7,306 Matching Annotations
  1. Sep 2020
    1. The Genesis stories provide two ethical alternatives, dominion and stewardship-both of which are anthropocentric. They do not explicitly acknowledge anthropocentric ethics, such as ecocentrism in which humanity is only one of a number of equal parts-an ecocentric ethic, nor is biocentrism a possibility, in which value is grounded in life itself, rather than being centered in humanity. But another form from of ethics is the partnership ethic I propose that posits nature and humanity as equal, interacting, mutually responsive partners. This ethic combines human actions and nature's actions in a dynamic relationship with each other. Here nature is not created specifically for human use, nor are women and animals seen as helpmates for "man." Rather, human life and biotic life exist in mutual support, reciprocity, and partnership with each other. Gardens could exemplify places in which the practice of gardening is a caretaking of the soul and the life it generates.

      There are two narratives about the origin, fall and reinvention of the Eden Garden, called Recovery Narratives. These two narratives have strongly influenced the relationship between human beings and nature. I think it's important to mention that both the Christian Narrative and the Environmentalist narrative seek to recover the lost, paradise, the Garden of Eden. However, the conceptions of what the garden (enclosure) is, and what you want to recover (relative to its extent) differs from one narrative to another. The Gilgamesh story we read the previous week may be related to the change of conception of ''recovering the lost.''

      Rooted in Genesis l, the Christian Narrative is the most influential rooted in Western culture. Is a traditional biblical narrative of the Fall from the Garden of the Eden, the guilt is charged to women and men is the agent to transform and savior with intention to redeem and recovered the garden by recreating the Eden in Earth with the possibility to exercise power and control over everything around them. Is the narrative that supports exploitation of nature to benefit human being and their cause, gender inequity.

      The environmentalist narrative follows what it is written in Genesis ll, God creates the man from dust, then The Eden Garden is made, followed by 4 rivers and trees for food, woman becomes partner of man. The landscape of this narrative is more abundant, fertile, rich. Human beings, animals and nature coexists in harmony and abundance. The Eden landscape is river based, spring-fed, everything to maintain abundance. The decline of the Eden is slow, and the way how male and female intend to recover it has an ecological vision. Nature is a victim of exploitation but also the main benefactor of the recovery of Eden.

      In summary Genesis l as ethical model is defined by human dominion, and Genesis ll as ethical model it's about stewardship.

      In this case Gilgamesh's story is a good example for relating the objective of the Christian Narrative as an ethical model. Or the human dominion over nature. Where the main benefactor is the human being, and the exploitation of the earth and what is obtained from it is merely for the satisfaction of ''humanity'' and ''civilization.'' Eden on earth is depicted in the ''walled kingdom of Gilgamesh'' and the ''clearing and appropriation of the forest'' which possesses the benefits given by nature, water, food, shelter, light, wealth.

      Andrew, George. The Epic of Gilgamesh, Penguin Group. 1999

    1. On page 4, the section that begins with "The mother of the Puetro Rican child..."particulary stood out to me as an adoptee myself. I found that my parents did this, reading all they could about Chinese adoptees, and reaching out to other parents. They tried to find the most information they could about the subject and flooded my sister and I with different articles, resources, Asian media, etc. as we grew older.

      This was intriguing to me because I think in this "Getting Ready" stage we have to understand that even when we try to find information about a certain group of people, not everyone in the group has the same experiences.

      Being someone who has experienced this from the point of view of the child in the situation given in this reading, I think although it is important to educate yourself on different minority/marginalized groups of people, there also must be an understanding that not all experiences are the same. As the person in the group, there also must be an understanding that there is good intent though.

      In my experience, I didn't relate fully with everything my parents would share on adoption or Chinese culture, and often felt a bit of "imposter syndrome," feeling as though I did not know enough to be considered a "true Asian," but also wondering if I indeed did fit in more with the white kids around me. My parents and those around me went through the getting ready and reaching out stages, but without fully understanding how my experiences with Asian culture or adoptee culture may be different from those they were reading about, sharing with me, or asking me about.

      I think that Harro makes good points about educating and seeking out exposure, however I think we need to think about this through an intersectional lens, and through the understanding not all people in a specific marginalized group have the same exposure/experiences.

    1. Note: This rebuttal was posted by the corresponding author to Review Commons. Content has not been altered except for formatting.

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      Reply to the reviewers

      Reviewer #1 (Evidence, reproducibility and clarity (Required)): The manuscript by Huh et al. reports that oxidative stress causes fragmentation of a specific tyrosine pre-tRNA, leading to two parallel outcomes. First, the fragmentation depletes the mature tRNA, causing translational repression of genes that are disproportionally rich in tyrosine codon. These genes are enriched for those involved in electron transport chain, cell cycle and growth. Second, the fragmentation generates tRNA fragments (tRFs) that bind to two known RNA binding proteins. Finally, the authors identify a nuclease that is needed for efficient formation of tyrosine tRFs. Comment 1: Th­­­­e authors should include a short diagram indicating the various known steps of pre-tRNA fragmentation (perhaps as a supplement) for general readers.

      Response: We thank the reviewer for their suggestion. Pre-tRNA fragmentation is still an unknown field but an initial introduction is best seen from pre-tRNA processing where there is a cleavage event for pre-tRNAs with an intron. This is a complex subject but a recent review from Hopper and Nostramo has done an excellent job in in describing the current field in yeast and vertebrate species (Hopper and Nostramo, Front. Genet., 2019). We have added this citation and new text in the manuscript about pre-tRNA processing for general readers to follow up on. We feel that a supplementary figure might be a bit too brief in describing the knowns and unknowns of pre-tRNA processing and fragmentation.

      Comment 2: I find the enrichment for mitochondrial electron transport chain (ETC) curious. The ETC includes several oxidoreductases, which may be rich in tyrosine as it is a common amino acid used in electron transfer. The depletion of the tyrosine tRNA from among many tRNAs under oxidative stress may not be incidental but related to an attempt by the cell to decrease oxygen consumption to avoid further oxidative damage. The authors could further mine their data to corroborate this hypothesis. For example, are the ETC genes among the targets of the RNA binding proteins targeted by tyrosine tRFs? This could potentially connect the effects of mature tRNA depletion and tRFs.

      Response: We thank the reviewer for this very interesting comment and insight, which had not occurred to us. The relationship between this response and oxidoreductase regulation could be a factor in both the tRNA and tRF modulations seen in our cells. Interestingly, we find that many oxidoreductases genes (such as the NDUF family) are bound by hnRNPA1 by CLIP. In new data, we have done stability experiments with the tRF (new Fig 7E-F) to show the regulon of hnRNPA1 is modulated with overexpression and LNA against the tRF, revealing that this tRNA fragmentation response modulates expression of certain oxidoreductase genes. However, we do not see clear and significant differences for ETC genes in particular. As hnRNPA1 is known to act as both a promoter and destabilizer of genes depending on context, it is likely that further and more detailed work will be needed to parse this hypothesis out in future studies.

      Comment 3: In figure 4A, the authors should provide the tyrosine codon content of the overlap genes and show how much it differs from a randomly selected sample.

      Response: We have identified an error in our manuscript where the overlap actually identifies 109 proteins rather than the 102 reported in the original manuscript. We apologize for this oversight. As for the overlap proteins, we plotted the downstream proteins detected in the proteome by mass spectrometry based off on Tyr-codon content. As explained in the text, the targets we tested were chosen for having higher than median levels of Tyr-codon, as seen in the histogram, and for showing some of the greatest reduction after Tyr tRNA-GUA depletion (Fig S4A). The other proteins found in the overlap will fall in a similar pattern along the histogram.

      Comment 4: Fig.6F, lower panel: the model should show pre-tRNA, as opposed to mature tRNA, because it is the former that is fragmented.

      Response: We apologize for the confusion. The model in Fig 7F was supposed to denote the pre-tRNA with the trailer and leader sequences intact initially, then lost with processing to mature tRNA. To make it clearer, we have now labeled the first species as “Pre-tRNA.”

      Reviewer #1 (Significance (Required)): This study is comprehensive and novel, and includes several orthogonal and complementary approaches to provide convincing evidence for the conclusions. The main discovery is significant because it presents an important advance in post-transcriptional control of gene expression. The process of tRF formation was previously thought not to affect the levels of mature tRNA. This study changes that understanding by describing for the first time the depletion of a specific mature tRNA as its precursor form is fragmented to generate tRFs. Finally, the authors identify DIS3L2 as a nuclease involved in fragmentation. This is also an important finding as the only other suspected nuclease, albeit with contradictory evidence, is angiogenin. Collectively, the findings of this study would be of interest to a broad group of scientists. I only have a few minor comments and suggestions (see above).

      Response: We thank the reviewer for their very positive and insightful comments and feedback.

      REFEREES CROSS-COMMENTING I have the following comments on other reviewers' critiques. Regarding the concern that the disappearance of the pre-tRNA could be a transcriptional response (reviewer 2), I think that the appearance of tRFs makes this scenario unlikely. If pre-tRNA levels decreased due to transcriptional repression, wouldn't one expect that both tRNA and the tRF levels diminish concomitantly? Reviewer 3 raises the issue of cross hybridization in Northern blots. The authors indicate that they "could not detect the other tyrosyl tRNA (tRNA Tyr AUA) in MCF10A cells by northern blot..." (page 6). Also, they gel extracted tRFs and sequenced them (figure S6B), directly identifying the fragments. I think these findings mitigate the concern of cross hybridization and clearly identify the nature of tRFs. Finally, I think that the codon-dependent reporter experiment (figure 5D) addresses many issues surrounding codon dependent vs indirect effects. In that experiment, the authors mutate 5 tyrosine codons of a reporter gene and demonstrate that the encoded protein is less susceptible to repression in response to oxidative stress.

      Response: We thank the reviewer for their tremendous insights. We are in agreement regarding the three points in the cross-comments.

      Reviewer #2 (Evidence, reproducibility and clarity (Required)): This very interesting study from Sohail Tavazoie's lab describes the consequences of oxidative stress on the tRNA pool in human epithelial cell lines. As previously described, the authors observed that tRNA fragments were generated upon exposure of cells to ROS. In addition, the authors made the novel observation that specific mature tRNAs were also depleted under these conditions. In particular, the authors focused on tyrosyl tRNA-GUA, which was decreased ~50% after 24 hours of ROS exposure, an effect attributable to a decrease in the pre-tRNA pool. Depletion of tyrosyl tRNA resulted in reduced translation of specific mRNAs that are enriched in tyr codons and likely contributed to the anti-proliferative effects of ROS exposure. In addition, the authors demonstrated that the tRFs produced from tyr tRNA-GUA can interact with specific RNA binding proteins (SSB and hnRNPA1). The major contribution of this paper is the novel finding that stress-induced tRNA fragmentation can result in a measurable reduction of specific mature tRNAs, leading to a selective reduction in translation of mRNAs that are enriched for the corresponding codons. Previously, studies of tRNA fragmentation largely focused on the functions of the tRFs themselves and it was generally believed that the mature tRNA pool was not impacted sufficiently to reduce translation. The findings reported here therefore add a new dimension to our understanding of the cellular consequences of stress-induced tRNA cleavage. Overall, the data are of high quality, the experiments are convincing, and the conclusions are well supported. I have the following suggestions that would further strengthen the study and bolster the conclusions. Comment 1: The authors have not formally demonstrated that the reduction in pre-tRNA in H2O2-treated cells is a consequence of pre-tRNA cleavage. It is possible that reduced transcription contributes to this effect. Pulse-chase experiments with nucleotides such as EU would provide a tractable approach to demonstrate that a labelled pool of pre-tRNA is rapidly depleted upon H2O2 treatment, which would further support their model. Since the response occurs rapidly (within 1 hour), it would be feasible to monitor the rate of pre-tRNA depletion during this time period in control vs. H2O2-treated cells.

      Response: We thank the reviewer for their suggestion and agree that testing for a transcriptional effect using a pulse-chase experiment would further support these findings. We are grateful to both reviewer 1 and reviewer 2 in the cross-comments for recognizing that the tRNA repression response we see is too rapid to be a transcriptional response and that the fact that this tRNA depletion response occurs concomitantly with the tRF generation supports our model that this is a pre-tRNA fragmentation response. It would be of interest for future studies to also examine the impact of cellular stress on tRNA transcription.

      Comment 2: To what extent is the growth arrest that results from H2O2 treatment attributable to tyr tRNA-GUA depletion (Fig. 3A)? Since the reduction in tRNA levels is only partial (~50%), it should be feasible to restore tRNA levels by overexpression (strategy used in Fig. 3E, S3B) and determine whether this measurably rescues growth in H2O2-treated cells.

      Response: We thank the reviewer for their suggestion. Originally, we had also thought of this experiment and attempted to test this hypothesis. Upon experimentation, we ran into technical challenges that prevented us from drawing any conclusions. The problems were that we were unable to develop a cell line that stably overexpressed the Tyr tRNA-GUA and had to settle for a transient overexpression that only lasted for a couple of days (Fig S3B). For transient transfection, we used Lipofectamine 3000 (Invitrogen) that has associated cell toxicities and requires a control RNA transfection in lipofectamine. In addition, H2O2 in itself is a stress. The simultaneous occurrence of these two stresses led to a combination of cell death and cell growth for the control and experimental group. Given the high variability, we were unable to draw any conclusions on cell growth with this combination. We hope to identify a way to stably overexpress Tyr tRNA-GUA in the future to address this hypothesis.

      Comment 3: Knockdown of YARS/tyr tRNA-GUA resulted in reduced expression of EPCAM, SCD, and USP3 at both the protein and mRNA levels (Fig. 4C-D, S4C). In contrast, H2O2-exposure reduced the abundance of these proteins without affecting mRNA levels (Fig. 5A-B, S5A). The authors should comment on this apparent discrepancy. Perhaps translational stalling induces No-Go decay, but it is unclear why this response would not also be triggered by ROS.

      Response: We would like to clarify that out of the three genes in Fig. S5A, only EPCAM mRNA levels were significantly reduced with H2O2-exposure while no changes were observed in the mRNA levels of USP3 or SCD. It is difficult to ascertain the reason for EPCAM mRNA reduction but one hypothesis is due to timing and steady state levels. Levels of mRNAs seen with knockdown of YARS or tRNA represent steady state levels where mRNA decay and transcriptional changes can be easily seen. Following H2O2, the data is collected at 24 hours, which may be before mRNA effects can be fully appreciated. We have edited the text to clarify the uncertainty involved. We agree with the reviewer’s insightful comment and find these differences to be interesting and will consider them in future studies to better understand the interplay between translation and mRNA levels in the context of tRNA depletion.

      Comment 4: In addition to the analyses of ribosome profiling in Fig. 5E-F, it might also be helpful to show a metagene analysis of ribosome occupancy centered upon UAC/UAU codons (for an example, see Figure 2 of Schuller et al., Mol Cell, 2017). This has previously been used as an effective way to visualize ribosome stalling at specific codons. Additionally, do the authors see a global correlation between tyrosine codon density and reduced translational efficiency in tRNA knockdown cells?

      Response: We thank the reviewer for their important suggestion. We have expanded the analysis to look at codon usage scatterplots across all codons for shTyr and shControl replicates (Fig S5D). The 5 most changed codons are labeled with UAC, a codon for the tyrosine amino acid, being the most affected (red arrow). Consistent with our model, a tyrosine codon, when at the ribosome A-site, is most affected with depletion of the corresponding tRNA. The text has also been edited to reflect our new analysis providing further evidence that ribosomal stalling could occur upon depletion of this tRNA. The gray outline around the regression line represents the 95% confidence interval.

      Fig S5D

      As seen in Fig 5F, a significant overlap was noted for genes with the lowest translational efficiency and tyrosine enrichment. We did further analysis to test if a direct and linear relationship exists between tyrosine codon density and reduced translational efficiency on the global scale (i.e. does more stalling occur with more tyrosine codons on a global scale). We again see that a reduced translational efficiency is significantly correlated with tyrosine codon enrichment (above median parameters) in the tRNA knockdown ribosome profiling data. However, our analysis on a direct relationship between codon density and translational efficiency is inconclusive. This analysis is limited given the sequencing depth and number of experimental replicates available and we lack the statistical power to draw strong conclusions. To prevent overstating our claims, we have omitted any conclusions regarding this second analysis.

      Comment 5: MINOR: On pg. 4, the authors state that tRF-tyrGUA is the most highly induced tRF, but Fig. S1B appears to show stronger induction of tRF-LeuTAA.

      Response: The reviewer is correct in that the data from Fig S1B shows Leu-tRFs with higher induction. Our text was meant to suggest we focused on tRF-TyrGUA due to higher band intensity seen on northern blot validation. We have edited the text in the manuscript to clarify this.

      Reviewer #2 (Significance (Required)): The major advance provided by this work is the demonstration that stress-induced tRNA cleavage can reduce the abundance of the mature tRNA pool sufficiently to impact translation. Moreover, the effect on mature tRNAs is selective, resulting in the reduced translation of a specific set of mRNAs under these conditions. These findings reveal previously unknown consequences of oxidative stress on gene expression and will be of interest to scientists working on cellular stress responses and post-transcriptional regulation.

      Response: We thank the reviewer for the kind comments and feedback.

      REFEREES CROSS-COMMENTING Regarding the concern that the disappearance of the pre-tRNA could be a transcriptional response (reviewer 2), I think that the appearance of tRFs makes this scenario unlikely. If pre-tRNA levels decreased due to transcriptional repression, wouldn't one expect that both tRNA and the tRF levels diminish concomitantly? Here is what I was thinking: The generation of tRFs does not generally result in reduction in levels of the mature tRNAs. So you can imagine a scenario where oxidative stress causes tRF generation from the mature tyr tRNA (which does not impact its steady-state levels), as is the case for other tRNAs. At the same time, decreased transcription would reduce the pre-tRNA pool, leading to a delayed reduction in mature tRNA, as observed. However, looking back at the data, I see that after only 5 min of H2O2 treatment, the authors observed reduced pre-tRNA and increased tRFs (Fig. 2A). This seems very fast for a transcriptional response, which would presumably require some kind of signal transduction. In addition, when you consider the amount of tRFs produced in Fig. S2C, it is hard to imagine that this would not impact the mature tRNA pool if they were derived from there. So I agree that the transcriptional scenario seems unlikely. Nevertheless, I think that looking at pre-tRNA degradation directly with the pulse-chase strategy would strengthen their story, so I would like to give the authors this suggestion. However, I am fine with listing this as an optional experiment which would enhance the paper but should not be essential for publication.

      Response: We thank the reviewer for these insightful comments. As mentioned above, five minutes is likely too rapid for a transcriptional response to be the main effect of H2O2 on Tyr-tRNA GUA. Moreover, the concomitant appearance of the tRF at this time-point makes tRNA fragmentation the most parsimonious and likely explanation rather than transcriptional repression, which would not cause a tRNA fragment to occur concurrently. Moreover, extraction and sequencing of the tRF shows it likely derives from the pre-tRNA as a 5’ leader sequence is present. We appreciate the reviewer’s suggestion and scholarly willingness to reassess their own hypothesis.

      Reviewer #3 (Evidence, reproducibility and clarity (Required)): The major findings in this manuscript are: 1.) Oxidative stress in human cells causes a decrease in tyrosine tRNA levels and accumulation of tyrosine tRNA fragments; 2.) The depletion of tyrosyl-tRNA synthetase or tyrosine tRNAs in human cells results in altered translation of certain genes and reduced cell growth and 3.) hnRNPA1 and SSB/La can bind tyrosine tRNA fragments. There is also preliminary evidence that the DIS3L2 endonuclease contributes to the appearance of tyrosine tRNA fragments upon oxidative stress. Based upon these results, the Authors conclude that tyrosine tRNA depletion is part of a conserved stress-response pathway to regulate translation in a codon-based manner. **Major comments:** Comment 1: There is a considerable amount of data in this paper and the experiments are performed in a generally rigorous manner. Sufficient details are provided for reproducing the findings and all results have been provided to appropriate databases (RNA-Seq and ribosome profiling).

      Response: We thank the reviewer for the positive comments and feedback.

      Comment 2: The manuscript uses a probe against the 5' half of Tyrosine tRNA for Northern blotting. However, tRNA probes can be prone to cross-hybridization, especially with some tRNA isoacceptors being similar in sequence. Thus, the blots in Figure 2 and Supplemental Figures should be probed with an oligonucleotide against the 3' half of tRNA-Tyr. This will confirm the pre- and mature tRNA-Tyr bands detected with the 5' probe. Moreover, this will determine whether 3' tRNA-Tyr fragments accumulate.

      Response: We agree that the reviewer is correct in suggesting that the 3’ tRNA-Tyr might also accumulate. However, we disagree that any accumulation of the 3’ tRF might be relevant in our particular model for multiple reasons. As supported by reviewer 1’s cross-comments, cross-hybridization between isoacceptors (GUA vs AUA) would be unlikely as Tyr-AUA could not even be detected by the initial 5’ tRF probe. Additionally, the sequences for Tyr-GUA are different with no nucleotide alignment from Tyr-AUA. Furthermore, the extraction and sequencing of the 5’ tRF (Fig S6B) confirms the 5’ leader sequence unique to the pre-tRNA (also noted by reviewer 1). While the 3’ half of many Tyr-GUA are similar, we find selective binding of our RNA binding proteins only to the 5’ tRF. The 3’ tRF may play some role in binding to other proteins in cell regulatory pathways but such experiments would be outside the scope of this study.

      Comment 3: The analysis of the proteomic and ribosome profiling experiments seem rather limited, or based upon what was presented in this manuscript. If additional analyses were performed, then they should be included as well, even if they yielded negative results. For example, the manuscript identifies 102 proteins that decrease after tRNA-Tyr depletion and YARS-depletion with a certain threshold of Tyr codon content. We realize the Authors were trying to find potential genes that are modulated under all three conditions. However, this does not provide information whether there is a relationship between a certain codon such as Tyr and protein abundance if only binning into two categories representing below and above a certain codon content. The Authors should plot the abundance change of each detected protein versus each codon and determine the correlation coefficient. This analysis is important for substantiating the conclusion of a codon-based system of specifically modulating transcripts enriched for certain codons. Otherwise, how could changes in tRNA-Tyr levels modulate codon-dependent gene expression if two different transcripts with the same Tyr codon content exhibit differences in translation? Moreover, this analysis should be performed with all the other codons as well.

      Response: We have identified an error in our manuscript where the overlap identified 109 proteins and not 102 as reported previously. We apologize for this oversight. While the reviewer is correct in that identifying codon dependent changes for all 3500+ proteins detected would offer greater insight, our study was specifically focused on tyrosine as we observed this tRNA to become depleted and our experimental system modulated this specific tRNA. As for the second point on Tyr tRNA level effects on translation, we felt that the most rigorous course would be to assess causality rather than an association for this tRNA and its codon in regulating a target gene. The only way to do this is to perform mutagenesis and reporter studies. Our codon dependent reporter clearly shows a direct effect on translation in a tyrosine-codon dependent manner. As for translational regulation for two different transcripts with the same Tyr codon content, it is unclear the molecular mechanisms that could dictate these differences. The reviewer has already brought up possibilities in the next comment regarding Tyr codons in 5’ or 3’ ends or consecutive Tyr codons. These are all interesting hypotheses that others in the field have devoted entire publications to try and understand how and why codon interactions and localizations impact translation (see Gamble et al., Cell 2016, Kunec and Osterreider, Cell Reports 2016, Gobet et al., PNAS 2020). While these further analyses would be interesting, our current experimental data would be insufficient to properly address these questions. We have focused on a specific tRNA, its fragment, and demonstrated direct effects of the tRNA on the codon-dependent translation of a specific growth-regulating target gene and the tRNA fragment on the modulation of the activity of the RNA binding protein it binds to with respect to its regulon. We believe that these findings individually reveal causal roles for this tRNA and tRF in downstream gene regulation and collectively reveal a previously unappreciated post-transcriptional response. We hope the reviewer agrees with us regarding the already deep extent of the studies and that further such analyses beyond this tRNA are outside the scope and focus of this current study.

      Comment 4: The Authors should provide the specific parameters used to calculate the median abundance of Tyr codons in a protein and the list of proteins containing higher than median abundance of Tyr codon content. Moreover, the complete list of 102 candidate genes should also be provided. This will allow one to determine what percentage of these Tyr-enriched proteins exhibited a decrease in levels. Moreover, is there anything special about these Tyr codon-enriched transcripts where they are affected at the level of translation but not the other Tyr-codon enriched transcripts? For example, are these transcripts enriched at the 5' or 3' ends for Tyr codons? Do these transcripts exhibit multiple consecutive Tyr codons? This deeper analysis would enrich the findings in this manuscript.

      Response: For the proteins identified in the mass spectrometry and overlap listed in Fig 4A, Tyr codon abundance was calculated by dividing the number of Tyr amino acids present by the total number of amino acids for each protein. For genes with different isoforms possible, the principal isoform, using ENSEMBL, was used for calculations. We are also happy to provide the entire list of proteins. Additionally, please see above response to comment 3. We wish to emphasize that the goal of identification of these proteins was to identify downstream targets of this response for functional studies, which we have done. We have identified downstream genes that become modulated by this response and that regulate cell growth, consistent with the phenotype of the tRNA. We then demonstrated a direct causal tRNA-dependent codon-based response with a specific target gene using mutagenesis.

      While we agree that the additional analysis the reviewer is requesting to determine what constitutes heightened translational sensitivity to this response is interesting, we believe this is a challenging question for future studies. It is possible that enrichment at 5’ or 3’ or concentration of tyrosine codons could cause increased sensitivity. Ideally, one would have information on a larger set of proteins so that such challenging questions could be better statistically bolstered. Ultimately, the requested experiments that go beyond our current work would require further analyses and experiments to allow firm conclusions to be drawn. As the other reviewers state and this reviewer agrees, we have uncovered the initial discovery regarding this tRNA fragmentation response and provided mechanistic characterization. Future studies, which are beyond the scope of the current work will undoubtedly further characterize features of this response.

      Comment 5: The ribosome profiling results are condensed into two panels of Figure 5E and 5F. We recommend the ribosome profiling experiment be expanded into its own figure with more extensive analysis and comparison beyond just looking at tRNA-Tyr. This could reveal insight into other codons that are impacted coordinately with Tyr codons and perhaps strengthen their conclusion. As an example of a more thorough analysis of ribosome profiling and proteomics, we point the Authors to this recent paper: Lyu et al. 2020 PLoS Genetics, https://journals.plos.org/plosgenetics/article?id=10.1371/journal.pgen.1008836

      Response: We thank the reviewer for their suggestion. We have expanded the analysis to look at codon usage scatterplots across all codons for shTyr and shControl replicates (Fig S5D). The 5 most changed codons are labeled with UAC, a codon for the tyrosine amino acid, being the most affected (red arrow). Consistent with our model, a tyrosine codon, when at the ribosome A-site, is most affected with depletion of the corresponding tRNA. The text has also been edited to reflect our new analysis providing further evidence that ribosomal stalling might occur with depletion of a given tRNA. The gray outline around the regression line represents the 95% confidence interval.

      Fig S5D

      Comment 6: Moreover, one would expect that the mRNAs encoding USP3, EPCAM and SCD would exhibit increased ribosome occupancy. Thus, the authors should at least provide relative ribosome occupancy information on these transcripts to provide evidence that the decrease in protein levels is indeed linked to ribosome pausing or stalling.

      Response: We would like to emphasize that resolution of ribosomal profiling data at the codon level for specific genes requires a high number of reads and replicates to draw accurate conclusions. There is an inherent level of stochasticity when mapping RPFs to specific genes and as a result, our analysis revolved around Tyr-enriched vs Tyr-low populations as this analysis was appropriate for our sequencing depth and number of replicates. To be able to conclusively make claims regarding ribosome pausing or stalling for specific genes, we would likely need further experimentation than can be currently done. However, we are currently conducting the requested bioinformatic analysis and have promising preliminary transcript-level data supporting our model.

      Comment 7: The results with hnRNPA1 and SSB/La are extremely preliminary and simply show binding of tRNA fragments but no biological relevance. We realize that the Authors attempted to see if Tyr-tRNA fragments impacted RNA Pol III RNA but found no effect. A potential experiment would be to perform HITS-CLIP on H2O2-treated cells to see if stress-induced tRNA fragments bind to SSB/La or hnRNPA1. In this case, at least the Authors would link the oxidative stress results found in Figure 1 and 2 with La/SSB and hnRNPA1.

      Response: We agree with the reviewer that a tRF function was not established in the manuscript. As a result, we have recently completed experiments looking at mRNA stability of the hnRNPA1 regulon in the context of overexpressing the tRF as well as using LNA to inhibit this Tyr-tRF (Fig 7E-F). Our data shows, in an hnRNPA1-dependent manner, that its regulon can be functionally regulated by Tyr-tRF. With tRF overexpression and RNAi-mediated depletion of hnRNPA1, a right shift in transcript stability is seen. Importantly, when we do the converse experiment with tRF inhibition in the same RNAi-mediated reduction of hnRNPA1, we see a left shift. These complementary experiments provide data that the Tyr-tRF has a functional role when bound to hnRNPA1 by modulating the regulon of hnRNPA1 and expand the scope of this manuscript and extend the pathway defined downstream of this tRNA fragmentation event.

      Fig 7E-F

      Comment 8: The manuscript concludes that "Tyrosyl tRNA-GUA fragments are generated in a DIS3L2-dependent manner" based upon data in Supplemental Figure S7. However, there is still a substantial amount of tyrosine tRNA fragments in both worms and human cells depleted of DIS3L2. Thus, DIS3L could play a role in the formation of Tyrosine tRNA fragments but it is too strong a claim to say that tRNA fragments are "dependent" upon DIS3L2. We suggest that the Authors soften their conclusions.

      Response: While there are certainly tRFs still apparent with DIS3L2 depletion (Fig S7F-I), we note significant impairment of tRF induction with DIS3L2 knockdown/knockout with multiple different methods in C. elegans and human cells. This data supports our conclusion that tRF generation is dependent on DIS3L2 as this ribonuclease is necessary to elicit the full Tyr-tRF response. We do not make claims that Tyr-tRFs are solely or completely dependent on DIS3L2. There must be other RNases involved given the data highlighted by the reviewer. To this point, we have added clarifying text that DIS3L2 depletion does not completely eliminate the tRF induction.

      Comment 9: Moreover, what is the level of DIS3L2 depletion in the worm and human cell lines? The Authors should provide the immunoblot of DIS3L2 that was described in the Materials and Methods.

      Response: An immunoblot of DIS3L2 depletion in human cells has now been added as a supplementary figure (Fig S7I). Depletion in C. elegans was confirmed through sequencing of a mutation, as is standard in the field. The wild-type PCR product is 1nt longer (859 bp) than the mutant product (858 bp) with CTC to TAG nonsynonymous mutation preceding a single nucleotide deletion.

      Wild-type disl-2: GTTGAAGCCGCAGGGC[CTC]ACTCAGACAGCTACAGG

      disl-2 (syb1033): GTTGAAGCCGCAGGGC[TAG]-CTCAGACAGCTACAGG

      Fig S7I

      Comment 10: The key conclusions of "a tRNA-regulated growth suppressive oxidative stress response pathway" and an "underlying adaptive codon-based gene regulatory logic inherent to the genetic code" are overstated. This is because of the major caveat that knockdown of tyrosine-tRNA or tyrosyl-tRNA synthetase are likely to trigger numerous indirect effects. While the authors validate that three proteins are expressed at lower levels under all three conditions (H2O2, tRNA-Tyr and YARS), they might overlap in some manner but not necessarily define a coordinated response. Thus, a glaring gap in this paper is a clear, mechanistic link between H2O2-induced changes in translation versus the changes in expression when either tRNA-Tyr or YARS is depleted. Thus, it is too preliminary to conclude that tRNA depletion is part of a "pathway" and "regulatory logic" when it could all be pleiotropic effects. At the very least, the authors should discuss the possibility of indirect effects to provide a more nuanced discussion of the results obtained using two different cell systems and oxidative stress.

      Response: We thank the reviewer for the feedback. While we agree that indirect effects may exist, we do not make any claims that our pathway is the only one required to have translation effects. The text for Fig 4A already acknowledges the pleiotropic effects of tRNA depletion. Our data shows that H2O2 stress leads to a depletion of Tyr tRNA-GUA and that depletion of this tRNA through multiple complementary methods has a codon-dependent effect on protein expression. We hope the reviewer agrees that the reduction of a specific target gene in a tyrosine codon-dependent manner (demonstrated by mutagenesis) and the binding of the tRF directly to an RBP and the modulation of the regulon of this RBP by this tRF (demonstrated by gain- and loss-of-function studies) demonstrates a direct role of this response on specific downstream target genes rather than pleiotropy. This is in keeping with the cross-comments of reviewer 1, where Fig 5D shows a direct Tyr codon link between H2O2 and downstream effects. As a result, we feel that our conclusions of a pathway (not the only pathway) are valid. However, the conclusion of a “regulatory logic” might not be interpreted in the same way by all readers and we have thus changed the text to reflect a more nuanced position.

      **Minor comments:** Comment 11: Tyrosyl-tRNAs refers to the aminoacylated form of tRNA. We recommend that all instances of tyrosyl-tRNA be changed to tyrosine tRNA or tRNA-Tyr which is more generic and provides no indication as to the aminoacylation status of a tRNA.

      Response: We thank the reviewer for their correction. We have changed all instances of “tyrosyl” to “tyrosine” in the text.

      Comment 12: In Figure 5C, the promoter is drawn as T7, which is a bacteriophage promoter. While the plasmid used in this manuscript (psiCHECK2) does contain a T7 promoter, mammalian gene expression is driven from the SV40 promoter. Thus, the relevant label in Figure 5C should be "SV40 promoter". Moreover, additional details should be provided on how the construct was made (such as sequence information etc.).

      Response: We thank the reviewer for their correction. We have changed the promoter text in the figure. In the methods for the construct, we have included which USP3 was used and would be happy to include further information if requested.

      Comment 13: Please provide original blots for each of the replicates in: Figure 4C, n=4 Figure 4A, n=9 Figure 4D, n=3 Figure 5D, n=3

      Response: There appears to be an unintentional mislabeling of the requested blots by the reviewer. The original blots for Fig 4C, Fig 5A, Fig 5D, and Fig 6D have been made available in a separate file for reviewers.

      Reviewer #3 (Significance (Required)): This manuscript provides evidence that specific tRNAs are depleted upon oxidative stress as part a conserved stress-response pathway in humans (and worms) to regulate translation in a codon-based manner. Unfortunately, the manuscript attempts to tie together results from different conditions and systems without providing any definitive links that suggest a "pathway" involved in the oxidative stress response. The findings in this paper provide a useful starting point but fall short of being a major advance due to the lack of a clear mechanism. However, there are intriguing results in this manuscript based upon the cell lines depleted of tRNA-Tyr or tyrosine synthetase that could interest researchers in the field of tRNA biology.

      Response: We thank the reviewer for the positive comments regarding our demonstration of a conserved stress response, acknowledging the intriguing nature of our findings that will be a starting point for future studies and that our work will be of interest to researchers in the field of tRNA biology. We hope that the very positive comments of reviewer 1 and 2, the cross-comments of reviewer 1 in response to reviewer 3’s comments regarding the specificity of this response, and our inclusion for reviewer 3 of additional data on the function of the tRF in regulating the activity of the hnRNPA1 RNA binding protein defining a post-transcriptional pathway and additional corroborating requested codon-level computational analyses provide compelling support that that our findings indeed represent a major advance for the field.

    1. Disabled people are also frequently excluded from dominant sexual scripts in two ways. They are commonly desexualized, in the sense that we tend to think of dis-abled people as not having sexual needs, experiencing sex-ual attraction, or having sex.

      I agree with this statement even if it maybe seem like disable people may not be talked about in sexual examples ,they still often have sexual desires and needs like other people. This argument shows how you can not exclude people just because you may not think of them as interested in sexual desire based on appearance and mentality’s.

    1. The Internet may cause our minds to wander off, and yet a quick look at the history of books suggests that we have been wandering off all along.

      This section of the reading strikes me as rather interesting. A lot of times we hear from people, especially the older generation that technology is such a nuisance. In reality, I don't think we as humans have ever been able to fully devote our focus on one thing or another, regardless of technology or not. So while the Internet is definitely much more prominent these days, and offers so much to do, the concept of getting distracted is not technology's fault on its own.

    1. Author Response

      Reviewer #1:

      This study was designed to determine whether there is a relationship among cranial suture closure patterns, the molecular causes for suture patency/closure, and phylogeny. The authors use correlative data to test causal hypotheses related to brain size, suture closure patterns, and diet and search for the genetic underpinnings of the relationships they identify using reference genomes. There are many ideas put forward and methods used that are not clearly explained in the body of the work or in the supplementary material. This made it difficult to provide a clear evaluation of the work. Even checking original sources on which they base their approach, I found some disconnect between original sources and ideas laid out here. I see some interesting ideas in the study but a lack of solid reasoning behind the hypotheses proposed, confusion about the data and/or ideas summarized from the literature (the confusion could be on my part, but it rests with the authors to explain this more fully), and lack of detail regarding methods used to support their conclusions.

      We take good note of this confusion and we will explain everything in more detail in a revised version of the manuscript.

      1) The entire study rests on the authors scoring of sutures as patent or closed but no information is given other than a suture was considered closed if it was not visible ( 'obliterated"), and a suture was considered open if visible. These are problematic definitions for distinguishing patent from closed sutures if we accept the authors' definition of sutures as growth and stress diffusion sites. A suture can be visible but still be "closed" as evidenced by bony connections or bridges linking the bones that border the suture. In the case of bridging, the suture would be visible, so would be scored as "open" according to the authors' criterion, but functionally, the suture is closed.

      Visual examination of sutures (e.g., from photos or in situ) is a common procedure in macroevolutionary studies of suture patency, where raw data is not always available for histological inspection (e.g., invasive procedures or CT are not permitted). In this regard, we follow previous literature. We would like to note that only photographic materials were available for most specimens during this project, because of the current exceptional circumstances (museums lockdown).

      Also, in some mammals (e.g., the laboratory mouse) most cranial sutures do not close in typically developing individuals.

      In this study we used specimens hosted in museum collections, which come from the wild or zoos. We did not use data from laboratory animals grown in controlled environments, which may indeed affect their suture patency (e.g., by feeding on pellets).

      2) Age estimates are not provided for the specimens used in analysis. In many mammalian species, suture closure occurs in a somewhat predictable fashion - this, coupled with tooth formation/eruption patterns is one of the ways that forensic scientists aged skeletal remains prior to the advent of modern technologies. The order of suture closure is not necessarily similar across vertebrates, or even across mammals. This means that, without known or estimated ages for each skull included in analysis, age becomes an unrecognized source of variation that will affect analytical outcome.

      Unfortunately, the exact age for museum specimens is often not available. For this reason, we focused on adult specimens, where suture patency tends to remain constant. We also excluded individuals with signs of senescence. To accommodate age and other source of intraspecific variation in adults, we collected information for as many individuals as possible, often more than 10 and sometimes up to 100. Thus, we coded suture patency as a frequency rather than as open/closed for each species.

      We only dichotomized suture patency as open/closed for the second part of the study. Here we used a sensible threshold to avoid ambiguity and be conservative. As a result, species with frequency of suture patency between 75% and 25% were excluded. This also means that if only 4 individuals were examined (small sample size was unavoidable for some rare species) and at least one showed a discrepancy, that species was excluded from the analysis. However, because suture patency is a very conserved trait, only a few taxa had to be excluded at the end.

      In any case, we will emphasize more this fact in the revised version.

      3) The authors' impact statement: "brain growth and skull ossification sequence cause suture closure in mammals evolution without common genetic factors causing premature suture closure diseases in humans" is hard to digest as brain growth is not considered by the authors but instead brain size. From a developmental perspective, brain size or even some form of the encephalization quotient (EQ) is not what is commonly proposed to drive suture closure/patency (or degree of patency). Instead it is the dynamics of brain growth that is proposed as a stimulus for the initiation of mineralization of cranial bones. As bones increase in size, new bone is added at the leading edge of opposing bones that line the suture, while the stem cells in the center of the suture remain to add to the mesenchymal cell population of the suture, keeping the suture patent. In short, the dynamics of brain growth (including any signaling emanating from the brain, dura, bones, or even the suture itself) contributes to suture patency. Because sutures tend to close later in life (after childhood in humans), normal suture closure appears to be associated with the termination of brain growth. Making the jump in their study from estimates of EQ (in some way estimated here) to dynamics of brain growth as a cause requires several steps and knowledge on timing and rate of growth that is not considered by the authors.

      We agree with the reviewer. A developmentally focused study on suture formation and closure dynamics must consider brain growth. However, this information is not available for most species selected for this study. Note that species selection depended on the availability of referenced genomes and multiple sequence alignments (some of which are rare, endangered species). Because we were comparing macroevolutionary dynamics in adults we decided to use brain size as a feasible proxy for brain influence (either due to growth or signalling). We aim to fill this gap in future research projects. In the meantime, we will revise the wording of the article to make sure that there are no misleading statements about brain growth influence.

      4) The authors assume a suture closure pattern across the skull that starts at the anterior (rostrally) and move posteriorly (caudally) and builds this into their model. This seems to be based on a work by Koyabu et al. (2014), but that study is about the appearance of ossification centers for bones (not suture formation or closure) and the study actually clumps the frontal and parietal into the same group in their final analysis so why this supports and anterior to posterior direction of suture closure is not clear.

      Note that we did not “assume” any closure pattern; we interpreted the published evidence on how the skull ossifies in mammals to make a plausible hypothesis. We also tested other 11 plausible hypotheses. It could have happened that such hypothesis was worse than the others, but we found that the best supported hypothesis includes an anterior-posterior relation of suture closure. We will try to explain the construction of our model and hypothesis testing better in the revised version.

      5) The authors conclusion: (Lines 289-292 does not follow from their analyses.) Brain growth was not analyzed. I am uncertain what they mean by suture self-regulation as I don't think their detection of genetic variants in common across a diverse set of species means that those are controlling suture patency/closure.

      The proposed idea of suture self-regulation refers to the fact that one suture closure may affect another suture closure (as theoretical models previously suggested), and it is not necessarily related to the genetic variants identified here. As explained before, we will revise any reference to brain growth.

      Reviewer #2:

      -Authors tested 4 hypotheses (page 5, lines 78-84), but rejected or questioned them later on (which is a fair approach to be realistic and point out possible weaknesses or methodological limitations, nevertheless, I find there are more questions or suggestions rather than actual answers).

      We have tried to offer an open and clear set of hypotheses, tested them with the available data, and discussed the results fairly. As it is often the case in science, research may bring more questions than answers; we do not see this as a weakness. Our answers are also contextualized within the limitations that we described in the methods. We believe this is the correct way of doing science: even if this forces us to reject all our hypotheses, negative results are also results. Since our object of study is not very well known, we hope this study can fuel more research.

      -Lots of repeating text

      -Frequent missing references for major statements, unclear formulations

      We will double-check our manuscript. However, the reviewer offers no details about what is repeated or missing.

      -Few contradicting or unclear information, for instance, "high conservation..enabled us to categorize phenotype as either open or closed" / "suture patency ranging from 0-1, only above 75% and below 25% was counted as open or closed" / authors involved species were >2 samples were available but excluded any ambiguous case (small number of samples per species?)

      As explained before, thresholding at 25/75 % was used to binarize species as having a suture open or closed. This binarization is only used for the convergent amino acid substation analysis. We excluded ambiguous cases (i.e., a suture half closed) prior to data collection. We will explain it better in the revised version to avoid confusion.

      -"Phylogenetic path analysis showed almost no effect of diet on the brain size; low to medium (what does that mean then?) effect of brain on suture closure and medium to high effect of 1 suture affecting the other sutures in AP direction" (in many species this is described-the timeline of suture closure)

      Not sure about what the reviewer means; we will revise these sentences to make them clearer to readers.

      -I am not able to evaluate if the assessment of diet hardness as an equivalent to mechanical forces in the skull is correct and hope other reviewers will be able to do that-in fact, also to evaluate the phylogenetic path analysis performed in this manuscript. Authors took information on % of nektar/soft-plants and invertebrates/hard food (seeds etc) that given species consumes and multiplied by an index but not an actual modeling or assessment of the forces... To a laymen it looks like, for instance, cow chewing all day long relatively soft grass, building very strong muscles will at the end develop much more force/tension within the skull than an animal cracking one nut.

      As the reviewer correctly points out, chewing grass all day long is harder than cracking one nut (cracking nuts “all day long” would be another issue). In any case, we have weighed each food item compared to others (e.g., grass is weighed as twice as hard as meat) and there is consensus that feeding on seeds and scavenging is one of the most biomechanically demanding feeding strategies. In addition, we would like to note that we critically discussed the caveats of diet hardness as a proxy for the effect of feeding biomechanics on sutures, and we did not blindly assume this as a hard truth.

      -Lots of attention is given to the three identified genes with convergent amino acid substitution despite the fact that none of these genes have ever been related to any aspect of craniofacial biology, nor to the suture pathological conditions.

      We discussed the three genes that our analysis revealed. We cannot discuss genes for which we found no support. For these three genes, we offered plausible scenarios for how they could be associated to craniosynostosis; it is for future studies to explore these scenarios and validate experimentally or clinically these genes. The fact that they are not currently known as part of pathological conditions does not preclude that we need to discuss them in the manuscript. Every year, new genetic variants are discovered to be associated with craniosynostosis. The lack of correspondence between these genes and pathology is in fact one of the findings of this study: the few genes that show convergent mutations are not associated to pathology. We agree that absence of evidence is not evidence of absence. However, we also think that this is a result to be discussed in this manuscript and for the readers to ponder.

    1. And you children that are unconverted, don’t you know that you are going down to hell, to bear the dreadful wrath of that God that is now angry with you every day, and every night? Will you be content to be the children of the devil, when so many other children in the land are converted, and are become the holy and happy children of the King of kings?

      I understand what Johnathan Edwards was attempting to do with this sermon. He was sharing the hard truths about the wrath of God so that people would leave their life a sin. However, I do not agree with this statement he made towards children. Maybe it's just me, but I don't think children should be "scared" into believing in God. We are all born sinners and it is up to the parents to teach their children how to live according to their beliefs and religions. Furthermore, a lot of people didn't practice Christianity then as there are different religions and belief systems as it is today. So if there were people present at the revival who didn't believe in God or practiced Christianity, then a speech like this could possibly scare them away from a relationship with God. I believe a sermon of this manner is for Christians who may have fell off and needed to be reminded of what could happen if they didn't correct their sinful ways. Overall, it was a good sermon that can be preached today.

    2. he looks upon you as worthy of nothing else, but to be cast into the fire

      ……. All saints are sinners.... According to Oscar Wilde, “The only difference between the saint and the sinner is that every saint has a past, and every sinner has a future.” Because of this, I think Jonathan Edwards Great Awakening Revival and the sermons delivered by George Whitefield are too judgmental. Even the so-called man of God is found in a circle of sinners sometimes, and they committed an atrocity, yet people look at them as a man of God. The sermon seems intimidating, and many people may end up committing suicide instead of going to hell and cast into a fire. God knows we are all sinners, and God still gives us abundant grace to changes from our iniquity and walk in his way!

    3. We know not who they are, or in what seats they sit, or what thoughts they now have: it may be they are now at ease, and hear all these things without much disturbance, and are now flattering themselves that they are not the persons, promising themselves that they shall escape. If we knew that there was one person, and but one, in the whole congregation that was to be the subject of this misery, what an awful thing would it be to think of! If we knew who it was, what an awful sight would it be to see such a person!

      Planting seeds of doubt in their personal salvation, after terrorizing them with his description of the pits of hell was brilliant on his part. I can imagine them all looking at each other as he delivered this part of the sermon wondering which one of them was going to hell.

    1. Likewise, Americans with a lot of trust in national news organizations have an easier time separating factual from opinion statements than those with less trust.

      In the first chapter of the textbook for the year, we read about why we have statistics, and a big reason for that was because we "can't always trust our gut". I think that this snippet is interesting because I would think that the less you trusted news outlets, the more vigilant and unbiased you would be in disseminating fact and opinion. However, this data seems to project to me that those who don't trust national news outlets say so because they don't trust a particular outlet(s), which may skew their decision-making.

    1. The general life of society cannot extend its sway without juridical life extending its sway at the same time and indirect relation. We can thus be certain of finding reflected in law all the essential varieties of social solidarity.The objection may be raised, it is true, that social relations can fix themselves without assuming a juridical for

      I think this is a factual statment as regardless of how the general population feels or acts with in society no major social/societal groth will truly be seen until the ruling goverment implements these changes into law.

    1. We can teach them about the influence of their votes on such issues as educational funding, employment, civil rights, andthe overall political climate. Asa result, they may beencouraged to understand their valueswithout being swayed bycampaignplatforms, one-sided messages, or biased media coverage.

      The tone throughout the piece seems to bounce around between different roles for faculty -- as one responsible for imparting knowledge or otherwise changing students, to mentor or guide supporting student's path towards self-discovery. In the first model, faculty have power and responsibility. In the second model, students hold power and responsibility. Which model is more aligned with your thinking for this work? Which do you think would be more accessible to faculty and more valuable for students?

    1. ce. On the other hand, there is no such thing as "Native American litera- ture," though it may yet, someday, come into be

      I think we definitely have to search harder for Native American Literature than World Lit, British Lit or American Lit, but I wouldn't go so far as to say it doesn't exist (maybe it only exists under American Lit, and not it's own)

    1. his term includes teaching practices thatBOX 6.1Literacy: Then and NowColonists were literate enough if they could sign their name, or even an X, ondeeds. When immigrants arrived in large numbers in the 1800s, educators urgedschools to deliver “recitation literacy” to the foreign children who filled the school-rooms. That literacy was the ability to hold a book and reel off memorized portionsof basic American texts such as the opening paragraph of the Declaration of Inde-pendence, a part of the Gettysburg address, or some Bryant or Longfellow. Withthe coming of World War I, and the prospect of large numbers of men handlingnew equipment in foreign countries, Army testers redefined reading. Suddenly, tothe dismay of men used to reading familiar passages, passing the army readingtest meant being able to make sense, on the spot, of never-before-seen text. Cur-rently, that kind of “extraction literacy,” revolutionary in 1914, looks meager. Find-ing out who, what, when, where or how simply does not yield the inferences,questions, or ideas we now think of as defining full or “higher literacy.” The ideaof a classroom where young women, poor and minority students, and learningdisabled students all read (not recite) and write about (not copy) Shakespeare orSteinbeck is a radical and hopeful departure from the long-running conception ofliteracy as serviceable skills for the many and generative, reflective reading andwriting for the few (Wolf, 1988:1).How People Learn: Brain, Mind, Experience, and School: Expanded EditionCopyright National Academy of Sciences. All rights reserved.How People Learn: Brain, Mind, Experience, and School: Expanded EditionCopyright National Academy of Sciences. All rights reserved.

      Why is the word "culturally" used? Why not "life" relevant? Since most of a child's experience may not be consider culture but real world experience with families, friends and neighbors. The above diagram seems to point toward the importance of community that might be very diverse instead of some monolithic cultural experience.

  2. Aug 2020
    1. “Joaquin’s Dilemma”

      Understanding the link between racial identity and school-related behaviors

      by Pedro Antonio Noguera Cambridge, Massachusetts

      Introduction

      When I am asked to speak or write about the relationship between racial identity and academic performance, I often tell the story of my eldest son, Joaquin. Joaquin did extremely well throughout most of his early schooling. He was an excellent athlete (participating in soccer, basketball, and wrestling), played piano and percussion, and did very well in his classes. My wife and I never heard any complaints about him. In fact, we heard nothing but praise about his behavior from teachers, who referred to him as “courteous,” “respectful,” and “a leader among his peers.” Then suddenly, in the tenth grade, Joaquin’s grades took a nosedive. He failed math and science, and for the first time he started getting into trouble at school. At home he was often angry and irritable for no particular reason.

      My wife and I were left asking ourselves, "What's going on with our son? What’s behind this sudden change in behavior?" Despite my disappointment and growing frustration, I tried not to allow his behavior to drive us apart. I started spending more time with him and I started listening more intently to what he had to tell me about school and his friends. As I did, several things became very clear to me. One was that all of the friends he had grown up with in our neighborhood in South Berkeley (one of the poorest areas of the city) were dropping out of school. These were mostly Black, working-class kids who didn't have a lot of support at home or at school and were experiencing academic failure. Even though Joaquin came from a middle-class home with two supportive parents, most of his reference group-that is, the students he was closest to and identified with --- did not. The other thing that was changing for Joaquin was his sense of how he had to present himself when he was out on the streets and in school. As he grew older, Joaquin felt the need to project the image of a tough and angry young Black man. He believed that in order to be respected he had to carry yourself in a manner that was intimidating and even menacing. To behave differently –- too nice, gentle, kind, or sincere -- meant that he would be vulnerable and preyed upon. I learned that for Joaquin, part of his new persona also involved placing less value on academics, and greater emphasis on being cool and hanging out with the right people.

      By eleventh grade, Joaquin gradually started working out of these behaviors, and by twelfth grade, he seemed to snap out of his angry state. He became closer to his family, his grades improved, he rejoined the soccer team, he resumed playing piano, and he even started producing music. As I reflected on the two years of anger and self-destructiveness that he went through I came to the conclusion that Joaquin was trying desperately to figure out what it meant to be a young Black man. As I reflect on that period I realize that like many Black male adolescents, Joaquin was trapped by stereotypes, and they were pulling him down. During this difficult period it was very hard for me to help him through this process of identity formation. While he was in the midst of it the only thing I could do was talk to him, listen to him, and try to let him know what it was like for me when I went through adolescence.

      As a high school student, I had coped with the isolation that came from being one of the few students of color in my advanced classes by working extra hard to prove that I could do as well as or better than my White peers. However, outside of the classroom I also worked hard to prove to my less studious friends that I was cool or “down” as we would say. For me this meant playing basketball, hanging out, fighting when necessary, and acting like “one of the guys.” I felt forced to adopt a split personality: I behaved one way in class, another way with my friends, and yet another way at home.

      The Emerging Awareness of Race

      Adolescence is typically a period when young people become more detached from their parents and attempt to establish an independent identity. For racial minorities, adolescence is also a period when young people begin to solidify their understanding of their racial identities. For many, understanding the significance of race means recognizing that membership within a racial category requires certain social and political commitments. Adolescence is often a difficult and painful period for many young people. However for young people struggling to figure out the meaning and significance of their racial identities, the experience can be even more difficult.

      Awareness of race and the significance of racial difference often begin in early childhood. We know from psychological research that the development of racial identity is very context-dependent, especially in the early years. Children who attend racially diverse schools or reside in racially diverse communities are much more likely to become aware of race at an earlier age than children in more homogenous (1) settings. In the latter context, race is often not a defining issue nor is it a primary basis for identity formation. When children see their race as the norm they are less likely to perceive characteristics associated with it (i.e. physical appearance) as markers of inferiority.

      In contrast, children who grow up in more integrated settings become aware of physical differences fairly early. Interacting with children from other racial and ethnic backgrounds in a society that has historically treated race as a means of distinguishing groups and individuals often forces young people to develop racial identities early. However, prior to adolescence they still do not usually understand the political and social significance associated with differences in appearance. For young children, being a person with different skin color may be no more significant than being thin or heavy, tall or short. Differences in skin color, hair texture, and facial features are simply seen as being among the many differences that all children have. In environments where racist and ethnocentric behavior is common, children may learn fairly early that racist speech is hurtful.(2) They may know that calling someone a nigger is worse than calling them stupid, but they may not necessarily understand the meaning of such words or know why their use inflicts hurt upon others.

      Four years ago I was conducting research with colleagues at an elementary school in East Oakland. We were interested in understanding how the practice of separating children on the basis of the language differences affected their social relationships and perceptions of students from other groups. As is true in many parts of California, East Oakland was experiencing a major demographic change as large numbers of Mexican and Central American immigrants were moving into communities that had previously been predominantly African Americans. As is often the case, schools in East Oakland serve as the place where children from these groups encounter one another, and at several of the high schools there had been a significant increase in inter-racial conflict. (3)

      In the elementary school where we did our research we found that most of the Black and Latino students had very little interaction with each other. Although they attended the same school, the students had been placed in separate classes, ostensibly for the purpose of serving their language needs. From our interviews with students we learned that even very young children viewed peers from the other racial group with suspicion and animosity, even though they could not explain why. Interestingly, when we asked the students why they thought they had been placed in separate classrooms, most thought it was to prevent them from fighting. We also found that the younger Mexican students (between ages five and eight) saw themselves as White, and the Black students also referred to the Mexican students as White. However, as the children entered early adolescence (age nine or ten), the Mexican youth began to realize that they were not considered White outside of this setting, and they began to understand for the first time that being Mexican meant something very different than being White.

      Depending on the context, it is not uncommon for minority children to express a desire to reject group membership based on skin color especially during early adolescence. As they start to realize that in this society to be Black or Brown means to be seen as “less than” -- whether it be less smart, less capable, or less attractive -- they will often express a desire to be associated with the dominant and more powerful group. This tendency was evident among some of the younger Mexican students in our study. However, as they grew older, the political reality of life in East Oakland served to reinforce their understanding that they were definitely not White. As one student told us “White kids go to nice schools with swimming pools and grass, not a ghetto school like we go to.”

      In adolescence, awareness of race and its implications for individual identity become even more salient. For many young men and women of color, racial identity development is affected by some of the same factors that influence individual identity development in general. According to Erikson and other theorists of child development, as children enter adolescence, they become extremely conscious of their peers and seek out acceptance from their reference group.(4) As they become increasingly aware of themselves as social beings their perception of self tends to be highly dependent on acceptance and affirmation by others. For some adolescents, identification with and attachment to peer groups sometimes takes on so much importance that it can override other attachments to family, parents, and teachers.

      For adolescents in racially integrated schools, racial and ethnic identity also frequently take on new significance with respect to friendship groups and dating. It is not uncommon in integrated settings for pre-adolescent children to interact and form friendships easily across racial boundaries -- if their parents or other adults allow them to do so.(5) However, as young people enter adolescence, such transgressions of racial boundaries can become more problematic. As they become increasingly aware of the significance associated with group differences, they generally become more concerned with how their peers will react to their participation in interracial relationships and they may begin to self-segregate. As they get older, young people also become more aware of the politics associated with race. They become more cognizant of racial hierarchies and prejudice, even if they cannot articulate the political significance of race. They can feel its significance, but they often cannot explain what it all means.

      For the past three years I have been working closely with fifteen racially integrated school districts in the Minority Student Achievement Network (MSAN). At the racially integrated high schools in MSAN, students often become much more aware that racial group membership comes with certain political commitments and social expectations. In these schools, high-achieving students of color (like my son Joaquin) are sometimes unwilling to enroll in advanced placement courses or engage in activities that have traditionally been associated with White students because they fear becoming estranged from their friends. If they appear to engage in behavior that violates racial norms, they may be seen as rejecting membership in their racial group and run the risk of being regarded as a race traitor. For this reason, I have urged the districts in MSAN not to rely upon the initiative of students to break down racial barriers but to put the onus on school leaders to take steps that will make this border crossing easier and more likely. (6)

      Theories of the identity/achievement connection

      For educators, understanding the process through which young people come to see themselves as belonging to particular racial categories is important because it has tremendous bearing upon the so-called “achievement gap.” Throughout the United States, schools are characterized by increasing racial segregation (7) and widespread racial disparities in academic achievement. (8) Blatant inequities in funding, quality, and organization are also characteristic of the American educational system. Despite overwhelming evidence of a strong correlation between race and academic performance, there is considerable confusion among researchers about how and why such a correlation exists.

      The scholars whose work has had the greatest influence on these issues are John Ogbu and Signithia Fordham, both of whom have argued that Black students from all socioeconomic backgrounds develop “oppositional identities” that lead them to view schooling as a form of forced assimilation to White cultural values. (9) Ogbu and Fordham argue that Black students and other “non-voluntary minorities” (e.g., Chicanos, Puerto Ricans, Native Americans and others whose groups that have been dominated by White European culture) come to equate academic success with "acting White." For these researchers, such perceptions lead to the de-valuation of academic pursuits and the adoption of self-defeating behaviors that inhibit possibilities for academic success. In this framework, the few students who aspire to achieve academically must pay a heavy price for success. Black students who perform at high levels may be ostracized by their peers as traitors and “sell outs” and may be forced to choose between maintaining ties with their peers or achieving success in school. (10) This would explain why middle class minority student like my son Joaquin would under-perform academically despite their social and economic advantages.

      My own research challenges Ogbu and Fordham’s “acting white” thesis. While carrying out research among high school students in Northern California, I discovered that some high achieving minority students are ostracized by their peers, but others (like me) learn how to succeed in both worlds by adopting multiple identities. Still others actively and deliberately challenge racial stereotypes and seek to re-define their racial identities by showing that it is possible to do well in school and be proud of who they are.

      Claude Steele’s work on the effects of racial stereotypes on academic performance helps to provide a compelling explanation for the identity-achievement paradox. Through his research on student attitudes toward testing, Steele (twin brother of the more conservative Shelby) has shown that students are highly susceptible to prevailing stereotypes related to intellectual ability. (11) According to Steele, when “stereotypes threats” are operative, they lower the confidence of vulnerable students and negatively affect their performance on standardized tests. Steele writes: "Ironically, their susceptibility to this threat derives not from internal doubts about their ability but from their identification with the domain and the resulting concern they have about being stereotyped in it.” (12) According to Steele, the debilitating effects of stereotypes can extend beyond particular episodes of testing, and can have an effect on overall academic performance.

      Race in the School Context

      Stereotypes and Expectations As Steele’s research illustrates, in the United States we have very deeply embedded stereotypes that connect racial identity to academic ability, and children become aware of these stereotypes as they grow up in the school context. Simply put, there are often strong assumptions made in schools that if you’re White you’ll do better in school than if you’re Black; or, if you’re Asian you’ll do better at school than if you’re Latino. These kinds of stereotypes affect both teachers’ expectations of students and students’ expectations of themselves.

      One of the groups most affected by these stereotypes is Asian-Americans. There is a perception in many schools that Asians are “naturally” academically gifted – especially in math. This stereotype is based on the following notions: 1) that Asians are inherently smart (either for genetic or cultural reasons); 2) that they have a strong work ethic; 3) that they are passive and deferential toward authority, and; 4) that unlike other minorities they don't complain about discrimination. These perceptions make up what is often called the “model minority” stereotype. (13)

      One of my former students, Julian Ledesma, now a researcher at the Office of the President of the University of California, has been doing research on the model minority stereotype at a high school in Oakland, California. He started his work by interviewing various teachers and students about who they believed were “the smartest kids." In nearly every case, those he asked reported that the Asians were the “smartest” students. Even Asian students who were doing poorly in school reported that Asians were the smartest. The surprising thing about their responses to this question is that the average grade point average for Asians at the school was a 1.8.

      One reason for the gross misconception at this school is that Asians were over-represented in the honors courses and among students with the highest ranks in their class. Yet, these successful students were not representative of Asians as a whole at the school. Overall, Asian students were dropping out in high numbers and not doing very well academically. The school where Julian did his research also had a considerable gang problem among Asians. Yet, because the stereotype is so powerful, students and teachers at the school were more likely to regard the majority of Asian students as the exceptions, and the smaller numbers who were successful as the norm.

      The stereotypical images we hold toward groups are powerful in influencing what people see and expect of students. Unless educators consciously try to undermine and work against these kinds of stereotypes, they often act upon them unconsciously. Our assumptions related to race are so deeply entrenched that it is virtually impossible for us not to hold them unless we take conscious and deliberate action.

      Sorting practices and “normal” racial separation Beyond these stereotypes, there are also the sorting practices that go on in schools that send important messages to students about the meaning of racial categories. For example, in many schools students the remedial classes are disproportionately Black and Brown, and students often draw conclusions about the relationship between race and academic ability based on these patterns. They might say to themselves, “Well, I guess the kids in these ‘slow’ classes are less smart than those other kids who are in the honors classes.” They also notice that the students who are most likely to be punished, suspended and expelled, also are more likely to be the darker students.

      In addition to reinforcing stereotypes, grouping practices, which teachers and administrators often say are not based on race but on ability or behavior, often have the effect of reinforcing racial separation. Unless the adults in a school are conscious of how this separation influences their own perceptions and that of students, over time this separation may be regarded as normal. For example, Black students may assume that because there are no Black students in advanced or honors courses that they cannot excel academically. Of course, Black students can distinguish themselves in sports because there are numerous examples of Black individuals that do. Similarly, White students may assume that they should not seek academic assistance from tutorial programs, especially if those programs primarily serve Black or Brown students. When the norms associated with race take on a static and determining quality they can be very difficult to undermine.

      Students who receive a lot of support and encouragement at home may be more likely to cross over and work against these separations. But, as my wife and I found for a time with Joaquin, middle class African-American parents who try to encourage their kids to excel in school often find this can’t be done because the peer pressures against crossing these boundaries are too great.

      The racial separation we see in schools might be also be seen as an element of the “hidden curriculum,” an unspoken set of rules that “teaches” certain students what they can and cannot do because of who they are. There are aspects of this hidden curriculum that are not being taught by the adults. It may well be that students are the ones teaching it to each other. No adult goes onto the playground and says, "I don't want the boys and girls to play together." The girls and boys do that themselves, and it's a rare child who crosses over. Why? Because those who violate gender norms are often ostracized by their peers. The girls who play with the boys become known as the tomboys, and the boys who play with the girls become known as the sissies. Although the children are sanctioning each other without instruction from adults, they are also engaging in behavior that has been learned from adults -- not explicitly, but implicitly. Adults can reinforce narrow gender roles by promoting certain activities such as physical sports for boys and other things such as dance for girls.

      With respect to race, children receive messages all the time about beauty standards. Who are the favored students, and what are their characteristics? Who are the people who get into trouble a lot, and what are their characteristics? Much of the time preferential (or non-preferential) treatment is very much related to race.

      In many schools there may not be many explicit messages about race, but students receive implicit messages about race all of the time that informs what they think it means to be a member of a particular racial group. When they see Black students over represented on the basketball team but under represented in advanced placement courses, or Latino students over represented among those who’ve gotten into trouble, but under represented among those receiving awards, they get a clear sense about the meaning of race. The hidden curriculum related to race presents racial patterns as normal and effectively reinforces racial stereotypes. When it is operative it can completely undermine efforts to raise student achievement because students may believe that altering racial patterns simply is not possible.

      Too often, educators assume because of the choices Black students make about who to socialize with, which classes to take, etc. that they are anti-intellectual. (14) However, the vast majority of Black students I meet express a strong desire to do well in school. The younger students don’t arrive at school with an anti-intellectual orientation. To the degree that such an orientation develops, it develops in school, and from their seeing these patterns and racial hierarchies as permanent. Because a great deal of this behavior plays out in schools, educators can do something about it.

      What can educators can do?

      Understanding and debunking racial stereotypes, breaking down racial separations, and challenging the hidden curriculum are challenges not just for teachers, but for principals, administrators and entire school communities. In addition, there are a number of things educators can do to support their students’ positive racial identity development.

      First, educators can make sure that students are not segregating themselves -- sitting in racially defined groups in the classroom. For teachers, this can be as simple as mixing students and assigning them seats. Or, if work groups are created students can be assigned to groups in ways that ensure that students of different backgrounds have an opportunity to work together. This approach to race-mixing is often far more effective than holding an abstract conversation about tolerance or diversity. By working together, friendships are more likely to form naturally, and as students gain familiarity with one another, they may be more willing to break racial norms. If teachers let students choose, they will more than likely choose those whom they perceive to be “their own kind.”

      Second, educators can encourage students to pursue things that are not traditionally associated with members of their group. If students of color are encouraged by adults to join the debating team or the science club, play music in the band, or to enroll in advanced courses, it will be possible for greater numbers to challenge racial norms. Extracurricular activities in particular can serve a very important role in this regard and give young people a chance to get to know each other in situations that are not racially loaded. As is true for work groups, in the course of playing soccer or writing for the newspaper students can become friends. Research on extracurricular activities has shown that sports, music, theater and other activities can play an important role in building connections among young people and breaking down the very insidious links between racial identity and academic achievement. (15)

      Third, teachers can find ways to incorporate information related to the history and culture of students into the curriculum. This is important in helping students to understand what it means to be who they are; an essential aspect of the identity formation process for adolescents. Literature – novels and short stories -- can be very effective in this regard because it can help students to identify and empathize with children who may be from different backgrounds. Field trips and out-of-class experiences that provide students with opportunities to learn about the experiences of others can also help in expanding their horizons.

      Finally, an effective teacher who is able to inspire students by getting to know them can actually do a great deal to overcome anti-academic tendencies. They can do this by getting students to believe in themselves, by getting them to learn how to work hard and persist, and by getting them to dream, plan for the future and set goals. Over and over again, when you talk to students who have been successful, they speak about the role that significant adults have played at various points in their lives. (16) They talk about how these adults helped them recognize their own potential, and how they opened doors that they previously did not know existed.

      I believe there are many young people who are crying out for supportive relationships with caring adults. Differences in race, gender, or sexual orientation need not limit a teacher’s ability to make a connection with a young person. In my own work with students and schools I have generally found kids to be the least prejudiced of all people. They tend to respond well to caring adults regardless of what they look like. However, they can also tell if the adults who work with them are sincere, and those acting out of guilt and faked concern, can generally be detected.

      Today, most social scientists recognize race as a social rather than as a biological construct. It is seen as a political category created largely for the purpose of justifying exploitation and oppression. (17) For many adults and kids, especially those of mixed heritage, the categories often do not even correspond to who they think they are. Rather than being a source of strength, the acquisition of racial identities may be a tremendous burden.

      For many years to come, race will undoubtedly continue to be a significant source of demarcation within the US population. For many of us it will continue to shape where we live, pray, go to school, and socialize. We cannot simply wish away the existence of race or racism but we can take steps to lessen the ways in which the categories trap and confine us. As educators who should be committed to helping young people realize their intellectual potential as they make their way toward adulthood, we have a responsibility to help them find ways to expand identities related to race so that they can experience the fullest possibility of all that they may become.

      Published in In Motion Magazine December 1, 2002

    1. Stars are obviously a case of appearance

      I agree with the idea that stars are a case of appearance because we really only know of them what is being presented to us. Many people may think they know a star based on the character they played on a TV show or based off of one interview they've seen. We constantly hear on news platforms when a celebrity does something bad in the public eye, or when they are wearing the wrong thing. In my opinion, us ordinary followers get presented the bad in celebrities more than the good, & it paints a picture of them in our minds despite the fact that we really don't know them as a person, yet we are convinced that we are. Similarly, with actual appearance, we often are believed to think that a celebrity looks a certain way when, in fact, they got something done to enhance features on themselves which we are taught to be normal (just to clarify it is totally okay for someone to do anything they want to their bodies! its theirs for a reason) but it makes many followers wonder why they don't have the same feature. We only see the outside of celebs, & what the media portrays them as.

    2. I agree with the idea that stars are a case of appearance because we really only know of them what is being presented to us. Many people may think they know a star based on the character they played on a TV show or based off of one interview they've seen. We constantly hear on news platforms when a celebrity does something bad in the public eye, or when they are wearing the wrong thing. In my opinion, us ordinary followers get presented the bad in celebrities more than the good, & it paints a picture of them in our minds despite the fact that we really don't know them as a person, yet we are convinced that we are. Similarly, with actual appearance, we often are believed to think that a celebrity looks a certain way when, in fact, they got something done to enhance features on themselves which we are taught to be normal (just to clarify it is totally okay for someone to do anything they want to their bodies! its theirs for a reason) but it makes many followers wonder why they don't have the same feature. We only see the outside of celebs, & what the media portrays them as.

    1. There is a new culture emerging, badly needed as we face questions that may have old origins but that remain at the very core of our pursuit of knowledge. The choices we make now, as we shape our curricula and create academic departments and divisions, will shape the minds of future generations and of intellectual cooperation.

      I think that students should be encouraged to take courses in the humanities even if they're not a requirement because they'll be able to learn how to think critically which may also aid them in their science courses. I also think that the sciences and the humanities should be considered equally important; and not that one is higher or lower than the other.

    2. as we face questions that may have old origins

      Yeah I don't think the questions that the humanities pose will ever go away or be irrelevant. Perhaps not appreciated, but never irrelevant. If the questions of philosophy are what propelled us, then any answers that come about will need philosophy to make meaningful sense out of them.

    1. I think that's a salient point. Prescriptivists follow consistent rules; while, descriptivists allow variation, adaption, when necessary. Because both are not mutually exclusive--I do think prescriptivism makes it easier to communicate. Albeit a linguistic might argue that sets of patterns for how words to form phrases of clauses (whether spoken or in writing) is surely important because to put simply, we do not live in a Shakespearan era--and as the world evolves, society should demand evolution in the English language. How people use language has certainly changed. We are now using slang phrases--freely and abbreviation of words--constantly. Some may argue (whether this trend is good or bad; while others, might argue that "change is inevitable" and that certain words and/or phrases are "no longer in use." I think prescriptivism in grammar is important and should be required widely. However, most importantly, there is enough room for 'You and me' (descriptivism) and 'you' and I" (prescriptivism), without people being rebuked for misusing 'I' and 'me'. Finally, as languages become, according to the test, "[...] a form of cultural capital" and that "stigmatized forms are...used by social groups" begs the question that the article posed ("who's right?") and is grammar declining? Are grammar rules arbitrary and should we be confined to follow them? Or, are we destroying the English language with such thinking?

    1. Our lessons, units, and courses should be logically inferred from the results sought, not derived from the methods, books, and activities with which we are most comfortable.

      I like that this is the result of backward design, as it puts a focus back on student learning and pushes teachers to think about what is best for delivering content versus what they may be used to doing for a unit. -AG

    1. They need to acquire more languageand culture in order to be ready for more situations

      Language and culture not only helps us as communicators, but also prepares us varies situations we may encounter. It's amazing to think how these two create such a major impact on our lives and shapes into the person we are today.

      This is a great example of why a social constructive classroom is important to have. Allowing students to be open about their experiences helps surrounding students add to their knowledge and culture. By learning from others, we can be more equipped situations we may encounter.

      Even as an educator this is important!

    1. Note: This rebuttal was posted by the corresponding author to Review Commons. Content has not been altered except for formatting.

      Learn more at Review Commons


      Reply to the reviewers

      Response to the References

      Reviewer #1 (Evidence, reproducibility and clarity (Required)):

      In this manuscript Yan et al describe a method to perform imaging based pooled CRISPR screens based on photoactivation followed by selection and sorting of the cells with the desired phenotypes.

      They establish a system in mammalian RPE-1 cells where they integrate a photo-activatable mCherry, identify the cells of interest under the microscope based on a phenotype, automatically activate the mCherry fluorescence in these cells and then sort the desired populations by FACS. They demonstrate the reliability of their enrichment method and finally use this approach to look for factors that regulate nuclear size by a targeted pooled CRISPR screen.

      **Major points:**

      1.This year Hassle et al described a very very similar approach that they name: Visual Cell Sorting . In this case, they use a photoconvertible fluorescent protein (green-to-red conversion) to select cells with a certain visual cellular phenotype and enrich those by FACS. The Hassle et al 2020 MSB paper is only mentioned together with the other methods in the introduction in one sentence (ref #19 in this manuscript):

      " Recently, several in situ sequencing15,16 and cell isolation methods17-20 were developed which allow microscopes to be used for screening. However, these methods contain non-high throughput steps that limit their scalability."

      I think the current citation of the Hassle et al paper, is not really fair. The idea and the execution of the two approaches are almost exactly the same. Here, the authors concentrate on a CRISPR based application, but obviously the applications of the method are not limited to that. The authors should discuss how these similar ideas can be used in several different applications.

      We agree with the reviewer that we need to describe more about the Hasle et al. paper (now ref #20 in the revised manuscript) and expand our description of other applications that could be performed with the method. For this purpose, we have made the following changes:

      We have modified the relevant paragraph in the Introduction.

      p.3 the second paragraph

      Recently, an imaging based method named “visual cell sorting” was described that uses the photo-convertible fluorescent protein Dendra2 to enrich phenotypes optically, enabling pooled genetic screens and transcription profiling(Hasle, N.; Cooke, A.; Srivatsan, S.; Huang, H.; Stephany, J. J.; Krieger, Z.; Jackson, D.; Tang, W.; Pendyala, S.; Monnat, R. J., Jr.; Trapnell, C.; Hatch, E. M.; Fowler, D. M. 2020). Here, we developed an analogous approach to execute an imaging-based pooled CRISPR screen using optical enrichment by automated photo-activation of the photo-activatable fluorescent protein, PA-mCherry.

      We have also added the following paragraph in the Discussion.

      p.14 line 1

      In our study, optical enrichment was utilized for pooled CRISPR screens on phenotypes identifiable through microscopy. However, optical enrichment can be used for other purposes, as demonstrated previously(Hasle, N.; Cooke, A.; Srivatsan, S.; Huang, H.; Stephany, J. J.; Krieger, Z.; Jackson, D.; Tang, W.; Pendyala, S.; Monnat, R. J., Jr.; Trapnell, C.; Hatch, E. M.; Fowler, D. M. 2020). In a recent study by Hasle et al.(Hasle, N.; Cooke, A.; Srivatsan, S.; Huang, H.; Stephany, J. J.; Krieger, Z.; Jackson, D.; Tang, W.; Pendyala, S.; Monnat, R. J., Jr.; Trapnell, C.; Hatch, E. M.; Fowler, D. M. 2020), the process of separating cells by FACS after optical enrichment was termed “visual cell sorting”. This method was used to evaluate hundreds of nuclear localization sequence variants in a pooled format and to identify transcriptional regulatory pathways associated with paclitaxel resistance using single cell sequencing(Hasle, N.; Cooke, A.; Srivatsan, S.; Huang, H.; Stephany, J. J.; Krieger, Z.; Jackson, D.; Tang, W.; Pendyala, S.; Monnat, R. J., Jr.; Trapnell, C.; Hatch, E. M.; Fowler, D. M. 2020), demonstrating the broad applicability and power of this approach beyond CRISPR screening.

      1. While I understand that the authors mean conversion from the dark state to fluorescent state when they describe their photo-activatable mCherry, I think the term "photo-activation" can be confusing for the general reader since typically photo-conversion refers to a change in color. I would here suggest stick to the term photo-activation.

      We thank the reviewer for pointing this out and to avoid future confusion, we restricted the usage of photo-conversion to specifically indicate conversion of fluorescence from one color into another: e.g. when talking about the published visual cell sorting paper in which Dendra2 is used as a photo-convertible fluorescent protein. We use photo-activation in reference to the activation of PA-mCherry in our work.

      1. For validation of the hits coming from the nuclear size screen: Did the authors have any controls making sure that the right targets were down-regulated? This might be obvious for some of the targets (e.g. CPC proteins that are known to induce division errors display the nuclear fragmentation that the authors also observe) but especially for the ones that are less known or unknown to induce any nuclear size change, it will be important to demonstrate the specificity of the targets.

      For validating hits coming from the nuclear size screen, we have verified the successful transduction of corresponding sgRNA constructs by FACS analysis, but have not confirmed the knockdown. Before final journal publication, we propose to perform rt-qPCR on our 15 gene hits before and after knockdown to measure the percentage of knockdown separately.

      In addition, it is not clear from the figure legends and the material and methods if these phenotypes are verified by 3-4 gRNAs they use in the validation. Are the histograms representative of a single experiment with one gRNA or a combination of gRNAs in different experiments? Methods of replication of the data presented in Fig4 is unclear.

      We apologize for the confusion. These phenotypes were verified with pools of 3-4 sgRNAs and the histograms are representative of a single replicate infected with a mixed 3-4 sgRNA pool. We have modified the legend to Figure 5 (original Fig. 4) and the method section to explain this point.

      Minor points:

      1. Related to major point #3: I could not find much experimental info on how the hits from the screen were verified in materials and methods.

      The description of the experiment and information about the selected sgRNAs has been added in the Method section as follows:

      p.23

      Verification of hits from nuclear size screen

      For each hit in the nuclear size screen, the two sgRNAs with the highest phenotypic score in the screen and the two sgRNAs with the highest score predicted by the CRISPRi-v2 algorithm24 were selected and pooled to generate a mixed sgRNA pool of 3-4 sgRNAs (detailed information in Supplementary file 8). Cells (hTERT-RPE1 dCas9-KRAB-BFP PA-mCherry H2B-mGFP) were transduced with pooled sgRNAs targeting each gene and puromycin selected for 2 days to prepare for imaging. Cells were then seeded into 96-well glass bottom imaging dishes. Images were collected the next day and nuclear size was measured using the Auto-PhotoConverter µManager plugin. To focus on cells with successful transduction, BFP was co-expressed on the sgRNA construct and only cells with BFP intensity above a threshold value were included in nuclear size measurements. This BFP threshold was established by comparing the average BFP intensity of cells with and without sgRNA transduction (Fig.S3a).

      We agree with this important point and have changed the figure legend of Fig. 5c (original Fig. 4c) to just describe the plot:

      c, The ratios between median level of nuclear size measured from microscopy and H2B-mGFP fluorescence or FSC signal measured from FACS after knockdown, were plotted separately. TACC3, confirmed to be a control gene, was used for comparison (Grey bar).

      The typo has been corrected.

      Reviewer #1 (Significance (Required)):

      I think the idea of performing pooled screens coupled to microscopy is exciting and this approach has definitely more potential than the Craft-ID approach that the authors also discuss in their manuscript. In addition, the approach that is described in this manuscript is convincing and although the fact that the analysis part will require more work (to adapt the software to recognise different types of phenotypic readouts) in the future to make it accessible to the scientific community, the authors present sufficient evidence that the system can be robust. They also present some clever ideas such as to calculate enrichments with different photo-activation times (2sec vs 100ms) followed by separation of these populations by FACS.

      Reviewer #2 (Evidence, reproducibility and clarity (Required)):

      In this manuscript, Yan et al. present optical enrichment, a method for conducing pooled optical screens. Optical enrichment works by combining microscopy to mark cells of interest using the PA-mCherry photo-activatable fluorescent protein with FACS to recover them. The method is similar to other methods (Photostick, Visual Cell Sorting), and provides an alternative to in situ sequencing/FISH methods. The authors use optical enrichment to conduct a pooled optical CRISPRi screen for nuclear size. They identify and exhaustively validate hits, showing that optical enrichment works for its intended purpose. The development of a uManager protocol and discussion of the number of sgRNA's required for a genetic screen using optical enrichment were welcome. The authors' reported throughput of 1.5 million cells per eight hour experiment is impressive; and the demonstrated use of low cell number input for next generation sequencing appears promising. Overall, the manuscript is well written, the methods clear and the claims supported by the data presented.

      **General comments**

      -I found the analysis and scoring methods to be lacking, both in terms of the clarity of description and in terms of what was actually done. The authors might consider using established methods (eg https://www.biorxiv.org/content/10.1101/819649v1.full). In any case, they should revise the text to clarify what was done and address the other concerns raised below.

      -Relatedly, details regarding how to perform the experiments described are lacking. It is not clear from the text, figures, "Online Methods" section, and Supplementary Files whether all imaging is performed before activation, or whether each field of view is subject to an individual round of imaging followed by activation. It is also unclear whether cells in 96 well plates are sorted as 96 separate tubes or pooled into a single tube prior to sorting. Furthermore, at a minimum, the following details are requested for each optical enrichment "run". These details are critical considerations for those who seek to use optical enrichment in their own laboratories:

      Seeding density

      Time elapsed (in hours) between cell plating and optical enrichment

      The number of fields of view examined

      The median number of cells per field of view; the proportion of each plate's surface area that is imaged and photo-converted

      The total time taken (in hours) to perform imaging and photoconversion

      The gating protocol used for sorting by FACS (preferably including a figure with example gates for one or two experiments). The gating protocol is described for the genetic screen but not for the control experiments.

      We agree with the reviewer and apologize for the confusion that arose from our description. We also thank the reviewer for suggesting using established methods. However, MAUDE, an analysis for sorting-based CRISPR screen with multiple expression bins, might not be suitable for our study since 1) the distribution of mCherry fluorescence intensity is a reflection of photo-activation efficiency and not sgRNA effect 2) only one sorting bin is collected for each experimental condition. Our analysis is adapted from an existing method from the Weissman lab (https://github.com/mhorlbeck/ScreenProcessing).

      We agree with the reviewer regarding clarifying other points and rewrote the following part in the Method section:

      p. 20

      mIFP proof-of-principle screen, Nuclear size screen, FSC screen and H2B-mGFP screen

      For the mIFP proof-of-principle screen, mIFP positive cells (hTERT-RPE1 dCas9-KRAB-BFP PA-mCherry H2B-mGFP mIFP-NLS) and mIFP negative cells (hTERT-RPE1 dCas9-KRAB-BFP PA-mCherry H2B-mGFP) were stably transduced with the “mIFP sgRNA library” (CRISPRa library with 860 elements, see Supplementary file 5) and the “control sgRNA library” (CRISPRa library with 6100 elements, see Supplementary file 6) separately. For the nuclear size screen, FSC screen and H2B-mGFP screen, cells (hTERT-RPE1 dCas9-KRAB-BFP PA-mCherry H2B-mGFP) were stably transduced with the “nuclear size library” (CRISPRi library with 6190 elements, see Supplementary file 7). To guarantee that cells receive no more than one sgRNA per cell, BFP was expressed on the same sgRNA construct and cells were analyzed by FACS the day after transduction. The experiment only continued when 10-15% of the cells were BFP positive. These cells were further enriched by puromycin selection (a puromycin resistance gene was expressed from the sgRNA construct) for 3 days to prepare for imaging. For FSC and H2B-mGFP screens, cells were then subjected to FACS sorting. Cells before FACS (unsorted sample for FSC and H2B-mGFP screens) and top 10% cells based on either FSC signal (high FSC sample) or GFP fluorescence signal (high GFP sample) were separately collected and prepared for high throughput sequencing. For mIFP proof-of-principle screen and nuclear size screen, cells were then seeded into 96-well glass bottom imaging dishes (Matriplate, Brooks) and imaged starting from the morning of the next day (around 15 hr after plating). A series of densities ranging from 0.5E4 cells/well to 2.5E4 cells/well with 0.5E4 cells/well interval were selected and seeded. The imaging dish with cells around 70% confluency was selected to be screened on the imaging day. For mIFP proof-of-principle screen, a single imaging plate was performed for each replicate while 4 imaging plates per replicate were imaged for the nuclear size screen. When executing multiple imaging runs, 2 consecutive runs could be imaged on the same day (day run and night run). 64 (8x8, day run) or 81 (9x9, night run) fields of view were selected for each imaging well and each field of view was subjected to an individual round of imaging directly followed by photo-activation. Around 200-250 cells were present in each given field of view and 60% to 80% surface area of each well was covered. Either mIFP positive cells or cells passing the nuclear size filter were identified and photo-activated automatically using the Auto-PhotoConverter µManager plugin. The total time to perform imaging and photo-activation of a single 96-well imaging dish with around 1.5 million cells was around 8 hr. The night run generally took longer, since more fields of view were included than in the day run. Cells were then harvested by trypsinization and pooled into a single tube for isolation by FACS. Sorting gates were pre-defined using samples with different photo-activation times (e.g. 0s, 200ms, 2s) and detailed gating strategies are described in Supplementary file 1. Sorted samples were used to prepare sequencing samples.

      -The authors use PA-mCherry. There are a variety of other photo-activatable fluorophores available, and it would be good for them to comment on why they chose PA-mCherry. Also, since the method is supposed to be used for generic pooled optical screens, it would be good for the authors to comment on what colors remain available for imaging cellular structures.

      To address these, we have added the following sentences:

      p. 4 line 16

      A photo-activatable fluorescent protein was chosen over a photo-convertible fluorescent protein to increase the number of channels available for imaging. PA-mCherry was chosen to leave the better performing green channel open for labeling of other cellular features. Moreover, non-activated PA-mCherry has low background fluorescence in the mCherry channel (Fig. S1b), and it can be activated to different intensities when photo-activated for various amounts of time.

      p. **14 line 10

      Phenotypes of interest should be identifiable under the microscope and generally require fluorescent labeling. Commonly used fluorescence microscopes use four channels for fluorescent imaging with little spectral overlap: blue, green, red and far red. In our study, the red channel was occupied by cell labeling with PA-mCherry and the blue channel was used to estimate sgRNA transduction efficiency. Since sgRNA transduction efficiency can be measured by other approaches, the blue channel could be used together with the remaining two channels to label cellular structures. Combining bright field imaging with deep learning can be used to reconstruct the localization of fluorescent labels(Ounkomol, C.; Seshamani, S.; Maleckar, M. M.; Collman, F.; Johnson, G. R. 2018), making it possible to use bright field imaging to further expand the phenotypes that can be studied with our technique.

      -In general, the figures are hard to read, with most space being dedicated to beautiful but complex schematics/workflows. Points and fonts should be bigger, and the authors should consider revising the schematics to take up less space.

      We thank the reviewer for this remark and revised all figures accordingly. Points and fonts were enlarged, and schematics were simplified or removed.

      -There is extensive use of editorialzing adverbs. Adverbs such as "highly" (abstract and page 15), "easily" (pages 4 and 11), "completely" (page 11), and "only" (page 12) are unnecessary at best and unsupported by the data at worst (e.g. cells are not "completely" separable with 100 ms photo-conversion, see page 11 and Figure 1C). Please remove "completely" from page 11 and consider removing other adverbs as well.

      We agree with the reviewer and the following adverbs have been removed: “highly” in abstract and page 15; “easily” on pages 4 and 11; “completely” on page 11 and three “only” on page 12.

      -Apologies if I missed it, but I couldn't find a data availability statement. Sequencing reads from the experiments should be deposited in SRA or GEO and made available upon publication.

      We apologize that we missed this, and the sequencing data has been deposited to GEO (GSE156623) which will be made available before final publication. The following part has been added to address this.

      p. 24

      DATA AND SOFTWARE AVAILABILITY

      The raw and processed data for the high throughput sequencing results have been deposited in NCBI GEO database with the accession number (GSE156623). The plugin Auto-PhotoConverter developed for open source microscope control software μManager(Edelstein, A. D.; Tsuchida, M. A.; Amodaj, N.; Pinkard, H.; Vale, R. D.; Stuurman, N. 2014) has been deposited on github (https://github.com/nicost/mnfinder).

      **Specific comments**

      Pages 5/6 - The authors present experiments that show that optical enrichment is highly specific for desired cells. But, they should consider presenting precision (fraction of called positives that are true positive) and recall (fraction of all true positives that are called positive) instead. I think these relate more directly to a pooled optical screen than specificity.

      We apologize for our poor terminology. Our original definition of “specificity” is the same as “precision” suggested by the reviewer. To avoid future confusion, we have changed all relevant occurrences of “specificity” into “precision”. The following sentence was modified to clarify the definition:

      p. 5 line 15

      To evaluate the precision (the fraction of called positives that are true positives) of this assay, all cells were collected and analyzed by FACS after image analysis and photo-activation (Fig. 2d and 2e). We calculated precision as the fraction of photo-activated cells (mCherry positive cells) that are true positives (mIFP-mCherry double positive cells) (Fig. 2f).

      Measuring recall is complicated because the microscope is unable to visit all locations in the imaging plate, hence recall will depend on the fraction of cells actually “seen” by the microscope. For the screening strategy employed in the nuclear size screen, recall is not as important as precision, since lower recall rates are compensated for by screening larger cell numbers. We therefore did not attempt to measure recall directly.

      Page 6 - Related to the above point, the authors state "These results indicate the assay yields reliable hit identification regardless of the percentage of hits in the library." This statement seems too strong given that the authors looked at specificity experimentally with a mixture of ~1% mIFP positive cells. In fact, hits might be much less than 1% of the total population of cells, and specificity would certainly fall from the 80% measured at 1% of the total population. The authors should do a bit more to fairly discuss their ability to find rare hits.

      We agree with the reviewer and have changed the following description:

      p. 5 line 20

      The precision varied with the initial percentage of mIFP positive cells and ranged from 80% to ~100% (initial percentage of mIFP positive cells ranging between 2.3% and 43.7%) (Fig. 2f). Precision is expected to fall below 80% with initial percentage of mIFP positive cells less than 2.3%. However, these results indicate that optical enrichment can be used to identify hits with high precision even at relatively low hit rates.

      Pages 6/7 - The authors perform a validation experiment using two different sgRNA libraries, infecting mIFP- and mIFP+ cells separately. Then, they demix these populations via optical enrichment, sequence and compute a phenotype score for sgRNAs or groups of sgRNAs. The way the experiment is described and visualized is extremely confusing. If I understood correctly (and I am not sure that I did), the bottom right panel of Figure 2b shows that if sgRNAs are (randomly?) paired AND two replicates are combined then optical enrichment nearly perfectly separates all (combined, paired) sgRNAs in the two libraries. The authors should rewrite this section, especially clarifying what is meant by "1 sgRNA/group and 2 sgRNA/group," and consider changing Figure 2b (perhaps just show the lower right panel?).

      We apologize for our confusing description. To avoid the confusion, we rewrote the paragraph describing the experiment and added a schematic (Fig. 3a) to better describe this experiment. We also simplified the result by just presenting the lower right panel of original Fig. 2b (current Fig. 3b) and moved the other data into supplementary figures (Fig. S2).

      p. 6 line 4

      mIFP negative cells and mIFP positive cells were separately infected with two different CRISPRa sgRNA libraries (6100 sgRNAs for mIFP negative cells; 860 sgRNAs for mIFP positive cells) at a low multiplicity of infection (MOI) to guarantee a single sgRNA per cell. Note that in these experiments, the sgRNAs only function as barcodes to be read out by sequencing, but do not cause phenotypic changes as the cells do not express corresponding CRISPR reagents. These two populations were then mixed at a ratio of 9:1 mIFP negative cells: mIFP positive cells. We again used mIFP expression as our phenotype of interest (outlined in Fig. 3a). Two biological replicates were performed and at least 200-fold coverage of each sgRNA library was guaranteed throughout the screen, including library infection, puromycin selection, imaging/photo-activation and FACS.

      Page 8 - Related to Supplementary Figure 3, why are there not clear BFP+ and BFP- populations but instead one continuous population? How was the gating determined (e.g. how was the boundary between red and gray picked)? Here, and generally, flow plots and histograms of flow plots should indicate the number of cells. If replicates were performed, they should be included.

      We have clarified our description. There are no clear BFP+ and BFP- populations but instead one continuous population due to the background expression of BFP from the dCas9 construct: dCas9-KRAB-BFP (which is now clearly indicated in the manuscript). On top of the dCas9-KRAB-BFP, another BFP is encoded on the sgRNA construct, which leads to a higher BFP expression level.

      There was no gating in the experiment, the grey dots in the figure represents wild type cells without viral transduction while the red dots (partially covered by the grey dots) were cells infected with the two negative control sgRNAs. We mistakenly wrote the legend of original Fig. S3 (current Fig. S3a) that these were FACS data; however, the data were acquired by imaging. We apologize for the confusion and thank the reviewer for detecting the issue. We completely rewrote the legend to Fig. S3a (original Fig. S3) to clarify.

      We now include the number of cells analyzed and the number of replicates for the other flow plots and histograms in the manuscript.

      Page 8 - "Nuclear sizes...". The authors should say in the main text what size metric was used.

      To address the reviewer’s point, we have included the following sentence:

      p. 8 line 23

      We defined nuclear size as the 2D area in square microns measured by H2B-mGFP using an epifluorescence microscope, as determined by automated image analysis (Fig. 4a and Supplementary file 2).

      Page 9 - I am a little confused about the statistical analysis of the screen. In Supplementary File 1, the authors state that p-values were "calculated based on comparison between the distribution of all the phenotypic scores of sgRNAs targeting to the gene/assigning in the group and the one of negative control sgRNAs in the libraries." I presume this means that all phenotypic scores (across replicates) of all sgRNAs targeting each gene were included in a Mann Whitney U test with a single randomized set of phenotypic scores. If that's right, it seems like an odd way to get p-values. Better would be a randomization test, where a null distribution of phenotypic scores for each gene is built by randomizing sgRNA-level scores many times. Then the actual phenotypic score is compared to the randomized null distribution, yielding a p-value. In any case, the authors must clarify what they did in the main text and Supplementary File 1.

      Page 9 - It does not appear that the p-values presented in Figure 3c have been adjusted for multiple hypothesis testing. This should be done.

      Page 9 - "A value of the top 0.1 percentile of control groups was used as a cutoff for hits." Why? This seems arbitrary. It seems like appropriate false-discovery rate control would enable a more rigorous method for choosing a cutoff.

      Page 9 - The same comments regarding analysis and scoring of the optical enrichment screen applies to the FSC and GFP screens.

      We clarified the description of the statistical analysis of the screen (see new/changed text below). Mann-Whitney p-values for the two replicates were calculated independently. The Mann-Whitney U test was not performed against a randomized set of phenotypic scores, but using the phenotypic scores of the 22 control non-targeting sgRNAs that were part of the library. Because there are only 22 control sgRNAs (adding more control sgRNAs would increase the size of the library, and reduce the number of genes that can be screened within a given amount of time), the statistical significance of testing genes against these controls is not expected to be very high, and using direct approaches such as multiple hypothesis testing are not expected to yield hits. Instead, we calculated a score combining the severity (phenotypic score) and the trustworthiness (Mann-Whitney p value) of the phenotype (a method previously developed in the Weissman lab at UCSF: https://github.com/mhorlbeck/ScreenProcessing24). We thank the reviewer for suggesting using false discovery rate control as a better method for choosing a cutoff. We modified our original analysis and now determine the threshold of our score based on a calculated empirical false discovery rate (eFDR). We used this approach to maximize the number of true hits and relied on a repeat of the screen and follow-up testing of hits to narrow down true hits. We added the following part in the method section and added an analysis example to the supplementary files (Supplementary file 9)."

      p. 22

      Bioinformatic analysis of the screen

      Analysis was based on the ScreenProcessing pipeline developed in the Weissman lab (https://github.com/mhorlbeck/ScreenProcessing)**(Horlbeck, M. A.; Gilbert, L. A.; Villalta, J. E.; Adamson, B.; Pak, R. A.; Chen, Y.; Fields, A. P.; Park, C. Y.; Corn, J. E.; Kampmann, M.; Weissman, J. S. 2016). The phenotypic score (ε) of each sgRNA was quantified as previously defined(Kampmann, M.; Bassik, M. C.; Weissman, J. S. 2013)** (Supplementary file 9). For the mIFP proof-of-principle screen, phenotypic score of each group was the average score of two sgRNAs assigned to the group and averaged between two replicates except otherwise described. For the nuclear size screen, FSC screen and H2B-mGFP screen, genes were scored based on the average phenotypic scores of the sgRNAs targeting them. For the nuclear size screen, phenotypic scores were further averaged between 4 runs for each replicate. For the nuclear size screen, FSC screen and H2B-mGFP screen, sgRNAs were first clustered by transcription start site (TSS) and scored by the Mann-Whitney U test against 22 non-targeting control sgRNAs included in the library. Since only 22 control sgRNAs were included, significance of hits was assessed by comparison with simulated negative controls that were generated by random assignment of all sgRNAs in the library and phenotypic scores of these simulated negative controls were scored in the same way as phenotypic scores for genes. A score η that includes the phenotypic score and its significance was calculated for each gene and simulated negative control. The optimal cut-off for score η was determined by calculating an empirical false discovery rate (eFDR) at multiple values of η as the number of simulated negative controls with score η higher than the cut-off (false positives) divided by the sum of genes and simulated negative controls with score η higher than the cut-off (all positives). The cut-off score η resulting in an eFDR of 0.1% was used to call hits for further analysis (Supplementary file 9). An example analysis is described in detail in Supplementary file 9 and raw counts and phenotypic scores for all four screens are listed in Supplementary file 10 and 11.

      Page 9 - "These data suggest that a direct measurement utilizing a microscope can provide significant improvement in hit yield even for phenotypes that could be indirectly screened with other approaches." I think this conclusion is too strong. It rests on the assumption that the FSC/GFP phenotypes should have the same set of hits as the microscope phenotype (larger nuclear area). This may not be the case. For example, genes whose inactivation increases GFP expression would be hits in the former, but not latter case. The authors should moderate this statement.

      We agree with the reviewer and have changed the sentence into:

      p. 10 line 17

      These data suggest that a direct measurement utilizing a microscope can provide different information and reveal hits that are inaccessible using other screening approaches.

      Page 11 - "This is significantly faster than the in situ methods." The authors should provide a citation and an actual comparison to the speed of in situ methods.

      We agree with the reviewer and have modified the sentence with a citation:

      p. 12 line 20

      This is significantly faster than in situ methods which process millions of cells over a period of a few days(Feldman, D.; Singh, A.; Schmid-Burgk, J. L.; Carlson, R. J.; Mezger, A.; Garrity, A. J.; Zhang, F.; Blainey, P. C. 2019).

      Page 12 - I think the authors could say a bit more about the possibility of low hit rate screens. How low do they think it is feasible to go? What hit rates are expected based on existing arrayed optical screens?

      We have added more description in the discussion section:

      p. 13 the second paragraph

      Optical enrichment screening also is possible for phenotypic screens with relatively low hit rates (defined as the fraction of all genes screened that are true hits). The ability to detect hits at low hit rates in our method depends on multiple factors, including: 1) the penetrance of the phenotype; 2) cellular fitness effect of the phenotype; 3) detection and photo-activation accuracy of the phenotype; 4) limitations imposed by FACS recovery and sequencing sample preparations of low cell numbers. The first three factors vary with the phenotype of interest. We optimized the genomic DNA preparation protocol (Methods), and are now able to process sequencing samples from a few thousand cells, enabling screens of low hit rate phenotypes. In our nuclear size screen, more than 1.5 millions cells were analyzed during each run with 2000-4000 cells recovered after FACS sorting. The hit rate of this screen was 2.76%, similar to optical CRISPR screens performed in an arrayed format(de Groot, R.; Luthi, J.; Lindsay, H.; Holtackers, R.; Pelkmans, L. 2018)**, demonstrating the possibility to apply our approach to investigate phenotypes with low hit rates.

      Page 14 - It is weird that the discussion includes a fairly important couple of paragraphs that seem to belong in the results (e.g. the text surrounding Figure 4b and c). Obviously, I don't want to prescribe stylistic changes, but I suggest the authors consider moving this description of the experiments/analyses to the results.

      The relevant description has been moved to the results.

      Page 14 - The authors validate their hits individually, and observe that expression of hit sgRNAs does increase nuclear size in some cells. But, many/most cells remain control-like in these validation experiments. The authors should comment on why this is the case (e.g. inefficient knockdown, cell cycle effects, etc).

      To address this point, we have added the following sentences in legend of Fig. 5:

      The cell population is heterogeneous due to inefficient knockdown, incomplete puromycin selection, and penetrance of the phenotype. A BFP was expressed from the same sgRNA construct. Only cells with high BFP intensity, indicating successfully sgRNA transduction, were included for data analysis as described in Methods.

      Page 14 - It would be nice to formally compare the control and sgRNA distributions in each panel of 4a and Supplementary Figure 5 (e.g. with a Komolgorov-Smirnov test, etc). That would allow a more precise statement to be substituted for "14 out of 15 hits (the exception was TACC3) were confirmed to be real hits, with cells exhibiting larger nuclei after knock down (Fig. 4a and Fig. S5)," which is not quantitative.

      We applied the Kolmogorov-Smirnov test and the corresponding sentence was changed into:

      p. 10 last line

      *14 out of 15 hits were confirmed to be real hits (Kolmogorov-Smirnov test two tailed p-value

      Figure 2a - I am not sure it is necessary to show the entire workflow again. The first and possibly last panels are the informative ones here.

      Figure 3a - Same comment as above - these workflow panels take up a lot of real estate and I suggest simplifying them if possible.

      The figures were simplified to just show the example images.

      Figure 3c - At least on my PDF/screen, the "scrambled control" points appear very light gray and are impossible to find. They should be an easier to spot color.

      We agree with the reviewer and changed the color.

      Figure 4b - "Most cells developed a larger cellular size and higher H2B-mGFP level after knock down." I think it would be more accurate to say that the median cell size/GFP level increased, or that some cells developed larger sizes/median GFP levels.

      We agree with the reviewer’s point; “most” has been changed to “some”.

      Figure 4c - I don't understand "Normalized FITC/nuclear size." Do the bars show the mean/median of a population (if so, why not show a dot plot or box plot or violin plot)? Also, what is FITC (I presume it's GFP levels)?

      Figure 4c - "Most cells maintained a constant ratio between nuclear size and DNA content..." I'm not sure where DNA content came from. Are the authors assuming that their H2B-mGFP is a proxy for DNA content? Or was some other measurement made? If the former, is there a citable reason why this is a good assumption?

      The bars represent the ratio of the median level of H2B-mGFP intensity (the axis is now labeled with "GFP" rather than "FITC", the colloquial name for the channel used on the FACS machine) measured by FACS and the median nuclear size of the same population of cells measured by microscopy. We plan to perform additional experiments to measure DNA content using a DNA dye in the same cell by microscopy so that we will be able to correlate these on a cell by cell basis. Data will be added before final publication.

      Reviewer #2 (Significance (Required)):

      I don't generally comment on significance in reviews. Since ReviewCommons is specifically asking, I'll say that this manuscript describes optical enrichment, a method that is an extension of previous work and is substantially similar to a previously published method, Visual Cell Sorting. However, given the timing, it is obvious that these authors have been working independently on optical enrichment. Since the application is distinct, and optical enrichment incorporates some nice features like software to make it easier to execute, it is clearly of independent value.

      Reviewer #3 (Evidence, reproducibility and clarity (Required)):

      This study reports a rapid and high-throughput CRISPR-based phenotypic screen approach consisting of selecting cells with phenotypes of interest, label them by photo-conversion and isolating them by FACS. The idea of the method is interesting (has been around) in principle. The key advantage is that is relatively simple, accessible to many groups as it does not require robotics. However, the manuscript is so badly written and hard to follow, that it makes it difficult to judge the technology, to really understand how the experiments were done and whether the results are interpreted correctly. Strictly speaking, it is unclear whether and how good scientific practices GSP have been followed, as the description of the experiments is sometimes lacking totally. Consequently, it is impossible to seriously evaluate this study and judge whether the technology described is really promising. It is probably less sensitive than arrayed screens, in all likelihood can miss hits that affect growth, cannot capture as many phenotypic classes as one would like from high-content screens and the computational and experimental workflow is more complicated. It is puzzling that the authors don't even compare the results with arrayed screens which are of course the current gold-standard.

      We do not in any way claim that the presented method replaces arrayed screens. However, most current sgRNA libraries are pooled libraries, and the few available arrayed sgRNA libraries are expensive and difficult to maintain, hence our methods to screen pooled sgRNA libraries are timely and useful. Comparisons with arrayed screens are unwarranted as no claims are made with respect to arrayed screens.

      We have clarified the manuscript in many places, and hope it is now readable and better understandable by more readers with diverse backgrounds.

      **Specific points:**

      The specificity test (Fig 1) does not make sense how it is described. If the authors spike a certain percentage of cells that can be photoconverted, when analysing the outcome, there will be three classes: mIFP positive, mIFP/mCherry positive and negative. How can they calculate specificity if they do not know whether they converted all mIFP cells? Also the formula used is questionable or is her an error? Furthermore, it is totally unclear how many cells were used and how they were scanned. If they took 90 negative cells and 10 mIFP cells, getting them all back is easy. If they start with 10e9 cells, the specificity should be quantified. Furthermore, the phenotype they pick is an easy and convenient one. Much more challenging is to apply it on a multi-parametric phenotype. Again, this is now the gold standard.

      We used the term specificity inadvertently and should have used precision, as also pointed out by Referee 2. This has been corrected in the current manuscript. We picked the mIFP phenotype as this was a proof of principle screen to clarify the performance of our screening approach and needed a phenotype that can be measured both by microscopy and FACS. We demonstrate that multi-parametric read-outs are possible, but do not think that the first demonstration of new technology needs such an application.

      In their first sgRNA assay, it is not possible to have a clear idea of what groups they are talking about. Do they mean they get phenotypic signatures which they group? How? They need to describe what they do. Here, only ~3500 genes are scanned (the 6843 is both populations and you only select from the mIFP neg population) and it took them 8hrs. This means for the genome it would require ~60h which is indeed fast. However, this experiment is not clearly described. They cannot select the negative population since there is no fluorescent marker (except false positive which are around 1.7%). So I assume they just randomly pick cells (they should really explain much better what they do!). Why go through the hassle? If these sequences are supposed to be a negative population, just pick them in the computer. Also, they cannot calculate an enrichment compared to the negative population, since two different libraries were infected. Again, I can't follow.

      We improved the description of this experiment. To clarify, we used mIFP in a proof of concept screen to validate whether sgRNAs infecting mIFP positive cells can be distinguished from those infecting mIFP negative cells No phenotypic signature other than the mIFP signal is used (as described in the text). As customary in pooled screens, a primary comparison was made between the positive (optically selected) cells and the complete population. To improve the clarity of this screen, we further described the concept of pooled sgRNA screens, which may have made this section harder to follow.

      I find their results about calculating scores based only on true negatives surprising. The average phenotypic score is improved from 3 to 5, which is enormous. This suggests that the phenotypes induced in the mIFP population are extremely common. These results are hard to interpret given the poor description of the experiment. It is possible that it is the same dataset as in 1, but in that case, the false negatives must be rare since the negatives can be selected by absence of both mCherry and mIFP.

      There are no phenotypes induced in the mIFP population (as now explicitly explained in the text). The mIFP population is isolated using optical enrichment, and we test our ability to discriminate the sgRNAs present in the enriched population. It is unsurprising that comparing to the negatively selected population (which is not possible in most other pooled screens) is significantly better than comparing against the total population (as customary in pooled screens).

      In the nuclear size screen, 6000 sgRNAs were screened. To array so many sequences would require 20 plates. They required ~40h for imaging one replicate. This is slow, imagine the time with a 60x lens.

      There are no arrayed screens performed in our study.

      Reviewer #3 (Significance (Required)):

      Overall, there is no sufficient evidence in this manuscript to convince this reviewer that this method is valid and truly powerful. I cannot support publication in its present form.

    1. But I wondered: Though agency may be good for you, does seeing yourself as a strong protagonist come at a cost to the other characters in your story? Are there implications for empathy if we see other people as bit players instead of protagonists in their own right?

      I think that people can balance agency and becoming a strong protagonist without it affecting other "characters" in their story. I'm not sure it's necessarily absolute.

    1. We don’t have to turn away from the mainstays of Victorian literature to study empire and racialization, although it may be useful to set these works in new constellations.

      I think a change of framing would be perfect for "undisciplining Victorian study." We can change what we take from these works when we view them not as "classics" but as the works produced by the rulers of an empire, or a higher, oppressive class.

    1. Note: This rebuttal was posted by the corresponding author to Review Commons. Content has not been altered except for formatting.

      Learn more at Review Commons


      Reply to the reviewers

      We would like to express our upmost gratitude to the three anonymous reviewers for their constructive and insightful comments on our manuscript. We broadly agree with all comments made and have uploaded a preliminary revised version with changes highlighted in bold. We now deal with each of the reviewer comments in turn.

      Reviewer #1

      L50-52: Can you predict where the unmapped read came from? Could viral infections be the source as in land plants?

      Having done a crude examination of unmapped reads, we couldn't find compelling evidence of them being of viral origin. The unmapped fraction in fact was in the same region as seen for other sRNA libraries in our lab which we found to occur for a number of reasons such as sequencing errors, incomplete assembly, differences between the sequenced lines and the reference line. Those all result in unmapped reads, which is also cause by since we employed a stringent mapping (0 mismatches).

      L67-68, which is the explanation?

      Thank you for querying this. After much closer inspection of the papers cited by Casas-Mollano et al. as evidence of the 23nt peak the evidence for the 23nt doesn't seem that strong and may even be a mistake on their part. Nonetheless, it is far from a critical piece of information for this paper and we have thus decided to remove this sentence.

      Fig 1D the reference to the A,C,G,U 5' should be re-positioned within Figure 1D panel space.

      Thanks, this has been addressed.

      Figure 3: it could be a supplementary figure based on the relevance given in the manuscript to this point.

      We agree, and have moved Fig3 to Supplement.

      *P5, line 107: while commenting on strand bias there seems to be a mistake in strong bias definition, it should be x 0.8, not "strong bias (0.2

      Thank you for pointing this out, we have now corrected this error. We have duly corrected it in the text.

      P5, line 110: marked changes regarding locus size are not as striking in my opinion, in particular log size 6 and following, which is not marked in the graph (the cut off between 6 and 8). Maybe this curve should be split into two distribution graphs based on some important features (as repetitiveness?) that might allow a better definition of cut-offs.

      Thank you for pointing this out. You are correct that the changes in the density distribution are not as striking for locus size. A great deal of deliberation on our part went into deciding what to do about this. In the end, we decided that for the size classes there was benefit in having several different classes with the understanding that having additional potentially redundant cut-offs would not adversely effect the analysis. In doing this, we were partially driven by the albeit subtle changes in the curve, but also by the desire to have size classes that were biologically relevant and informative. For example, a locus 3000nt captures the long tail. However, we neglected to fully explain these subtleties in our decision-making, something we have now rectified through some added explanation in the text. These choices were validated by the way size classes are differentially associated with different locus clusters in Figure 8.

      Fig 5: the legend has the C subfigure twice, the second should be D.

      Thank you for highlighting this. It has now been corrected.

      Table 1: I believe the data would be better presented in a plot, potentially something similar to the plot in Figure 1 A and B. The numbers are already presented in the supplementary spreadsheet.

      Thanks for pointing this out. We agree with this suggestion and have replaced Table 1 with a Figure (Fig 5) which is indeed a better way to present those results.

      Fig 6A: The boxplots regarding Stability of the clusters should be better described. What exactly does the y-axis in each "small plot" represent?

      Thank you for pointing this out, we understand that this isn't clear at the moment. Briefly, for this analysis we performed the clustering multiple times each time with a random sample of the loci (with replacement) of the same size as the original dataset. We then calculated the proportion of loci that retained their original clustering. We have clarified this in the figure legend and also elaborated on the approach in the methods section to ensure that it is better described.

      P6, line 142: analyses of stability and variance shows 7 as the optimal k, while gap statistics and NMI suggested 6 as the optimal. It is not clear why 6 was preferred. The MCA section in Methods is unclear regarding this point too.

      Thank you for querying this. The process of choosing the appropriate value of k is a complicated one and we appreciate that the explanation could be clearer. After your comment, we re-visited our decision-making process and were reassured that a k value of 6 rather than 7 was indeed appropriate. The stability plots in Fig. 6A start with k=2 and it can be clearly seen for k=6 that stability is comparatively high for dimensions 7-10. Indeed, k values of 2,3 and 6 seem to be the only feasible values. k=7 is fairly unstable for all dimensions from 1-8. We have done some rewording of the methods to hopefully make this clearer.

      Fig S2-S5: please check legends, they are identical, although they should cover examples of loci in LC2 through LC5. These figures are not cited in the text, only S1 and S2.

      Thanks for pointing this out. This is now corrected and we have referenced all figures in the main text.

      Fig 9: I suggest using different colors in density plots to ease interpretation. LC tracks could share a color and Gene, TEs, DNA meth, and All loci should have a different color each.

      A good suggestion - this has been replotted with different colours.

      Supplementary Files S1: The full-annotated locus map should be provided as a spreadsheet file or as a text (.csv) file, not as a pdf file.

      Thanks for pointing this out. We originally submitted this file as a gff format. We are not sure why this got converted. We will make sure this is going to be in appropriate format in the final form, especially having suffered from the pains of pdf tables ourselves in the past.

      I may be misunderstanding Fig. 6E, but it looks strange that the observed sum-of-squares is smooth, but the expected is not. Is it possible that the in-figure reference is inverted?

      Indeed, the colours were inverted. Thanks a lot for that spot, we have now swapped them around.

      Reviewer #2

      I am concerned that the methodology used does not adequately distinguish small RNA loci that are attributable to random RNA degradation products from loci that are truly fit the DCL / AGO paradigm. I think this is critical to maximize the utility of the annotations for the community. This issue was not directly addressed in the current version of the manuscript. There is cause for concern: 64% of the annotations overlap with protein-coding genes (lines 116-117), 55% with exons (line 118), and 41% of loci show strong strand bias (lines 123-124). These are all associations expected for breakdown products of mRNAs. Furthermore, only 11% of the loci were found to be dependent on CrDCL3 (line 123). Small RNA sequencing data from the other 2 DCL mutants are not yet available (line 211). One way that has been effective in angiosperms is to track the proportion of "DCL-sized" RNAs within all RNAs from each locus. Loci comprised of random degradation products will be single-stranded, generally touching exons, and have a very wide size distribution. In contrast, loci where the small RNAs are truly created by a DCL protein will have a very narrow size distribution. In any event, I think a strong effort to identify and flag small RNA loci that are less likely to be DCL / AGO silencing RNAs, and more likely to be degradation products, would be an important change to this study.

      Thank you for this very insightful comment which has helped us to reflect on the methodological approach. While it is likely that there are some RNA breakdown products picked-up in the sRNA sequencing, we do not think that the locus-map as a whole is undermined by this. For example 54% of loci have a predominance for 21-nt sRNAs and 18% for 20-nt sRNAs, so the majority of sRNA loci do have a predominance for a specific RNA size.

      However, your point does raise a very valid concern with implications for the interpretation of LC4. Although we posit some explanations for these loci (e.g. DCL-mediated sRNA production without an accessory protein to provide PAZ domain-like sRNA measurement), given the very strong strand bias and association with genic regions we do agree that there is a risk that these loci predominantly represent degradation fragments. Therefore, we have now reworded how we discuss LC4 in the discussion to reflect this. This also reveals a key advantage of the clustering approach in that should LC4 indead represent degradation products, they have been successfully grouped together into a seperate cluster such that they don't undermine the insights gained from the other locus clusters.

      One of the key results likely to be used by others is the final GFF3 file (Sup File S1). The Description fields in this file are extremely verbose. Do these load well on a genome browser? I suggest it might be good to store most of the information currently in the Description field in a separate flat file, and limit the GFF3 descriptions to key information (locus name, the LC group).

      Thank you for pointing this out. In a pursuit to share as many details as possible, we appreciate that this can be too verbose, as righlfully noticed here. In order to not compromise detail too much, we have created a second, toned down, version as csv which now includes essential details such as name, position and LC. As for the gff, we kept all details in since it loads quickly in a genome browser, but also into other tools such R in which those feature can be used as efficient filters.

      Sup Table S1 would be much more useful for future researchers if it had a column with the direct accession numbers for the raw sequencing libraries.

      We have included another table which includes direct accession number for ENA as well as numerous other meta data in Sup Table S6 i.e. "Supp_Table_S6_library_ENA_accession"

      Figures showing genome browser snapshots are too small; the text is mostly illegible on screen and when printed. This includes Figure 4 and Figures S1-S5.

      The snapshots have been improved to ensure better readability.

      Lines 67-68: This is unclear to me. Did the authors do Northerns? Please clarify / re-write.

      Thank you for querying this. After much closer inspection of the papers cited by Casas-Mollano et al. as evidence of the 23nt peak the evidence for the 23nt doesn't seem that strong and may even be a mistake on their part. Nonetheless, it is far from a critical piece of information for this paper and we have thus decided to remove this sentence.

      Figure 2B: X-axis label, perhaps change to "number of reads in library" for clarity.

      We agree and have changed it accordingly

      Figure 4 caption: The acronym "CRSL" should be defined.

      CRSL is now been duly defined in the manuscript

      Line 387: Reference #29 (line 509): There is not enough information here to find the data.

      We have used the appropriate bibtex code to reference this Zenodo share (https://zenodo.org/record/3862405/export/hx). The current cite format does somehow omit some information. We hope this will be fixed by the publisher but we have also provided the full DOI address in the “additional information” section just in-case. We will keep an eye on how it comes out.

      Style suggestion on title: What is "secret" about the genome? I didn't really understand that first part of the title. Perhaps consider revision to make it more factual and less literary. Just "A small RNA locus map for Chlamydomonas reinhardtii"?

      Thank you for this suggestion, we have adapted the title to make it more descriptive.

      Reviewer #3

      …the evolutionary implications are not clear. The authors state in the abstract that "These results are consistent with the idea that there was diversification in sRNA mechanisms after the evolutionary divergence of algae from higher plant lineages." Although in the end this may prove to be correct, the only species compared are Arabidopsis thaliana (as representative of land plants) and Chlamydomonas reinhardtii (as representative of green algae). With this very limited information it is not possible to infer the sRNA loci (much less sRNA mechanisms) in an ancestral species. It remains formally possible that an ancestral progenitor species had a greater diversity of sRNA loci that were subsequently lost in a selective manner in specific lineages. Moreover, the diversity of sRNA loci may not correlate strictly with the diversity of the RNAi machinery since, at least some loci, do not appear to be associated with RNAi components such as Dicer or Argonaute.

      Thank you for these insightful comments. As we followed a very similar methodological approach to that used to produce the Arabidopsis sRNA locus map published in Hardcastle et al. (2018), we wanted to take the opportunity to compare the results and build upon the ongoing discussion concerning the evolution of sRNA mechanisms in Chlamydomonas (e.g. Valli et al. 2016). Your point about the possibility of an ancestral progenitor with greater diversity that was then lost is very valid. You are also of course correct about the limitations to what can be concluded from this study and the limited comparisons that can be made. We see our approach as a useful tool for hypothesis generation which can be complemented by more in-depth exploration in the future. With this in mind, and taking on board your comments, we have elaborated on our discussion of the evolutionary implications of our study, which we hope now gives a more balanced account.

      I may have missed it but I could not find a table listing the specific sRNA loci assigned to each of the locus classes. It would be very useful to provide the class annotation of each sRNA locus in order to facilitate future analyses of sRNA biogenesis and function.

      That information was indeed missing, thanks for bringing it up. We have now included this in the gff file (column LC) as well as in another cleaner table (Supp_Table_S7_loci_class_annotation).

      Figures S2 to S5 have the same legend but they correspond to different loci. It would be useful to provide for each locus class, as supplementary figures, two examples of typical sRNA loci.

      Thanks for pointing this out, this was an error on our part, the captions have now been corrected. Unfortunately, due to the ongoing pandemic-related restrictions we were unable to run to get a genome browser session to run to this point to create more loci figures.

      If information is available, the paper would be strengthened by some locus class validation based on features not used to generate the classification.

      Thank you for this suggestion. In fact, not all annotation features were used predictively in the MCA and clustering process, and so these "supplementary" annotations as outlined in supplementary table S3 can provide some cross-validation. With that in mind, we have now included an additional heatmap as a supplementary figure which shows associations for some of these supplementary annotations as well as corresponding explanations in the text. Further validation is provided by the chromosome tracks in figure 9 showing the distinct genomic distributions of each locus cluster despite chromosomal location not being a factor in the clustering.

      Pg 5, line 108. I think you mean "strong bias (0.2 > x > 0.8)."

      Thank you for pointing this out, we have now corrected this error.

      Pg 7, Table 1. Some of the annotation features are obvious but some abbreviations may need clarification using footnotes.

      Table 1 has been replaced by the new Fig 5, annotation/abbreviations should now be more obvious.

      Pg 8, lines 156-157. This sentence is not clear. Additionally, the legends to Figures S2-S5 do not refer to LC2 paragon (CSRL003890).

      Thank you for pointing this out. We have now moved the reference to the paragons to earlier in the section where we introduce the six clusters. We hope this is now clearer.

    1. “Most Americans don’t know very much about Reconstruction, and in many cases what they may think they know is wrong,”

      This stood out to me immediately as I thought back to a question that was posed to us in my previous history class, "Do you think History was written by the winners?"Often times we are quick to only skim the surface of what happened throughout history. There is so much fake news on the internet that we can get confused on what to believe and what not to believe. It is important to fact check our sources, continue to learn, and have an open mind.

    1. Resume and Its Impacts on Your Future JobYunic SolutionsAug 26 · 5 min readResume Writing Services assist employers with settling on recruitment choices and assist you with getting your first meeting. That is the reason it makes a difference in how you structure your resume and what data you choose to incorporate. In this article, you’ll realize why a resume is significant and get noteworthy resume tips that may assist you with accomplishing your next profession move.<img alt="Image for post" class="s t u dt ai" src="https://miro.medium.com/max/590/1*gLu8G5hNr8JOA2cRhnCKbA.png" width="295" height="171" srcSet="https://miro.medium.com/max/552/1*gLu8G5hNr8JOA2cRhnCKbA.png 276w, https://miro.medium.com/max/590/1*gLu8G5hNr8JOA2cRhnCKbA.png 295w" sizes="295px"/>Subsequently, the significance of a resume can never be disparaged. In this way, to establish the main connection, it is basic that your resume stands apart from the group first. 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  3. media.carnegie.org media.carnegie.org
    1. t is obvious that if today’s youngsters cannotread with understanding, think about and analyze what they’ve read, and then write clearly and effectively about what they’ve learned and what they think, then they may never be able to do justiceto their talents and their potential

      As educators, particularly in the English field, we have to think about the broader context of what we are teaching our students. We are not only preparing our students to be better writers, but to be better citizens and better prepare them to think critically and communicate effectively. It is interesting to think about how this perspective can change our approaches to teaching writing.

    1. it on Anti-Semitism and Hate Remarks by Sacha Baron Cohen, Recipient of ADL's International Leadership Award November 21, 2019 <img src="https://www.adl.org/sites/default/files/styles/cropped_img_md/public/2019-11/never-is-now-2019-sacha-baron-cohen-award-800w-543h.jpg?h=6b028cc2&amp;itok=CZMBCXlF" alt="Sacha Baron Cohen gives award speech"/> <!--/*--><![CDATA[/* ><!--*/ .resp-container { position: relative; overflow: hidden; padding-top: 56.25%; } .resp-iframe { position: absolute; top: 0; left: 0; width: 100%; height: 100%; border: 0; } /*--><!]]>*/ Thank you, Jonathan, for your very kind words.  Thank you, ADL, for this recognition and your work in fighting racism, hate and bigotry.  And to be clear, when I say “racism, hate and bigotry” I’m not referring to the names of Stephen Miller’s Labradoodles. Now, I realize that some of you may be thinking, what the hell is a comedian doing speaking at a conference like this!  I certainly am.  I’ve spent most of the past two decades in character.  In fact, this is the first time that I have ever stood up and given a speech as my least popular character, Sacha Baron Cohen.  And I have to confess, it is terrifying. I realize that my presence here may also be unexpected for another reason.  At times, some critics have said my comedy risks reinforcing old stereotypes. The truth is, I’ve been passionate about challenging bigotry and intolerance throughout my life.  As a teenager in the UK, I marched against the fascist National Front and to abolish Apartheid.  As an undergraduate, I traveled around America and wrote my thesis about the civil rights movement, with the help of the archives of the ADL.  And as a comedian, I’ve tried to use my characters to get people to let down their guard and reveal what they actually believe, including their own prejudice. Now, I’m not going to claim that everything I’ve done has been for a higher purpose.  Yes, some of my comedy, OK probably half my comedy, has been absolutely juvenile and the other half completely puerile.  I admit, there was nothing particularly enlightening about me—as Borat from Kazakhstan, the first fake news journalist—running through a conference of mortgage brokers when I was completely naked.  But when Borat was able to get an entire bar in Arizona to sing “Throw the Jew down the well,” it did reveal people’s indifference to anti-Semitism.  When—as Bruno, the gay fashion reporter from Austria—I started kissing a man in a cage fight in Arkansas, nearly starting a riot, it showed the violent potential of homophobia.  And when—disguised as an ultra-woke developer—I proposed building a mosque in one rural community, prompting a resident to proudly admit, “I am racist, against Muslims”—it showed the acceptance of Islamophobia.    That’s why I appreciate the opportunity to be here with you.  Today around the world, demagogues appeal to our worst instincts.  Conspiracy theories once confined to the fringe are going mainstream.  It’s as if the Age of Reason—the era of evidential argument—is ending, and now knowledge is delegitimized and scientific consensus is dismissed.  Democracy, which depends on shared truths, is in retreat, and autocracy, which depends on shared lies, is on the march.  Hate crimes are surging, as are murderous attacks on religious and ethnic minorities. What do all these dangerous trends have in common?  I’m just a comedian and an actor, not a scholar.  But one thing is pretty clear to me.  All this hate and violence is being facilitated by a handful of internet companies that amount to the greatest propaganda machine in history. The greatest propaganda machine in history. Think about it.  Facebook, YouTube and Google, Twitter and others—they reach billions of people.  The algorithms these platforms depend on deliberately amplify the type of content that keeps users engaged—stories that appeal to our baser instincts and that trigger outrage and fear.  It’s why YouTube recommended videos by the conspiracist Alex Jones billions of times.  It’s why fake news outperforms real news, because studies show that lies spread faster than truth.  And it’s no surprise that the greatest propaganda machine in history has spread the oldest conspiracy theory in history—the lie that Jews are somehow dangerous.  As one headline put it, “Just Think What Goebbels Could Have Done with Facebook.” On the internet, everything can appear equally legitimate.  Breitbart resembles the BBC.  The fictitious Protocols of the Elders of Zion look as valid as an ADL report.  And the rantings of a lunatic seem as credible as the findings of a Nobel Prize winner.  We have lost, it seems, a shared sense of the basic facts upon which democracy depends. When I, as the wanna-be-gansta Ali G, asked the astronaut Buzz Aldrin “what woz it like to walk on de sun?” the joke worked, because we, the audience, shared the same facts.  If you believe the moon landing was a hoax, the joke was not funny. When Borat got that bar in Arizona to agree that “Jews control everybody’s money and never give it back,” the joke worked because the audience shared the fact that the depiction of Jews as miserly is a conspiracy theory originating in the Middle Ages. But when, thanks to social media, conspiracies take hold, it’s easier for hate groups to recruit, easier for foreign intelligence agencies to interfere in our elections, and easier for a country like Myanmar to commit genocide against the Rohingya. It’s actually quite shocking how easy it is to turn conspiracy thinking into violence.  In my last show Who is America?, I found an educated, normal guy who had held down a good job, but who, on social media, repeated many of the conspiracy theories that President Trump, using Twitter, has spread more than 1,700 times to his 67 million followers.  The President even tweeted that he was considering designating Antifa—anti-fascists who march against the far right—as a terror organization.   So, disguised as an Israel anti-terrorism expert, Colonel Erran Morad, I told my interviewee that, at the Women’s March in San Francisco, Antifa were plotting to put hormones into babies’ diapers in order to “make them transgender.”  And he believed it. I instructed him to plant small devices on three innocent people at the march and explained that when he pushed a button, he’d trigger an explosion that would kill them all.  They weren’t real explosives, of course, but he thought they were.  I wanted to see—would he actually do it? The answer was yes.  He pushed the button and thought he had actually killed three human beings.  Voltaire was right, “those who can make you believe absurdities, can make you commit atrocities.”  And social media lets authoritarians push absurdities to billions of people. In their defense, these social media companies have taken some steps to reduce hate and conspiracies on their platforms, but these steps have been mostly superficial. I’m speaking up today because I believe that our pluralistic democracies are on a precipice and that the next twelve months, and the role of social media, could be determinant.  British voters will go to the polls while online conspiracists promote the despicable theory of “great replacement” that white Christians are being deliberately replaced by Muslim immigrants.  Americans will vote for president while trolls and bots perpetuate the disgusting lie of a “Hispanic invasion.”  And after years of YouTube videos calling climate change a “hoax,” the United States is on track, a year from now, to formally withdraw from the Paris Accords.  A sewer of bigotry and vile conspiracy theories that threatens democracy and our planet—this cannot possibly be what the creators of the internet had in mind. I believe it’s time for a fundamental rethink of social media and how it spreads hate, conspiracies and lies.  Last month, however, Mark Zuckerberg of Facebook delivered a major speech that, not surprisingly, warned against new laws and regulations on companies like his.  Well, some of these arguments are simply absurd.  Let’s count the ways. First, Zuckerberg tried to portray this whole issue as “choices…around free expression.”  That is ludicrous.  This is not about limiting anyone’s free speech.  This is about giving people, including some of the most reprehensible people on earth, the biggest platform in history to reach a third of the planet.  Freedom of speech is not freedom of reach.  Sadly, there will always be racists, misogynists, anti-Semites and child abusers.  But I think we could all agree that we should not be giving bigots and pedophiles a free platform to amplify their views and target their victims. Second, Zuckerberg claimed that new limits on what’s posted on social media would be to “pull back on free expression.”  This is utter nonsense.  The First Amendment says that “Congress shall make no law” abridging freedom of speech, however, this does not apply to private businesses like Facebook.  We’re not asking these companies to determine the boundaries of free speech across society.  We just want them to be responsible on their platforms. If a neo-Nazi comes goose-stepping into a restaurant and starts threatening other customers and saying he wants kill Jews, would the owner of the restaurant be required to serve him an elegant eight-course meal?  Of course not!  The restaurant owner has every legal right and a moral obligation to kick the Nazi out, and so do these internet companies. Third, Zuckerberg seemed to equate regulation of companies like his to the actions of “the most repressive societies.”  Incredible.  This, from one of the six people who decide what information so much of the world sees.  Zuckerberg at Facebook, Sundar Pichai at Google, at its parent company Alphabet, Larry Page and Sergey Brin, Brin’s ex-sister-in-law, Susan Wojcicki at YouTube and Jack Dorsey at Twitter.  The Silicon Six—all billionaires, all Americans—who care more about boosting their share price than about protecting democracy.  This is ideological imperialism—six unelected individuals in Silicon Valley imposing their vision on the rest of the world, unaccountable to any government and acting like they’re above the reach of law.  It’s like we’re living in the Roman Empire, and Mark Zuckerberg is Caesar.  At least that would explain his haircut. Here’s an idea.  Instead of letting the Silicon Six decide the fate of the world, let our elected representatives, voted for by the people, of every democracy in the world, have at least some say. Fourth, Zuckerberg speaks of welcoming a “diversity of ideas,” and last year he gave us an example.  He said that he found posts denying the Holocaust “deeply offensive,” but he didn’t think Facebook should take them down “because I think there are things that different people get wrong.”  At this very moment, there are still Holocaust deniers on Facebook, and Google still takes you to the most repulsive Holocaust denial sites with a simple click.  One of the heads of Google once told me, incredibly, that these sites just show “both sides” of the issue.  This is madness. To quote Edward R. Murrow, one “cannot accept that there are, on every story, two equal and logical sides to an argument.”  We have millions of pieces of evidence for the Holocaust—it is an historical fact.  And denying it is not some random opinion.  Those who deny the Holocaust aim to encourage another one. Still, Zuckerberg says that “people should decide what is credible, not tech companies.”  But at a time when two-thirds of millennials say they haven’t even heard of Auschwitz, how are they supposed to know what’s “credible?”  How are they supposed to know that the lie is a lie? There is such a thing as objective truth.  Facts do exist.  And if these internet companies really want to make a difference, they should hire enough monitors to actually monitor, work closely with groups like the ADL, insist on facts and purge these lies and conspiracies from their platforms. Fifth, when discussing the difficulty of removing content, Zuckerberg asked “where do you draw the line?”  Yes, drawing the line can be difficult.  But here’s what he’s really saying: removing more of these lies and conspiracies is just too expensive. These are the richest companies in the world, and they have the best engineers in the world.  They could fix these problems if they wanted to.  Twitter could deploy an algorithm to remove more white supremacist hate speech, but they reportedly haven’t because it would eject some very prominent politicians from their platform.  Maybe that’s not a bad thing!  The truth is, these companies won’t fundamentally change because their entire business model relies on generating more engagement, and nothing generates more engagement than lies, fear and outrage.    It’s time to finally call these companies what they really are—the largest publishers in history.  And here’s an idea for them: abide by basic standards and practices just like newspapers, magazines and TV news do every day.  We have standards and practices in television and the movies; there are certain things we cannot say or do.  In England, I was told that Ali G could not curse when he appeared before 9pm.  Here in the U.S., the Motion Picture Association of America regulates and rates what we see.  I’ve had scenes in my movies cut or reduced to abide by those standards.  If there are standards and practices for what cinemas and television channels can show, then surely companies that publish material to billions of people should have to abide by basic standards and practices too. Take the issue of political ads.  Fortunately, Twitter finally banned them, and Google is making changes, too.  But if you pay them, Facebook will run any “political” ad you want, even if it’s a lie.  And they’ll even help you micro-target those lies to their users for maximum effect.  Under this twisted logic, if Facebook were around in the 1930s, it would have allowed Hitler to post 30-second ads on his “solution” to the “Jewish problem.”  So here’s a good standard and practice: Facebook, start fact-checking political ads before you run them, stop micro-targeted lies immediately, and when the ads are false, give back the money and don’t publish them. Here’s another good practice: slow down.  Every single post doesn’t need to be published immediately.  Oscar Wilde once said that “we live in an age when unnecessary things are our only necessities.”  But is having every thought or video posted instantly online, even if it is racist or criminal or murderous, really a necessity?  Of course not! The shooter who massacred Muslims in New Zealand live streamed his atrocity on Facebook where it then spread across the internet and was viewed likely millions of times.  It was a snuff film, brought to you by social media.  Why can’t we have more of a delay so this trauma-inducing filth can be caught and stopped before it’s posted in the first place? Finally, Zuckerberg said that social media companies should “live up to their responsibilities,” but he’s totally silent about what should happen when they don’t.  By now it’s pretty clear, they cannot be trusted to regulate themselves.  As with the Industrial Revolution, it’s time for regulation and legislation to curb the greed of these high-tech robber barons.  In every other industry, a company can be held liable when their product is defective.  When engines explode or seatbelts malfunction, car companies recall tens of thousands of vehicles, at a cost of billions of dollars.  It only seems fair to say to Facebook, YouTube and Twitter: your product is defective, you are obliged to fix it, no matter how much it costs and no matter how many moderators you need to employ. In every other industry, you can be sued for the harm you cause.  Publishers can be sued for libel, people can be sued for defamation.  I’ve been sued many times!  I’m being sued right now by someone whose name I won’t mention because he might sue me again!  But social media companies are largely protected from liability for the content their users post—no matter how indecent it is—by Section 230 of, get ready for it, the Communications Decency Act.  Absurd! Fortunately, Internet companies can now be held responsible for pedophiles who use their sites to target children.  I say, let’s also hold these companies responsible for those who use their sites to advocate for the mass murder of children because of their race or religion.  And maybe fines are not enough.  Maybe it’s time to tell Mark Zuckerberg and the CEOs of these companies: you already allowed one foreign power to interfere in our elections, you already facilitated one genocide in Myanmar, do it again and you go to jail. In the end, it all comes down to what kind of world we want.  In his speech, Zuckerberg said that one of his main goals is to “uphold as wide a definition of freedom of expression as possible.”  Yet our freedoms are not only an end in themselves, they’re also the means to another end—as you say here in the U.S., the right to life, liberty and the pursuit of happiness.  But today these rights are threatened by hate, conspiracies and lies. Allow me to leave you with a suggestion for a different aim for society.  The ultimate aim of society should be to make sure that people are not targeted, not harassed and not murdered because of who they are, where they come from, who they love or how they pray If we make that our aim—if we prioritize truth over lies, tolerance over prejudice, empathy over indifference and experts over ignoramuses—then maybe, just maybe, we can stop the greatest propaganda machine in history, we can save democracy, we can still have a place for free speech and free expression, and, most importantly, my jokes will still work. Thank you all very much. Antisemitism in the US Cyberhate Extremism, Terrorism & Bigotry

      hi

    1. Author Response

      Summary:

      The bacterial ribosome from E. coli has traditionally been a reference model in structural biology. Basic studies in translation and the mode of action and resistance to antibiotics, have greatly benefited from the mechanistic framework derived from structural studies of this cellular machinery. Recently, electron cryo-microscopy has surpassed the resolution limits X-ray crystallography studies of bacterial ribosomes historically reported. In the present manuscript, Watson et al present a landmark work where these limits are pushed even further, reporting a ribosome cryo-EM reconstruction with an overall resolution of 2Å, and even better than that in the best areas of the map. The achieved resolution is impressive and one thus expects major findings, methodological highlights and comparisons with previous structures. However, these could be better developed. Instead, the usage of map-to-model Fourier shell correlation (already known in the field) is stressed to estimate the resolution, but it is not clear what the advantage is here as the values are the same when estimated from half map FSCs. Therefore, it is suggested that the discussion about the model-to-map FSC is toned down considerably in (or even removed), while adding in more information about the new findings in the map, along the lines of the comments below.

      We thank the reviewers for their interest in this work, and for their helpful comments on the first version of the manuscript. We provide responses to the individual points below.

      Reviewer #1:

      This paper describes a 2A cryo-EM reconstruction of the E.coli 70S ribosome. This structure represents the highest resolution ribosome structure, by any method, available thus far and highlights interesting modifications that were not possible to see in previous structures. I'll let the ribosome experts comment on the relevance of these and focus my review on the cryo-EM technical parts. The paper is clearly written and the figures are informative and beautiful.

      The first author is particularly gratified that the figures were well received.

      Major comments:

      1) The authors make a big deal out of resolution assessment by model-to-map FSCs. It is unclear to me why they do this. First of all, model-to-map FSC is not a new resolution measure: it is in widespread use already. Second, it is unclear why the authors are so forceful in stating that it is better than the half-map FSC. They say "While map-to-model FSC carries intrinsic bias from the model's dependence on the map, in a high resolution context it does provide additional information about the overall confidence with which to interpret the model, not captured in half-map FSCs." What additional information does it provide? I would say it only provides true additional information if the atomic model comes from another experiment! In the way it is used here: by refining the model inside the very same map, there is a danger of increasing model-to-map FSC values through overfitting of the model (see also below). This danger is not recognized enough in the text (it is only hinted at in the sentence above), and overfitting is not measured explicitly for this case. Yes, half-map FSC measures self-consistency, but in practical terms (when done right!), this doesn't matter for the resolution estimate. The same is true for model-to-map FSCs: when done right they convey the right information, but the danger of self-consistency (through overfitting) also exists here. As the paper is mainly about the high-resolution ribosome structure, and no proper evaluation of the relative merits of half-map FSC versus model-to-map FSC is performed, I would suggest that the authors remove (or at least tone down considerably) their statements about resolution assessment from the manuscript.

      All three reviewers commented on our emphasis on using the map-to-model FSC criterion. We thank the reviewers for pointing out our motivation to discuss FSC metrics was not clear. We agree with the reviewers that the map-to-model FSC metric has been available for some time. However, in the ribosome field, the half-map FSC is still very commonly used as the sole resolution-dependent metric, including in recent literature that we cited (Nürenberg-Goloub, 2020; Tesina, 2020; Stojković, 2020; Pichkur, 2020; Halfon, 2019), as well as in a newer publication (Loveland, 2020, Nature, https://doi.org/10.1038/s41586-020-2447-x ). We mention some of the shortcomings of half-map FSC, which the third reviewer alludes to in their comment on “intense debate” in the field. While it is acknowledged as best practice to examine both maps and models, many visitors to the PDB likely will download only the model. Therefore we find it prudent to communicate confidence in the model resolution and not just the half-maps, particularly in this resolution regime. Again, this is not common in recent ribosome literature, which we will clarify in the Discussion. We will make changes throughout the manuscript to streamline and clarify our discussion of the two metrics, including an additional comparison to a newly released ribosome structure, as detailed below.

      When we discuss “additional information provided by map-to-model FSC,” we recognize that there may be semantic issues with the word “information” as map-to-model FSC depends on the same information content of the maps. However, the map-to-model FSC provides new information about the model quality to the reader. While half-map FSC tells us something about the best model one might achieve, new practical information lies in the authors’ handling of the model, which will vary among individuals (as discussed further below). Furthermore, model refinement procedures leverage well-defined chemical properties (i.e. bond lengths, angles, dihedrals, and steric restraints) that the map “knows” nothing about, which has value for keeping the realism in check. This is also why we originally included the sentence, “Sub-Ångstrom differences in nominal resolution as reported by half-map FSCs have significant bearing on chemical interactions at face value but may lack usefulness if map correlation with the final structural model is not to a similar resolution.” We will rewrite portions of this section for clarity.

      Comparisons to other recent high-resolution cryo-EM ribosome structures show discrepancies in the reported half-map FSC and map-to-model FSC calculated by us (see beginning of section “High-resolution structural features of the 50S ribosomal subunit”), with the map-to-model FSC values being to lower resolution. These structures report half-map FSCs only, which we could not replicate because of unavailability of half-maps, but we describe our calculation of map-to-model FSC with their deposited maps. We did not explicitly highlight the comparisons with their reported half-map FSC resolutions in the original manuscript, and we will include further discussion to more clearly communicate our point. We will also include another comparison to the newly released structure by Pichkur et al. (Pichkur, 2020) which has become available during the review process and is the closest to our map resolution. The map-to-model FSC with their model and map yields 2.29 Å resolution, while a simple rigid-body fit of our model into their map without further adjustment yields 2.07 Å. This difference highlights the practical insufficiency of focusing only on half-map FSC and the value of our model as a reference for future work.

      2) To test for the presence of overfitting their atomic models in the maps, the authors should shake-up the atomic models and refine them in the first independently refined half-map. The FSC of that model versus that half-map (FSC_work) should be compared with the FSC of that very same model versus the second half-map (FSC_test). Deviations between the two would be an indicating of overfitting. If that were to be observed, the weights on the stereochemical restraints should be tightened until the overfitting disappears. The same weighting scheme should then be used for the final model refinement against the sum of the half-maps.

      In lieu of what the reviewers have suggested, we think the additional map-to-model comparison of our model rigid-body docked into the 2.1 Å 50S map by Pichkur et al. provides reasonable evidence that our model suffers from minimal overfitting. Without any additional refinement of our model into their map, the map-to-model FSC resolution is 2.07 Å. We will include the new comparison in the revised manuscript.

      For model refinement, we used default parameters for phenix.real_space_refine, which internally optimizes weights for hundreds of different “chunks” during the refinement. This “black box” aspect does not give us facile control over the weighting scheme. However, we also note that the final model is not “fresh” out of Phenix; rather, the macromolecules have been meticulously reviewed and adjusted manually in Coot, with blurred maps to aid in accurate modeling for areas that are not as well connected/resolved. RSR in Coot was also required to “stitch” sections of the model together, since the models were refined in multiple focus-refined maps. Further, we think that for models that are ⅔ RNA, manually optimizing the Ramachandran restraints is unlikely to provide much new insight into RSR of this structure.

      3) Figure 1 -supplement 7: if radiation damage breaks the ribose rings, they should still be OK during early movie frames. This could be investigated by performing per-frame (or per-few-frames) reconstructions. The radiation damage argument would be a lot stronger if the density is present in early frames, yet disappears in the later ones. There will be a balance between dose-resolution and achievable spatial-resolution to see this of course. But it may be worth investigating.

      This is a great suggestion, and we have now carried out this analysis. We have performed the early-frame reconstruction and now have an alternative hypothesis that may make more sense. We will include the alternative hypothesis that we are likely seeing disorder due to conformational flexibility in the RNA backbone, rather than radiation damage, which seems unlikely given the features in the early-frame map. We will also update Figure 1–figure supplement 7 with new panels to aid this discussion.

      Reviewer #2:

      The manuscript by Watson et al. presents the structural analysis of a bacterial ribosome at high resolution. The achieved resolution is impressive and one thus expects major findings, methodological highlights and comparisons with previous structures. However, these are missing or not well developed. Instead, the usage of map-to-model Fourier shell correlation (already known in the field) is stressed to estimate the resolution, but it is not clear what this actually brings here as the values are the same when estimated from half map FSCs. The structure visualizes chemical modifications of ribosomal RNA and amino acids and water molecules, which together are interesting and important. However, here one would expect a comparison with structures of previously analyzed bacterial ribosomes, e.g. E. coli and T. thermophilus, e.g. from the same group and from the work by Fischer et al., Nature 2015: how far are the sites conserved? How do the maps compare? Are the same features seen? It is surprising to see that the main chemical modifications are not discussed and shown (only summarized in the Suppl. Data). Pseudo-uridines are mentioned, but how were these identified? It should be mentioned here that due to their isomeric nature these can be discussed only from their typical hydrogen bond pattern. The paper discusses new sites with chemical modifications, but this could benefit from a more thorough discussion of existing biochemical data or from including new biochemical characterization. The structural role of these modifications is not much described. The side chain of IAS119 has no density, hence one should be careful in interpreting an isomerization of this residue, not sure whether the data allow the conclusions to be made. Similar for the mSAsp89 residue for which the density is uncertain, hence it is not clear whether the conclusions stay on a safe ground.

      We thank the reviewer for their interest in this work. We addressed our emphasis on the map-to-model FSC in response to reviewer #1.

      For the majority of rRNA modifications, we included the supplementary figure as a reference for comparison to the published 4YBB and 4Y4O maps and models. These modifications have been extensively described in the structural biology literature, including in the recent cryo-EM study of the 50S ribosomal subunit (Stojković, 2020) and warrant no detailed comment by us at this time. Instead, we focused on new features that were not previously observed, such as hypomodifications and new modifications. The new modifications are the isoAsp observed in uS11 and the thioamide modification in uL16.

      IAS119 modeling in uS11: We thoroughly analyzed Asn or isoAsp modeled at this residue, and will provide additional evidence that isoAsp is correctly modeled at residue 119. In the original maps, although the side chain density is weak, the backbone density is unequivocal. There is clear density for the extra methylene group (marked with an asterisk in Fig. 4A). We have now calculated a map of the 30S subunit using the first three frames in the image stacks corresponding to a ~3 electron/Å2 dose. In this map, the side chain of isoAsp is more clearly visible (we will include a new figure panel with this density in the supplement). In addition to visual inspection, PHENIX provides a quantitative measure of the fit that also rules out Asn at this position. As we noted in the Methods, “Initial real-space refinement of the 30S subunit against the focused-refined map using PHENIX resulted in a single chiral volume inversion involving the backbone of N119 in ribosomal protein uS11, indicating that the L-amino acid was being forced into a D-amino acid chirality, as reported by phenix.real_space_refine.” Of the 10,564 chiral centers in the 30S subunit, only that for N119 stands out, having an energy residual nearly 2 orders of magnitude larger than the next highest deviation. This stereochemical problem was resolved by modeling isoAsp at this position. We will add these refinement details to the Methods.

      Furthermore, as we noted in the manuscript, isoAsp has been identified in E. coli uS11 by biochemical means (see David, 1999). We examined the phylogenetic conservation of the neighboring sequences in uS11, finding that the N is nearly universal in bacteria and organelles, and D is nearly universal in archaea and eukaryotes (Figure 4 and original Figure 4–figure supplement 1). Finally, even in lower-resolution maps of the archaeal and eukaryotic ribosomes, we find that isoAsp better fits the density, visually with respect to the backbone, and quantitatively based on correlations between RSR models and the density (original Figure 4–figure supplement 2). We therefore think we have been careful in interpreting the isoAsp in uS11, structurally, phylogenetically, and in light of available biochemical evidence. We also provided an in-depth analysis of the neighboring 16S/18S rRNA residues that are in intimate contact with the isoAsp119 region of uS11. See Figure 4B and Supplementary Table 2 and accompanying description.

      mSAsp89: Density for mSAsp89 has been seen previously in the X-ray crystal structure of the 70S ribosome (Noeske, 2015). Here, we also see density for mSAsp89 at lower contour levels. See Figure 1–figure supplement 5. We should have noted in the legend of this panel that we used a lower contour level for mSAsp89 and m7G527, to reveal the modifications. This will be added. Notably, at higher contours that still enclose the standard nucleobase and amino acid side chains, we do not see clear density for the mSAsp89 and m7G527 modifications, in Figure 1–figure supplement 6. In the section of the manuscript covering hypomodifications in RNA, we will clarify this point.

      Pseudouridines: We will clarify how pseudouridines are inferred in the main text. These can be inferred if a solvent molecule or other polar atom is within hydrogen-bonding distance of the N3 in pseudouridine (would be C5 in uridine). We will update Figure 1–figure supplement 5 to better show solvent molecules within hydrogen bonding distance of pseudouridine N3 atoms.

      From a methodological point of view it would be interesting to discuss in more detail how this high resolution structure was obtained, what the specific aspects of high-resolution data collection were and which were the important parameters to refine the structure. Also, how were the thousands of water molecules validated? Regarding the discussion on electrostatic potentials, in contrast to what might be intuitive, the contribution of electron scattering is actually stronger at medium resolution, i.e. its effect does not need high resolution per se. The discussion on radiation damage is a hypothesis at this stage and should be done more carefully including processing of the data using less electron dose (see detailed points below). Taken together, this work describes some interesting findings, but some remain unclear in the discussion because for some no biochemical data are available yet. However, this analysis provides useful hints to design future experiments. Also, there are no developments of tools in this paper in contrast to what is stated.

      We will add some additional information to the Discussion and Methods. In terms of the water molecules, we have not gone through these one by one at this point. We actually do not claim to have introduced new tools, but we note that our water modeling spurred the incorporation of phenix.douse into the latest PHENIX releases. This will be more clearly stated, and we will acknowledge Pavel Afonine for helping us as he developed this functionality. (He indicated we should cite Liebschner, 2019.) Solvent modeling is ripe for future development, as we note in the Discussion.

      Although scattering is stronger at medium resolution, it is not absent at < 2 Å. See the recent atomic-resolution structures of ferritin for examples. In fact, we have now examined the 2.1 Å map deposited by Pichkur et al. (Pichkur, 2020), in which the thioamide is barely visible. The thioamide in the 2.2 Å map deposited by Stojković (Stojković, 2020) is not obviously visible. We will add panels showing this in the revised manuscript.

      We have now used the early frames to address the question of ribose damage and the carboxylate of IAS119 in uS11, as noted above.

      Reviewer #3:

      The bacterial ribosome from E.coli has traditionally been a reference model in structural biology. Basic studies in translation and the mode of action and resistance to antibiotics, have greatly benefited from the mechanistic framework derived from structural studies of this cellular machinery. Recently, electron cryo-microscopy has surpassed the resolution limits X-ray crystallography studies of bacterial ribosomes historically reported. In the present manuscript, Watson et al present a landmark work where these limits are pushed even further, reporting a ribosome cryoEM reconstruction with features compatible with a resolution in the range of overall 2Å and below that resolution in the best areas of the map. With this level of detail, a chemical interpretation of many and fundamental aspects of translation and antibiotic interaction can be discerned in physicochemical terms, greatly improving our understanding of this key component of bacterial cells. The manuscript is well presented with clear evidence supporting the author's claims and interpretations. Specially remarkable is the detailed and accurate handling of the reference list.

      We thank the reviewer for their interest in our work. In the revision, we will keep the references mostly as-is, but will add a few based on the revisions we need to make.

      Mayor concern:

      There is an intense debate within the cryoEM community regarding which is the best way to estimate the resolution of a cryoEM reconstruction. In this manuscript, the authors claim map-to-model FSC values could "in a high resolution context [...] provide additional information about the overall confidence with which to interpret the model, not captured in half-map FSCs." Regardless of the opinion of this reviewer about this specific point, if a map-to-model FSC is to be used as a claim of "high-resolution" a convincing overfitting test proving the absence of overfitting in the refined model should be presented. Otherwise, map-to-model FSC values may be artificially high due to unrealistic deformation of the model. The authors thus, should prove their refined model is not overfitted.

      This was a concern of all the reviewers, which we addressed above. We think the comparisons to other recent structures, especially the 2.1 Å 50S map by Pichkur et al., makes the case for using the map-to-model FSC criterion.

    1. Background The COVID-19 pandemic has unprecedented consequences on population health, with governments worldwide issuing public health directives which have major impacts on normal living. In the absence of a vaccine, a key way to control the pandemic is through behavioural change: people adhering to transmission-reducing behaviours (TRBs), such as physical distancing, regular hand washing, and wearing face covering, especially when physical distancing is difficult. The application of behavioural science is central to understanding factors that influence adherence to TRBs. Non-adherence may be explained by theories of how people think about the illness (the common-sense model of self-regulation) and/or how they think about the TRBs (social cognition theory and protection motivation theory). In addition, outbreaks of infectious diseases and the measures employed to curb them are likely to have detrimental effects on people’s mental and general health. Therefore, in representative repeated surveys we will apply behavioural theories to model adherence to TRBs, explain variations in adherence, and the effects on mental and general health in the Scottish population from June to November 2020, following the initial outbreak of the COVID-19 pandemic. Methods Repeated 20-minute structured telephone surveys will be conducted with nationally representative random samples of 500 adults from throughout Scotland. The first 6 weeks the survey will be conducted weekly, thereafter fortnightly, for a total of 14 waves (total n=7000). Ipsos MORI will recruit participants through random digit dialling. The core survey will measure adherence to TRBs, mental and general health, and explanatory variables from the theories. Further questions will be added, enabling more detailed measurement of constructs in the core survey, additional themes, and questions that align with the evolving pandemic. Discussion This study will provide insights into the link between (changes in) adherence to TRBs and explanatory factors, and their effects on mental and general health, including event-related changes (e.g., when government directives change). Governments and other decision-makers may use these findings to tailor public health promotion, target specific population groups, and develop behaviourally informed interventions over the duration of the COVID-19 pandemic (along with any subsequent equivalent events) to protect health and limit the spread of COVID-19.
    1. Religion and science are two major sources of knowledge. Some accounts suggest that religious belief inhibits people from trusting scientific information, and encourages conflict between religion and science. We draw from theories of human motivation to challenge this claim, instead suggesting that religious people perceive less conflict between science and religion than non-religious people, that religious—but not non-religious—people use both science and religion when they explain phenomena, and that religious people rely on science more than non-religious people think they do. Five studies support our account. A pilot study uses a large representative sample of Americans to show that religious people perceive less conflict between science and religion than non-religious people. Studies 1-2 show that religious people view religion and science as equally and moderately instrumental for explaining extraordinary events (Study 1) and life’s “big questions” (Study 2), whereas non-religious people view science as highly instrumental and religion as not at all so. Study 3 finds that non-religious people mischaracterize religious people as more reliant on religion and less reliant on science than they really are, and also suggests that religious people view science and religion as orthogonal whereas non-religious people view them as hydraulic. Study 4 applies these findings to the COVID-19 pandemic, showing that faith-based strategies of avoiding infection do not inhibit adoption of science-based strategies. Religious people may be more open to science than many non-religious people think.
    1. To talk about what happens in classrooms, let alone in children’s heads, as moving forward or backward in specifiable degrees, is not only simplistic because it fails to capture much of what is going on, but also destructive because it may change what is going on for the worse. 

      I think each class has different types of students,for example some children are quiet,but some children are active and playful,but all of them are still in their developmental age, so we just need advice and education,everything will be fine

    1. Thus we have a basic distinction between fantasy and science fiction

      Here, one cannot help but think about the fact that we as humans do not, and likely will not ever, know what is truly science and what is supernatural. The argument that we must separate the two because "bringing experiences [of both] is to destroy one or the other, is not made because the universe cannot handle both— it is because we are humans are incapable of handling both, and of realizing that there is no line that separates the two. The supernatural, in many ways, may just be the parts of science that we have not figured out yet, and separating them is easier for us as people to handle.

    1. Asking them to assign themselves grades, would challenge the dominant power of teacher as the almighty transmitter of knowledge which is consumed by students and scribed into a grade.

      I think students are very much aware of this power. That is often why we under-grade ourselves when given the option to self grade. Even though we know we put in the effort and time we still feel as though we don't have the authority to give ourselves that A. In our minds we may be thinking “only a teacher could be truly unbiased and determine if this is A work or not”. That is why also having classmates grade you as well is good. They may confirm what the teacher thinks of your work. If your group members are giving you A’s as well, perhaps that boosts your confidence when you self-grade the next time.

    1. How do we experience border-crossing? Alter-natively, what does it feel like to exist as a border – as, for example,unwelcome migrants and minority groups are forced to do?

      I think this question is particularly interesting question to pose as the answer would look different for the person you ask (even currently) from those from different periods of time. Meaning if we went way back into monarchism—those individuals would likely say the stone wall around their kingdom (potentially). Currently, there isn't necessaryily a need for a physical border wall (unless you are certain people) because there, arguably, aren't really any borders in modern civilization. Most countries allow people to travel to their country and there isn't a physical border separating them rather a sort of marker. Another interesting concept this begins to dip into, is the idea of ideologies. Even though there is a "border" separating different countries—is there a border separating ideologies that may be similar even though they exist in different countries.

    1. Note: This rebuttal was posted by the corresponding author to Review Commons. Content has not been altered except for formatting.

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      Reply to the reviewers

      Reviewer1

      Reviewer #1 (Evidence, reproducibility and clarity (Required)):

      The manuscript is clearly written and the figures appropriate and informative. Some descriptions of data analyses are a little dense but reflect what would appear long hard efforts on the part of the authors to identify and control for possible sources of misinterpretation due to sensitivities of parameters in their fitness model. The authors efforts to retest interactions under non-competition conditions allay fears of most concerns that I would have. One problem though that I could not see explicitly addressed was that of potential effects of interactions between methotrexate and the other conditions and how this is controlled for. Specifically, I could be argued that the fact that a particular PPI is observed under a specific condition could have more to do with a synthetic effect of treatment of cells with a drug plus methotrexate. Is this controlled for and how? I raise this because in a chemical genetic screen for fitness it was shown that methotrexate is particularly promiscuous for drug-drug interactions (Hillenmeyer ME ,et al. Science 2008). I tried to think of how this works but couldn't come up with anything immediately. I'd appreciate if the authors would take a crack at resolving this issue. Otherwise I have no further concerns about the manuscript.

      We thank the reviewer for the kind comments. We agree with the reviewer’s point that methotrexate could be interacting with drugs or other perturbagens, similar to how the chosen nitrogen source, carbon source, or other growth conditions may interact with a drug. However, the methotrexate concentration is held constant across all conditions, as is the rest of the media components such as the nitrogen and carbon source (with the exception of the raffinose perturbation). Any interactions with methotrexate, or other media components, is undetectable without systematically varying all components for all stressors. Therefore, we use the typical experimental design of measuring molecular variation from a reference, holding invariant media components (such as methotrexate, glucose, or vitamins) fixed between conditions. This is a general practice, and we describe that every condition contains methotrexate on page 3, line 10.

      The library was grown under mild methotrexate selection in 9 environments for 12-18 generations in serial batch culture, diluting 1:8 every ~3 generations, with a bottleneck population size greater than 2 x 109 cells (Table S1).

      We also list the full details of each environment in Table S1.

      Reviewer #1 (Significance (Required)):

      Lui et al expand on previous work from the Levy group to explore a massive in vivo protein interactome in the yeast S. cerevisiae. They achieve this by performing screens cross 9 growth conditions, which, with replication, results in a total of 44 million measurements. Interpreting their results based on a fitness model for pooled growth under methotrexate selection, they make the key observation that there is a vastly expanded pool of protein-protein interactions (PPI) that are found under only one or two condition compared to a more limited set of PPI that are found under a broad set of conditions (mutable versus immutable interactors). The authors show that this dichotomy suggests some important features of proteins and their PPIs that raise important questions about functionality and evolution of PPIs. Among these are that mutable PPIs are enriched for cross-compartmental, high disorder and higher rates of evolution and subcellular localization of proteins to chromatin, suggesting roles in gene regulation that are associated with cellular responses to new conditions. At the same time these interactions are not enriched for changes in abundance. These results are in contrast to those of immutable PPIs, which seem to form a core background noise, more determined by changes in abundance than what the authors interpret must be post-translational processes that may drive, for instance, changes in subcellular localization resulting in appearance of PPIs under specific conditions. The authors are also able to address a couple of key issues about protein interactomes, including the controversial Party-date Hub hypothesis of Vidal, in which they could now affirm support for this hypothesis based on their results and notably negative correlation of PPIs to protein abundance for mutable PPIs. Finally, they also addressed the problem of predicting the upper limit of PPIs in yeast, showing the remarkable results that it may be no more than about 2 times the number of proteins expressed by yeast. Such an upper limit is profoundly important to modelling cellular network complexity and, if it holds up, could define a general upper limit on organismal complexity.

      This manuscript is a very important contribution to understanding dynamics of molecular networks in living cells and should be published with high priority.

      Reviewer 2

      Reviewer #2 (Evidence, reproducibility and clarity (Required)):

      Report on Liu et al. "A large accessory protein interactome is rewired across environments"

      Liu et al. use a mDHFR-based, pooled barcode sequencing / competitive growth / mild methotrexate selection method to investigate changes of PPI abundance of 1.6 million protein pairs across different 9 growth conditions. Because most PPI screens aim to identify novel PPIs in standard growth conditions, the currently known yeast PPI network may be incomplete. The key concept is to define immutable" PPIs that are found in all conditions and "mutable" PPIs that are present in only some conditions.

      The assay identified 13764 PPIs across the 9 conditions, using optimized fitness cut offs. Steady PPI i.e. across all environments, were identified in membrane compartments and cell division. Processes associated with the chromosome, transcription, protein translation, RNA processing and ribosome regulation were found to change between conditions. Mutable PPIs are form modules as topological analyses reveals.

      Interestingly, a correlation on intrinsic disorder and PPI mutability was found and postulated as more flexible in the conformational context, while at the same time they are formed by less abundant proteins.

      I appreciate the trick to use homodimerization as an abundance proxy to predict interaction between heterodimers (of proteins that homodimerize). This "mass-action kinetics model" explains the strength of 230 out of 1212 tested heterodimers.

      A validation experiment of the glucose transporter network was performed and 90 "randomly chosen" PPIs that were present in the SD environment were tested in NaCl (osmotic stress) and Raffinose (low glucose) conditions through recording optical density growth trajectories. Hxt5 PPIs stayed similar in the tested conditions, supported by the current knowledge that Hxt5 is highly expressed in stationary phase and under salt stress. In Raffinose, Hxt7, previously reported to increase the mRNA expression, lost most PPIs indicating that other factors might influence Hxt7 PPIs.

      **Points for consideration:**

      *) A clear definition of mutable and immutable is missing, or could not be found e.g. at page 4 second paragraph.

      We thank the reviewer for pointing this out. We have now added better definition of mutable and immutable on line 19 page 4:

      We partitioned PPIs by the number of environments in which they were identified and defined PPIs at opposite ends of this spectrum as “mutable” PPIs (identified in only 1-3 environments) and “immutable” (identified in 8-9 environments).

      *) Approximately half of the PPIs have been identified in one environment. Many of those mutable PPIs were detected in the 16{degree sign}C condition. Is there an explanation for the predominance of this specific environment? What are these PPIs about?

      The reviewer is correct that ~40% of the PPIs identified in only one environment were found in the 16 ℃ environment. One reason for this could be technical: the positive predictive value (PPV) is the lowest amongst the conditions (16 ℃: 31.6%, mean: 57%, Table SM6). It must be noted, however, that PPVs are calculated using reference data that has generally been collected in standard growth conditions. So, it might be expected that the most divergent environment from standard growth conditions (resulting in the most differences in PPIs) would result in a lower PPV in our study even if the true frequency of false positives was equivalent across environments. We have attempted to be transparent about the quality of the data in each environment by reporting PPVs and other metrics in Table SM6. However, we suspect that the large number of PPIs unique to 16 ℃ is due in part to the fact that it causes the largest changes in the protein interactome, and believe that it should be included, even at the risk of lowering the overall quality of the data. The main reason for this is that this data is likely to contain valuable information about how the cell copes with this stress. For example, we find, but do not highlight in the manuscript, that 16 ℃-specific PPIs contain two major hubs (DID4: 285 PPIs involved in endocytosis and vacuolar trafficking, and DED1: 102 PPIs involved in translation), both of which are reported to be associated with cold adaptation in yeast (Hilliker et al., 2011; Isasa et al., 2015).

      To assess whether the potentially higher false-positive rate in 16 ℃ could be impacting our conclusions related to PPI network organization and features of immutable and mutable PPIs, we repeated these analyses leaving out the 16 ℃ data and found that our main conclusions did not change. This new analysis is now presented in Figure S8 and described on page 5, line 10.

      Finally, we used a pair of more conservative PPI calling procedures that either identified PPIs with a low rate of false positives across all environments (FPR

      We have also added references to other panels in Figure S8 throughout the manuscript, where appropriate.

      *) 50 % overall retest validation rate is fair and reflects a value comparable to other large-scale approaches. However what is the actual variation, e.g. between mutable PPIs and immutable or between condition. e.g. at 16{degree sign}C.

      We validated 502 PPIs present in the SD environment and an additional 36 PPIs in the NaCl environment. As the reviewer suggests, we do indeed observe differences in the validation rate across mutability bins. This data is reported in Figures 3B and S6B, and we use this information to provide a confidence score for each PPI on page 5, line 4.

      To better estimate how the number of PPIs changes with PPI mutability, we used these optical density assays to model the validation rate as a function of the mean PPiSeq fitness and the number of environments in which a PPI is detected. This accurate model (Spearman's r =0.98 between predicted and observed, see Methods) provided confidence scores (predicted validation rates) for each PPI (Table S5) and allowed us to adjust the true positive PPI estimate in each mutability bin. Using this more conservative estimate, we still found a preponderance of mutable PPIs (Figure S6E).

      The validation rate in NaCl is similar to SD (39%, 14/36), suggesting that validation rates do not vary excessively across environments. Because validation experiments are time consuming (we performed 6 growth experiments per PPI), performing a similar scale of validations in all environments as in SD would be resource intensive. Insead, we report a number of metrics (true positive rate, false positive rate, positive predictive value) in Table SM6 using large positive and random reference sets. We believe these metrics are sufficient for readers to compare the quality of data across environments.

      *) What is the R correlation cutoff for PPIs explained in the mass equilibrium model vs. not explained?

      We do not use an R correlation cutoff to assess if a PPI is explained by the mass-action equilibrium model. We instead rely on ordinary least-squares regression as detailed in the methods on page 68, line 13.

      ...we used ordinary least-squares linear regression in R to fit a model of the geometric mean of the homodimer signals multiplied by a free constant and plus a free intercept. Significantly explained heterodimer PPIs were judged by a significant coefficient (FDR 0.05, single-test). This criteria was used to identify PPIs for which protein expression does or does not appear to play as significant of a role as other post-translational mechanisms.

      The first criterion identifies a quantitative fit to the model of variation being related. The second criterion is used to filter out PPIs for which the relationship appears to be explained by more than just the homodimer signals. This approach is more stringent, but we believe this is the most appropriate statistical test to assess fit to this linear model.

      *) 90 "randomly chosen" PPIs for validation. It needs to be demonstrated that these interaction are a random subset otherwise is could also mean cherry picked interactions.

      We selected 90 of the 284 glucose transport-related PPIs for validation using the “sample” function in R (replace = FALSE). We have now included text that describes this on page 63, line 3 in the supplementary methods:

      Diploids (PPIs) on each plate were randomly picked using the “sample” function in R (replace = FALSE) from PPIs that meet specific requirements.

      *) Figure 4 provides interesting correlations with the goal to reveal properties of mutable and less mutable PPIs. PPIs detected in the PPIseq screen can partially be correlated to co-expression (4A) as well as co-localization. Does it make sense to correlate the co-expression across number of conditions? Are the expression correlation condition specific. In this graph it could be that expression correlation stems from condition 1 and 2 and the interaction takes place in 4 and 5 still leading to the same conclusion ... Is the picture of the co-expression correlation similar when you simply look at individual environments like in S4A?

      We use co-expression mutual rank scores from the COXPRESdb v7.3 database (Obayashi et al., 2019). These mutual rank scores are derived from a broad set of 3593 environmental perturbations that are not limited to the environments we tested here. By using this data, we are asking if co-expression in general is correlated with mutability and report that it is in Figure 4A. We thank the reviewer for pointing out that this was not clear and have now added text to clarify that the co-expression analysis is derived from external data on page 6, line 7.

      We first asked whether co-expression is indeed a predictor of PPI mutability and found that it is: co-expression mutual rank (which is inversely proportional to co-expression across thousands of microarray experiments) declined with PPI mutability (Figures 4A and S11) (Obayashi and Kinoshita, 2009; Obayashi et al., 2019).

      The new figure S11 examines how the co-expression mutual rank changes with PPI mutability for PPIs identified in each environment, as the reviewer suggested. For each environment, we find the same general pattern as in Figure 4A (which considers PPIs from all environments).

      *) Figure 4C: Interesting, how dependent are the various categories?

      It is well known that many of these categories are correlated (e.g. mRNA expression level and protein abundance, and deletion fitness effect and genetic interaction degree). However, we believe it is most valuable to report the correlation of each category with PPI mutability independently in Figures 4C and S12, since similar correlations with related categories provide more confidence in our conclusions.

      *) Figure 4 F: When binned in the number of environments in which the PPI was found, the distribution peaks at 6 environments and decreases with higher and lower number of environments. The description /explanation in the text clearly says something else.

      We reported on page 7, line 15:

      We next used logistic regression to determine what features may underlie a good or poor fit to the model (Figure S14C) and found that PPI mutability was the best predictor, with more mutable PPIs being less frequently explained (Figure 4F). Unexpectedly, mean protein abundance was the second best predictor, with high abundance predicting a poor fit to the model, particularly for less mutable PPIs (Figure S14D and S14E).

      As the reviewer notes, Figure 4F shows that the percent of heterodimers explained by the model does appear to decrease for PPIs observed in the most environments. We suspect that the reviewer is correct that something more complicated is going on. One possibility is that extraordinarily stable PPIs (stable in all conditions) would have less quantitative variation in protein or PPI abundance across environments. If this is true, it would be statistically difficult to fit the mass action kinetics model for these PPIs (lower signal relative to noise), thereby resulting in the observed dip.

      A second possibility is that multiple correlated factors are associated with contributing positively or negatively to a good fit, and the simplicity of Figure 4F or a Pearson correlation does not capture this interplay. This second possibility is why we used multivariate logistic regression (Figure S14C) to dissect the major contributing factors. In the text quote above, we report that high abundance is anti-correlated with a good fit to the model (S14D, S14E). Figure 4C shows that immutable PPIs tend to be formed from highly abundant proteins. One possible explanation is that highly abundant proteins saturate the binding sites of their binding partners, breaking from the assumptions of mass action kinetics model. We have now changed the word “limit” to “saturate” on page 7, line 22 to make this concept more explicit.

      Taken together, these data suggest that mutable PPIs are subject to more post-translational regulation across environments and that high basal protein abundance may saturate the binding sites of their partners, limiting the ability of gene expression changes to regulate PPIs.

      A third possibility is that the dip is simply due to noise. Given the complexity of the possible explanations and our uncertainty about which is more likely, we chose to leave this description out of the main text and focus on the major finding: that PPIs detected in more environments are generally associated with a better fit to the mass action kinetics model.

      *) Figure 6: I apologize, but for my taste this is not a final figure 6 for this study. Investigation of different environments increases the PPI network in yeast, yes, yet it is very well known that a saturation is reached after testing of several conditions, different methods and even screening repetition (sampling). It does not represent an important outcome. Move to suppl or remove.

      We included Figure 6 to summarize and illustrate the path forward from this study. This is an explicit reference to impactful computational analyses done using earlier generations of data to assess the completeness of single-condition interaction networks (Hart et al., 2006; Sambourg and Thierry-Mieg, 2010). Here, we are extending PPI measurement of millions-scale networks across multiple environments, and are using this figure to extend these concepts to multi-condition screens. We agree that the property of saturation in sampling is well known, but it is surprising that we can quantitatively estimate convergence of this expanded condition-specific PPI set using only 9 conditions. Thus, we agree with Reviewer 1 that these are “remarkable results” and that the “upper limit is profoundly important to modelling cellular network complexity and, if it holds up, could define a general upper limit on organismal complexity.” We think this is an important advance of the paper, and this figure is useful to stimulate discussion and guide future work.

      Reviewer #2 (Significance (Required)):

      Liu et al. increase the current PPI network in yeast and offer a substantial dataset of novel PPIs seen in specific environments only. This resource can be used to further investigate the biological meaning of the PPI changes. The data set is compared to previous DHFR providing some sort of quality benchmarking. Mutable interactions are characterized well. Clearly a next step could be to start some "orthogonal" validation, i.e. beyond yeast growth under methotrexate treatment.

      The reviewer makes a great point that we also discuss on page 9, line 33:

      While we used reconstruction of C-terminal-attached mDHFR fragments as a reporter for PPI abundance, similar massively parallel assays could be constructed with different PCA reporters or tagging configurations to validate our observations and overcome false negatives that are specific to our reporter. Indeed, the recent development of “swap tag” libraries, where new markers can be inserted C- or N-terminal to most genes (Weill et al., 2018; Yofe et al., 2016), in combination with our iSeq double barcoder collection (Liu et al., 2019), makes extension of our approach eminently feasible.

      Reviewer 3

      Reviewer #3 (Evidence, reproducibility and clarity (Required)):

      **Summary**

      The manuscript "A large accessory protein interactome is rewired across environments" by Liu et al. scales up a previously-described method (PPiSeq) to test a matrix of ~1.6 million protein pairs of direct protein-protein interactions in each of 9 different growth environments.

      While the study found a small fraction of immutable PPIs that are relatively stable across environments, the vast majority were 'mutable' across environments. Surprisingly, PPIs detected only in one environment made up more than 60% of the map. In addition to a false positive fraction that can yield apparently-mutable interactions, retest experiments demonstrate (not surprisingly) that environment-specificity can sometimes be attributed to false-negatives. The study authors predict that the whole subnetwork within the space tested will contain 11K true interactions.

      Much of environment-specific rewiring seemed to take place in an 'accessory module', which surrounds the core module made of mostly immutable PPIs. A number of interesting network clustering and functional enrichment analyses are performed to characterize the network overall and 'mutable' interactions in particular. The study report other global properties such as expression level, protein abundance and genetic interaction degree that differ between mutable and immutable PPIs. One of the interesting findings was evidence that many environmentally mutable PPI changes are regulated post-translationally. Finally, authors provide a case study about network rewiring related to glucose transport.

      **Major issues**

      -The results section should more prominently describe the dimensions of the matrix screen, both in terms of the set of protein pairs attempted and the set actually screened (I think this was 1741 x 1113 after filtering?). More importantly, the study should acknowledge in the introduction that this was NOT a random sample of protein pairs, but rather focused on pairs for which interaction had been previously observed in the baseline condition. This major bias has a potentially substantial impact on many of the downstream analyses. For example, any gene which was not expressed under the conditions of the original Tarrasov et al. study on which the screening space was based will not have been tested here. Thus, the study has systematically excluded interactions involving proteins with environment-dependent expression, except where they happened to be expressed in the single Tarrasov et al. environment. Heightened connectivity within the 'core module' may result from this bias, and if Tarrasov et al had screened in hydrogen peroxide (H2O2) instead of SD media, perhaps the network would have exhibited a code module in H2O2 decorated by less-densely connected accessory modules observed in other environments. The paper should clearly indicate which downstream analyses have special caveats in light of this design bias.

      We have now added text the matrix dimensions of our study on page 3, line 3:

      To generate a large PPiSeq library, all strains from the protein interactome (mDHFR-PCA) collection that were found to contain a protein likely to participate in at least one PPI (1742 X 1130 protein pairs), (Tarassov et al., 2008) were barcoded in duplicate using the double barcoder iSeq collection (Liu et al., 2019), and mated together in a single pool (Figure 1A). Double barcode sequencing revealed that the PPiSeq library contained 1.79 million protein pairs and 6.05 million double barcodes (92.3% and 78.1% of theoretical, respectively, 1741 X 1113 protein pairs), with each protein pair represented by an average of 3.4 unique double barcodes (Figure S1).

      We agree with the reviewer that our selection of proteins from a previously identified set can introduce bias in our conclusions. Our research question was focused on how PPIs change across environments, and thus we chose to maximize our power to detect PPI changes by selecting a set of protein pairs that are enriched for PPIs. We have now added a discussion of the potential caveats of this choice to the discussion on page 9, line 4:

      Results presented here and elsewhere (Huttlin et al., 2020) suggest that PPIs discovered under a single condition or cell type are a small subset of the full protein interactome emergent from a genome. We sampled nine diverse environments and found approximately 3-fold more interactions than in a single environment. However, the discovery of new PPIs began to saturate, indicating that most condition-specific PPIs can be captured in a limited number of conditions. Testing in many more conditions and with PPI assays orthogonal to PPiSeq will undoubtedly identify new PPIs, however a more important outcome could be the identification of coordinated network changes across conditions. Using a test set of ~1.6 million (of ~18 million) protein pairs across nine environments, we find that specific parts of the protein interactome are relatively stable (core modules) while others frequently change across environments (accessory modules). However, two important caveats of our study must be recognized before extrapolating these results to the entire protein interactome across all environment space. First, we tested for interactions between a biased set of proteins that have previously been found to participate in at least one PPI as measured by mDHFR-PCA under standard growth conditions (Tarassov et al., 2008). Thus, proteins that are not expressed under standard growth conditions are excluded from our study, as are PPIs that are not detectable by mDHFR-PCA or PPiSeq. It is possible that a comprehensive screen using multiple orthogonal PPI assays would alter our observations related to the relative dynamics of different regions of the protein interactome and the features of mutable and immutable PPIs. Second, we tested a limited number of environmental perturbations under similar growth conditions (batch liquid growth). It is possible that more extreme environmental shifts (e.g. growth as a colony, anaerobic growth, pseudohyphal growth) would introduce new accessory modules or alter the mutability of the PPIs we detect. Nevertheless, results presented here provide a new mechanistic view of how the cell changes in response to environmental challenges, building on the previous work that describes coordinated responses in the transcriptome (Brauer et al., 2007; Gasch et al., 2000) and proteome (Breker et al., 2013; Chong et al., 2015).

      -Related to the previous issue, a quick look at the proteins tested (if I understood them correctly) showed that they were enriched for genes encoding the elongator holoenzyme complex, DNA-directed RNA polymerase I complex, membrane docking and actin binding proteins, among other functional enrichments. Genes related to DNA damage (endonuclease activity and transposition), were depleted. It was unclear whether the functional enrichment analyses described in the paper reported enrichments relative to what would be expected given the bias inherent to the tested space?

      We did two functional enrichment analyses in this study: network density within Gene Ontology terms (related to Figure 2) and gene ontology enrichment of network communities (related to Figure 3). For both analyses, we performed comparisons to proteins included in PPiSeq library. This is described in the Supplementary Materials on page 63, line 35:

      To estimate GO term enrichment in our PPI network, we constructed 1000 random networks by replacing each bait or prey protein that was involved in a PPI with a randomly chosen protein from all proteins in our screen. This randomization preserves the degree distribution of the network.

      And on page 66, line 38:

      The set of proteins used for enrichment comparison are proteins that are involved in at least one PPI as determined by PPiSeq.

      -Re: data quality. To the study's great credit, they incorporated positive and random reference sets (PRS and RRS) into the screen. However, the results from this were concerning: Table SM6 shows that assay stringency was set such that between 1 and 3 out of 67 RRS pairs were detected. This specificity would be fine for an assay intended for retest or validate previous hits, where the prior probability of a true interaction is high, but in large-scale screening the prior probability of true interactions that are detectable by PCA is much lower, and a higher specificity is needed to avoid being overwhelmed by false positives. Consider this back of the envelope calculation: Let's say that the prior probability of true interaction is 1% as the authors' suggest (pg 49, section 6.5), and if PCA can optimistically detect 30% of these pairs, then the number of true interactions we might expect to see in an RRS of size 67 is 1% * 30% * 67 = 0.2 . This back of the envelope calculation suggests that a stringency allowing 1 hit in RRS will yield 80% [ (1 - 0.2) / 1 ] false positives, and a stringency allowing 3 hits in RRS will yield 93% [ (3 - 0.2) / 3] false positives. How do the authors reconcile these back of the envelope calculations from their PRS and RRS results with their estimates of precision?

      We thank the reviewer for bringing up with this issue. We included positive and random reference sets (PRS:70 protein pairs, RRS:67 protein pairs) to benchmark our PPI calling (Yu et al., 2008). The PRS reference lists PPIs that have been validated by multiple independent studies and is therefore likely to represent true PPIs that are present in some subset of the environments we tested. For the PRS set, we found a rate of detection that is comparable to other studies (PPiSeq in SD: 28%, Y2H and yellow fluorescent protein-PCA: ~20%) (Yu et al., 2008). The RRS reference, developed ten years ago, is randomly chosen protein pairs for which there was no evidence of a PPI in the literature at the time (mostly in standard growth conditions). Given the relatively high rate of false negatives in PPI assays, this set may in fact contain some true PPIs that have yet to be discovered. We could detect PPIs for four RRS protein pairs in our study, when looking across all 9 environments. Three of these (Grs1_Pet10, Rck2_Csh1, and YDR492W_Rpd3) could be detected in multiple environments (9, 7, and 3, respectively), suggesting that their detection was not a statistical or experimental artifact of our bar-seq assay (see table below derived from Table S4). The remaining PPI detected in the RRS, was only detected in SD (standard growth conditions) but with a relatively high fitness (0.35), again suggesting its detection was not a statistical or experimental artifact. While we do acknowledge it is possible that these are indeed false positives due to erroneous interactions of chimeric DHFR-tagged versions of these proteins, the small size of the RRS combined with the fact that some of the protein pairs could be true PPIs, did not give us confidence that this rate (4 of 70) is representative of our true false positive rate. To determine a false positive rate that is less subject to biases stemming from sampling of small numbers, we instead generated 50 new, larger random reference sets, by sampling for each set ~ 60,000 protein pairs without a reported PPI in BioGRID. Using these new reference sets, we found that the putative false positive rate of our assay is generally lower than 0.3% across conditions for each of the 50 reference sets. We therefore used this more statistically robust measure of the false positive rate to estimate positive predictive values (PPV = 62%, TPR = 41% in SD). We detail these statistical methods in Section 6 of the supplementary methods and report all statistical metrics in Table SM6.

      PPI

      Environment_number

      SD

      H2O2

      Hydroxyurea

      Doxorubicin

      Forskolin

      Raffinose

      NaCl

      16℃

      FK506

      Rck2_Csh1

      7

      0.35

      0.35

      0

      0.20

      0.54

      0.74

      0

      0.17

      0.59

      Grs1_Pet10

      9

      0.44

      0.39

      0.34

      0.25

      0.65

      1.19

      0.2

      0.16

      0.95

      YDR492W_Rpd3

      3

      0

      0.18

      0

      0

      0

      0

      0

      0.17

      0.61

      Mrps35_Bub3

      1

      0.35

      0

      0

      0

      0

      0

      0

      0

      0

      Positive_control

      9

      1

      0.8

      0.73

      0.62

      1.4

      2.44

      0.4

      0.28

      1.8

      Table. Mean fitness in each environment

      -Methods for estimating precision and recall were not sufficiently well described to assess. Precision vs recall plots would be helpful to better understand this tradeoff as score thresholds were evaluated.

      We describe in detail our approach to calling PPIs in section 6.6 of the supplementary methods, including Table SM6, and Figures SM3, SM4, SM6, and now Figure SM5. We identified positive PPIs using a dynamic threshold that considers the mean fitness and p-value in each environment. For each dynamic threshold, we estimated the precision and recall based on the reference sets (described supplementary methods in section 6.5). We then chose the threshold with the maximal Matthews correlation coefficient (MCC) to obtain the best balance between precision and recall. We have now added an additional plot (Figure SM5) that shows the precision and recall for the chosen dynamic threshold in each environment.

      -Within the tested space, the Tarassov et al map and the current map could each be compared against a common 'bronze standard' (e.g. literature curated interactions), at least for the SD map, to have an idea about how the quality of the current map compares to that of the previous PCA map. Each could also be compared with the most recent large-scale Y2H study (Yu et al).

      We thank the reviewer for this suggestion. We have now added a figure panel (Figure S4) that compares PPiSeq in SD (2 replicates) to mDHFR PCA (Tarassov et al., 2008), Y2H (Yu et al., 2008), and our newly constructed ‘bronze standard’ high-confidence positive reference set (PRS, supplementary method section 6.4).

      • Experimental validation of the network was done by conventional PCA. However, it should be noted that this is a form of technical replication of the DHFR-based PCA assay, and not a truly independent validation. Other large-scale yeast interaction studies (e.g., Yu et al, Science 2008) have assessed a random subset of observed PPIs using an orthogonal approach, calibrated using PRS and RRS sets examined via the same orthogonal method, from which overall performance of the dataset could be determined.

      We appreciate the reviewer’s perspective, since orthogonal validation experiments have been a critical tool to establish assay performance following early Y2H work. We know from careful work done previously that modern orthogonal assays have a low cross validation rate ((Yu et al., 2008) and that they tend to be enriched for PPIs in different cellular compartments (Jensen and Bork, 2008), indicating that high false negative rates are the likely explanation. High false negative rates have been confirmed here and elsewhere using positive reference sets (e.g. Y2H 80%, PCA 80%, PPiSeq 74% using the PRS in (Yu et al., 2008)). Therefore, the expectation is that PPiSeq, as with other assays, will have a low rate of validation using an orthogonal assay -- although we would not know if this rate is 10%, 30% or somewhere in between without performing the work. However, the exact number -- whether it be 10% or 30% -- has no practical impact on the main conclusions of this study (focused on network dynamics rather than network enumeration). Neither does that number speak to the confidence in our PPI calls, since a lower number may simply be due to less overlap in the sets of PPIs that are callable by PPiSeq and another assay. Our method uses bar-seq to extend an established mDHFR-PCA assay (Tarassov et al., 2008). The validations we performed were aimed at confirming that our sequencing, barcode counting, fitness estimation, and PPI calling protocols were not introducing excessive noise relative to mDHFR-PCA that resulted in a high number of PPI miscalls. Confirming this, we do indeed find a high rate of validation by lower throughput PCA (50-90%, Figure 3B). Finally, we do include independent tests of the quality of our data by comparing it to positive and random reference sets from literature curated data. We find that our assay performs extremely well (PPV > 61%, TPR > 41%) relative to other high-throughput assays.

      -The Venn diagram in Figure 1G was not very informative in terms of assessing the quality of data. It looks like there is a relatively little overlap between PPIs identified in standard conditions (SD media) in the current study and those of the previous study using a very similar method. Is there any way to know how much of this disagreement can be attributed to each screen being sub-saturation (e.g. by comparing replica screens) and what fraction to systematic assay or environment differences?

      We have now added a figure panel (Figure S4) that compares PPiSeq in SD (2 replicates) to mDHFR-PCA (Tarassov et al., 2008), Y2H (Yu et al., 2008), and our newly constructed ‘bronze standard’ high-confidence positive reference sets (PRS, supplementary methods section 6.4). We find that SD replicates have an overlap coefficient of 79% with each other, ~45% with mDHFR-PCA, ~45% the ‘bronze standard’ PRS, and ~13% with Y2H. Overlap coefficients between the SD replicates and mDHFR-PCA are much higher than those found between orthologous methods ((Yu et al., 2008), indicating that these two assays are identifying a similar set of PPIs. We do note that PPiSeq and mDHFR-PCA do screen for PPIs under different growth conditions (batch liquid growth vs. colonies on agar), so some fraction of the disagreement is due to environmental differences. PPIs that overlap between the two PPiSeq SD replicates are more likely to be found in mDHFR-PCA, PRS, and Y2H, indicating that PPIs identified in a single SD replicate are more likely to be false positives. However, we do find (a lower rate of) overlaps between PPIs identified in only one SD replicate and other methods, suggesting that a single PPiSeq replicate is not finding all discoverable PPIs.

      -In Figure S5C, the environment-specificity rate of PPIs might be inflated due to the fact that authors only test for the absence of SD hits in other conditions, and the SD condition is the only condition that has been sampled twice during the screening. What would be the environment-specific verification rate if sample hits from each environment were tested in all environments? This seems important, as robustly detecting environment-specific PPIs is one of the key points of the study.

      We use PPIs found in the SD environment to determine the environment-specificity because this provides the most conservative (highest) estimate of the number of PPIs found in other environments that were not detectable by our bar-seq assay. To identify PPIs in the SD environment, we pooled fitness estimates across the two replicates (~ 4 fitness estimates per replicate, ~ 8 total). The higher number of replicates results in a reduced rate of false positives (an erroneous fitness estimate has less impact on a PPI call), meaning that we are more confident that PPIs identified in SD are true positives. Because false positives in one environment (but not other environments) are likely to erroneously contribute to the environment-specificity rate, choosing the environment with the lowest rate of false positives (SD) should result in the lowest environment-specificity rate (highest estimate of PPIs found in other environments that were not detectable by our bar-seq assay).

      **Minor issues**

      -Re: "An interaction between the proteins reconstitutes mDHFR, providing resistance to the drug methotrexate and a growth advantage that is proportional to the PPI abundance" (pg 2). It may be more accurate to say "monotonically related" than "proportional" here. Fig 2 from the cited Freschi et al ref does suggests linearity with colony size over a wide range of inferred complex abundances, but non-linear at low complex abundance. Also note that Freschi measured colony area which is not linear with exponential growth rate nor with cell count.

      We agree with the reviewer and have changed “proportional” to “monotonically related” on page 2, line 41.

      -Re: "Using putatively positive and negative reference sets, we empirically determined a statistical threshold for each environment with the best balance of precision and recall (positive predictive value (PPV) > 61% in SD media, Methods, section 6)." (pg 3). Should state the recall at this PPV.

      We agree with the reviewer and have added the recall (41%) in the main text (line 26, page3).

      Using putatively positive and negative reference sets, we empirically determined a statistical threshold for each environment with the best balance of precision and recall (positive predictive value (PPV) > 61% and true positive rate > 41% in SD media, Methods, section 6).

      -Authors could discuss the extent to which related methods (e.g. PMID: 28650476, PMID: 27107012, PMID: 29165646, PMID: 30217970) would be potentially suitable for screening in different environments.

      We have now added a reference to a barcode-based Y2H study that examined interactions between yeast proteins to the introduction on page 2, line 2:

      Yet, little is known about how PPI networks reorganize on a global scale or what drives these changes. One challenge is that commonly-used high-throughput PPI screening technologies are geared toward PPI identification (Gavin et al., 2002; Ito et al., 2001; Tarassov et al., 2008; Uetz et al., 2000; Yu et al., 2008, Yachie et al., 2016), not a quantitative analysis of relative PPI abundance that is necessary to determine if changes in the PPI network are occurring. The murine dihydrofolate reductase (mDHFR)‐based protein-fragment complementation assay (PCA) provides a viable path to characterize PPI abundance changes because it is a sensitive test for PPIs in the native cellular context and at native protein expression levels (Freschi et al., 2013; Remy and Michnick, 1999; Tarassov et al., 2008).

      We have excluded the references to other barcode-based Y2H studies that reviewer mentions because they test heterologous proteins within yeast, and the effect of perturbations to yeast on these proteins would be difficult to interpret in the context of our questions. The yeast protein Y2H study, although a wonderful approach and paper, would also not be an appropriate method to examine how PPI networks change across environments because protein fusions are not expressed under their endogenous promoters and must be transported to, in many cases, a non-native compartment (cell nucleus) to be detected. Rather than explicitly discuss the caveats of this particular approach, we have instead chosen to discuss why we use PCA.

      • the term "mutable" is certainly appropriate according to the dictionary definition of changeable. The authors may wish to consider though, that in a molecular biology context the term evokes changeability by mutation (a very interesting but distinct topic). Maybe another term (environment-dependent interactions or ePPIs?) would be clearer. Of course this is the authors' call.

      We thank the reviewer for this suggestion, and have admittedly struggled with the terminology. For clarity of presentation, we strived to have a single word that describes the property of a PPI that is at the core of this manuscript -- how frequently a PPI is found across environments. However, the most descriptive words come with preloaded meanings in PPI research (e.g. transient, stable, dynamic), as does “mutable” with another research field. We are, quite frankly, open to suggestions from the reviewers or editors for a more appropriate word that does not raise similar objections.

      -Some discussion is warranted about the phenomenon that a PPI that is unchanged in abundance could appear to change because of statistical significance thresholds that differ between screens. This would be a difficult question for any such study, and I don't think the authors need to solve it, but just to discuss.

      We agree with the reviewer that significance thresholds could be impacting our interpretations and discuss this idea at length on page 4, line 23 of the Results. This section has been modified to include an additional analysis (excluding 16 ℃ data) in response to another reviewer’s comment:

      Immutable PPIs were likely to have been previously reported by colony-based mDHFR-PCA or other methods, while the PPIs found in the fewest environments were not. One possible explanation for this observation is that previous PPI assays, which largely tested in standard laboratory growth conditions, and variations thereof, are biased toward identification of the least mutable PPIs. That is, since immutable PPIs are found in nearly all environments, they are more readily observed in just one. However, another possible explanation is that, in our assay, mutable PPIs are more likely to be false positives in environment(s) in which they are identified or false negatives in environments in which they are not identified. To investigate this second possibility, we first asked whether PPIs present in very few environments have lower fitnesses, as this might indicate that they are closer to our limit of detection. We found no such pattern: mean fitnesses were roughly consistent across PPIs found in 1 to 6 conditions, although they were elevated in PPIs found in 7-9 conditions (Figure S6A). To directly test the false-positive rate stemming from pooled growth and barcode sequencing, we validated randomly selected PPIs within each mutability bin by comparing their optical density growth trajectories against controls (Figures 3B). We found that mutable PPIs did indeed have lower validation rates in the environment in which they were identified, yet putative false positives were limited to ~50%, and, within a bin, do not differ between PPIs that have been previously identified and those that have been newly discovered by our assay (Figure S65B). We also note mutable PPIs might be more sensitive to environmental differences between our large pooled PPiSeq assays and clonal 96-well validation assays, indicating that differences in validation rates might be overstated. To test the false-negative rate, we assayed PPIs identified in only SD by PPiSeq across all other environments by optical density growth and found that PPIs can be assigned to additional environments (Figure S6C). However, the number of additional environments in which a PPI was detected was generally low (2.5 on average), and the interaction signal in other environments was generally weaker than in SD (Figure S6D). To better estimate how the number of PPIs changes with PPI mutability, we used these optical density assays to model the validation rate as a function of the mean PPiSeq fitness and the number of environments in which a PPI is detected. This accurate model (Spearman's r =0.98 between predicted and observed, see Methods) provided confidence scores (predicted validation rates) for each PPI (Table S5) and allowed us to adjust the true positive PPI estimate in each mutability bin. Using this more conservative estimate, we still found a preponderance of mutable PPIs (Figure S6E). Finally, we used a pair of more conservative PPI calling procedures that either identified PPIs with a low rate of false positives across all environments (FPR

      We later examine major conclusions of our study using more conservative calling procedures, and find that they are consistent. On page 6, line 14:

      Both the co-expression and co-localization patterns were also apparent in our higher confidence PPI sets (Figures S7B, and S7C, S8B, S8C ), indicating that they are not caused by different false positive rates between the mutability bins.

      And on page 6, line 19:

      We binned proteins by their PPI degree, and, within each bin, determined the correlation between the mutability score and another gene feature (Figure 4C and S12A, Table S8) (Costanzo et al., 2016; Finn et al., 2014; Gavin et al., 2006; Holstege et al., 1998; Krogan et al., 2006; Levy and Siegal, 2008; Myers et al., 2006; Newman et al., 2006; Östlund et al., 2010; Rice et al., 2000; Stark et al., 2011; Wapinski et al., 2007; Ward et al., 2004; Yang, 2007; Yu et al., 2008). These correlations were also calculated using our higher confidence PPI sets, confirming results from the full data set (Figures S7D and, S7E, S8D, S8E). We found that mutable hubs (> 15 PPIs) have more genetic interactions, in agreement with predictions from co-expression data (Bertin et al., 2007; Han et al., 2004), and that their deletion tends to cause larger fitness defects.

      -More discussion would be helpful about the idea that immutability may to some extent favor interactions that PCA is better able to detect (possibly including membrane proteins?)

      We agree with the reviewer and now added a discussion of this potential caveats to the discussion on page 9, line 4:

      Results presented here and elsewhere (Huttlin et al., 2020) suggest that PPIs discovered under a single condition or cell type are a small subset of the full protein interactome emergent from a genome. We sampled nine diverse environments and found approximately 3-fold more interactions than in a single environment. However, the discovery of new PPIs began to saturate, indicating that most condition-specific PPIs can be captured in a limited number of conditions. Testing in many more conditions and with PPI assays orthogonal to PPiSeq will undoubtedly identify new PPIs, however a more important outcome could be the identification of coordinated network changes across conditions. Using a test set of ~1.6 million (of ~18 million) protein pairs across nine environments, we find that specific parts of the protein interactome are relatively stable (core modules) while others frequently change across environments (accessory modules). However, two important caveats of our study must be recognized before extrapolating these results to the entire protein interactome across all environment space. First, we tested for interactions between a biased set of proteins that have previously been found to participate in at least one PPI as measured by mDHFR-PCA under standard growth conditions (Tarassov et al., 2008). Thus, proteins that are not expressed under standard growth conditions are excluded from our study, as are PPIs that are not detectable by mDHFR-PCA or PPiSeq. It is possible that a comprehensive screen using multiple orthogonal PPI assays would alter our observations related to the relative dynamics of different regions of the protein interactome and the features of mutable and immutable PPIs. Second, we tested a limited number of environmental perturbations under similar growth conditions (batch liquid growth). It is possible that more extreme environmental shifts (e.g. growth as a colony, anaerobic growth, pseudohyphal growth) would introduce new accessory modules or alter the mutability of the PPIs we detect. Nevertheless, results presented here provide a new mechanistic view of how the cell changes in response to environmental challenges, building on the previous work that describes coordinated responses in the transcriptome (Brauer et al., 2007; Gasch et al., 2000) and proteome (Breker et al., 2013; Chong et al., 2015).

      -Re: "As might be expected, we also found that mutable hubs, but not non-hubs, are more likely to participate in multiple protein complexes than less mutable proteins." (pg 6) This is a cool result. To what extent was this result driven by members of one or two complexes? If so, it would worth noting them.

      We thank the reviewer for this question. We have now included Figue S13, which shows the number and size of protein complexes that underlie the finding that mutable hubs are more likely to participate in multiple protein complexes. We find that proteins in our screen that participate in multiple complexes are distributed over a wide range of complexes, indicating that this observation is not driven by one or two complexes. On page 6, line 34:

      As might be expected, we also found that mutable hubs, but not non-hubs, are more likely to participate in multiple protein complexes than less mutable proteins (Figures S13A-C) (Costanzo et al., 2016).

      -Re: "Borrowing a species richness estimator from ecology (Jari Oksanen et al., 2019), we estimate that there are ~10,840 true interactions within our search space across all environments, ~3-fold more than are detected in SD (note difference to Figure 3, which counts observed PPIs)." (pg 8) Should note that this only allows estimation of the number of interactions that are detectable by PCA methods. Previous work (Braun et al, 2019) showed that every known protein interaction assay (including PCA approaches) can only detect a fraction of bona fide interactions.

      We agree with the reviewer and have modified the discussion to make this point explicit on page 9, line 4:

      Results presented here and elsewhere (Huttlin et al., 2020) suggest that PPIs discovered under a single condition or cell type are a small subset of the full protein interactome emergent from a genome. We sampled nine diverse environments and found approximately 3-fold more interactions than in a single environment. However, the discovery of new PPIs began to saturate, indicating that most condition-specific PPIs can be captured in a limited number of conditions. Testing in many more conditions and with PPI assays orthogonal to PPiSeq will undoubtedly identify new PPIs, however a more important outcome could be the identification of coordinated network changes across conditions.

      We continue in this paragraph to discuss the implications:

      Using a test set of ~1.6 million (of ~18 million) protein pairs across nine environments, we find that specific parts of the protein interactome are relatively stable (core modules) while others frequently change across environments (accessory modules). However, two important caveats of our study must be recognized before extrapolating these results to the entire protein interactome across all environment space. First, we tested for interactions between a biased set of proteins that have previously been found to participate in at least one PPI as measured by mDHFR-PCA under standard growth conditions (Tarassov et al., 2008). Thus, proteins that are not expressed under standard growth conditions are excluded from our study, as are PPIs that are not detectable by mDHFR-PCA or PPiSeq. It is possible that a comprehensive screen using multiple orthogonal PPI assays would alter our observations related to the relative dynamics of different regions of the protein interactome and the features of mutable and immutable PPIs.

      -Re: "This analysis shows that the number of PPIs present across all environments is much larger than the number observed in a single condition, but that it is feasible to discover most of these new PPIs by sampling a limited number of conditions." (pg 8). The main point is surely correct, but it is worth noting that extrapolation to the number of true interactions depends on the nine chosen environments being representative of all environments. The situation could change under more extreme, e.g., anaerobic, conditions.

      We agree with the reviewer and make this point explicit, continuing from the paragraph quoted above on page 9, line 22:

      Second, we tested a limited number of environmental perturbations under similar growth conditions (batch liquid growth). It is possible that more extreme environmental shifts (e.g. growth as a colony, anaerobic growth, pseudohyphal growth) would introduce new accessory modules or alter the mutability of the PPIs we detect. Nevertheless, results presented here provide a new mechanistic view of how the cell changes in response to environmental challenges, building on the previous work that describes coordinated responses in the transcriptome (Brauer et al., 2007; Gasch et al., 2000) and proteome (Breker et al., 2013; Chong et al., 2015).

      -It stands to reason that proteins expressed in all conditions will yield less mutable interactions, if 'mutability' is primarily due to expression change at the transcriptional level. They should at least discuss that measuring mRNA levels could resolve questions about this. Could use Waern et al G3 2013 data (H202, SD, HU, NaCl) to predict the dynamic interactome purely by node removal, and see how conclusions would change

      We agree with the reviewer that mRNA abundance could potentially be used as a proxy for protein abundance and have added this point on page 10, line 28:

      Here we use homodimer abundance as a proxy for protein abundance. However, genome-wide mRNA abundance measures could be used as a proxy for protein abundance or protein abundance could be measured directly in the same pool (Levy et al., 2014) by, for example, attaching a full length mDHFR to each gene using “swap tag” libraries mentioned above (Weill et al., 2018; Yofe et al., 2016).

      However, using mRNA abundance as a proxy for protein abundance in this study has several important caveats that would make interpretation difficult. First, mRNA and protein abundance correlate, but not perfectly (R2 = 0.45) (Lahtvee et al., 2017), and our findings suggest that post-translational regulation may be important to driving PPI changes. Second, mRNA abundance measures are for a single time point, while our PPI measures coarse grain over a growth cycle (lag, exponential growth, diauxic shift, saturation). Although we may be able to take multiple mRNA measures across the cycle, time delays between changes in mRNA and protein levels, combined with the fact that we do not know when a PPI is occurring or most prominent over the cycle, would pose a significant challenge to making any claims that PPI changes are driven by changes in protein abundance. We instead chose to focus on a subset of proteins (homodimers) where abundance measures can be coarse grained in the same way as PPI measures. In the above quote, we point to a potential method by which this can be done for all proteins. We also point to how a continuous culturing design could be used to better determine how protein (or mRNA proxy) abundance impacts PPI abundance on page 10, line 6:

      Finally, our assays were performed across cycles of batch growth meaning that changes in PPI abundance across a growth cycle (e.g. lag, exponential growth, saturation) are coarse grained into one measurement. While this method potentially increases our chance of discovering a diverse set of PPIs, it might have an unpredictable impact on the relationship between fitness and PPI abundance (Li et al., 2018). To overcome these issues, strains containing natural or synthetic PPIs with known abundances and intracellular localizations could be spiked into cell pools to calibrate the relationship between fitness and PPI abundance in each environment. In addition, continuous culturing systems may be useful for refining precision of growth-based assays such as ours.

      -The analysis showing that many interactions are likely due to post-translational modifications is very interesting, but caveats should be discussed. Where heterodimers do not fit the expression-level dependence model, some cases of non-fitting may simply be due to measurement error or non-linearity in the relationship between abundance and fitness.

      We show the measurement error in Figures 1, S2, S3. While we agree with the reviewer that measurement error is a general caveat for all results reported, we do not feel that it is necessary to point to that fact in this particular case, which uses a logistic regression to report that PPI mutability was the best predictor of fit to the expression-level dependence model. We discuss the non-linearity caveat on page 9, line 41:

      Our assay detected subtle fitness differences across environments (Fig S5B and S5C), which we used as a rough estimate for changes in relative PPI abundance. While it would be tempting to use fitness as a direct readout of absolute PPI abundance within a cell, non-linearities between fitness and PPI abundance may be common and PPI dependent. For example, the relative contribution of a reconstructed mDHFR molecule to fitness might diminish at high PPI abundances (saturation effects) and fitness differences between PPIs may be caused, in part, by differences in how accessible a reconstructed mDHFR molecule is to substrate. In addition, environmental shifts might impact cell growth rate, initiate a stress response, or result in other unpredictable cell effects that impact the selective pressure of methotrexate and thereby fitness (Figure S2 and S3).

      -Line numbers would have been helpful to note more specific minor comments

      We are sorry for this inconvenience. We have added line numbers in our revised manuscript.

      -Sequence data should be shared via the Short-Read Archive.

      The raw sequencing data have been uploaded to the Short-Read Archive. We mentioned it in the Data and Software Availability section on page 68, line 41.

      Raw barcode sequencing data are available from the NIH Sequence Read Archive as accession PRJNA630095 (https://trace.ncbi.nlm.nih.gov/Traces/study/?acc=SRP259652).

      Reviewer #3 (Significance (Required)):

      Knowledge of protein-protein interactions (PPIs) provides a key window on biological mechanism, and unbiased screens have informed global principles underlying cellular organization. Several genome-scale screens for direct (binary) interactions between yeast proteins have been carried out, and while each has provided a wealth of new hypotheses, each has been sub-saturation. Therefore, even given multiple genome-scale screens our knowledge of yeast interactions remains incomplete. Different assays are better suited to find different interactions, and it is now clear that every assay evaluated thus far is only capable (even in a saturated screen) of detecting a minority of true interactions. More relevant to the current study, no binary interaction screen has been carried out at the scale of millions of protein pairs outside of a single 'baseline' condition.

      The study by Liu et al is notable from a technology perspective in that it is one of several recombinant-barcode approaches have been developed to multiplex pairwise combinations of two barcoded libraries. Although other methods have been demonstrated at the scale of 1M protein pairs, this is the first study using such a technology at the scale of >1M pairs across multiple environments.

      A limitation is that this study is not genome-scale, and the search space is biased towards proteins for which interactions were previously observed in a particular environment. This is perhaps understandable, as it made the study more tractable, but this does add caveats to many of the conclusions drawn. These would be acceptable if clearly described and discussed. There were also questions about data quality and assessment that would need to be addressed.

      Assuming issues can be addressed, this is a timely study on an important topic, and will be of broad interest given the importance of protein interactions and the status of S. cerevisiae as a key testbed for systems biology.

      *Reviewers' expertise:* Interaction assays, next-generation sequencing, computational genomics. Less able to assess evolutionary biology aspects.

      References

      Brauer, M.J., Huttenhower, C., Airoldi, E.M., Rosenstein, R., Matese, J.C., Gresham, D., Boer, V.M., Troyanskaya, O.G., and Botstein, D. (2007). Coordination of Growth Rate, Cell Cycle, Stress Response, and Metabolic Activity in Yeast. Mol. Biol. Cell 19, 352–367.

      Breker, M., Gymrek, M., and Schuldiner, M. (2013). A novel single-cell screening platform reveals proteome plasticity during yeast stress responses. J. Cell Biol. 200, 839–850.

      Chong, Y.T., Koh, J.L.Y., Friesen, H., Kaluarachchi Duffy, S., Cox, M.J., Moses, A., Moffat, J., Boone, C., and Andrews, B.J. (2015). Yeast Proteome Dynamics from Single Cell Imaging and Automated Analysis. Cell 161, 1413–1424.

      Gasch, A.P., Spellman, P.T., Kao, C.M., Carmel-Harel, O., Eisen, M.B., Storz, G., Botstein, D., and Brown, P.O. (2000). Genomic Expression Programs in the Response of Yeast Cells to Environmental Changes. Mol. Biol. Cell 11, 4241–4257.

      Hart, G.T., Ramani, A.K., and Marcotte, E.M. (2006). How complete are current yeast and human protein-interaction networks? Genome Biol. 7, 120.

      Hilliker, A., Gao, Z., Jankowsky, E., and Parker, R. (2011). The DEAD-box protein Ded1 modulates translation by the formation and resolution of an eIF4F-mRNA complex. Mol. Cell 43, 962–972.

      Isasa, M., Suñer, C., Díaz, M., Puig-Sàrries, P., Zuin, A., Bichmann, A., Gygi, S.P., Rebollo, E., and Crosas, B. (2015). Cold Temperature Induces the Reprogramming of Proteolytic Pathways in Yeast. J. Biol. Chem. jbc.M115.698662.

      Jensen, L.J., and Bork, P. (2008). Not Comparable, But Complementary. Science 322, 56–57.

      Lahtvee, P.-J., Sánchez, B.J., Smialowska, A., Kasvandik, S., Elsemman, I.E., Gatto, F., and Nielsen, J. (2017). Absolute Quantification of Protein and mRNA Abundances Demonstrate Variability in Gene-Specific Translation Efficiency in Yeast. Cell Syst. 4, 495-504.e5.

      Obayashi, T., Kagaya, Y., Aoki, Y., Tadaka, S., and Kinoshita, K. (2019). COXPRESdb v7: a gene coexpression database for 11 animal species supported by 23 coexpression platforms for technical evaluation and evolutionary inference. Nucleic Acids Res. 47, D55–D62.

      Sambourg, L., and Thierry-Mieg, N. (2010). New insights into protein-protein interaction data lead to increased estimates of the S. cerevisiae interactome size. BMC Bioinformatics 11, 605.

      Tarassov, K., Messier, V., Landry, C.R., Radinovic, S., Molina, M.M.S., Shames, I., Malitskaya, Y., Vogel, J., Bussey, H., and Michnick, S.W. (2008). An in Vivo Map of the Yeast Protein Interactome. Science 320, 1465–1470.

      Yu, H., Braun, P., Yıldırım, M.A., Lemmens, I., Venkatesan, K., Sahalie, J., Hirozane-Kishikawa, T., Gebreab, F., Li, N., Simonis, N., et al. (2008). High-Quality Binary Protein Interaction Map of the Yeast Interactome Network. Science 322, 104–110.

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      Referee #3

      Evidence, reproducibility and clarity

      Summary

      The manuscript "A large accessory protein interactome is rewired across environments" by Liu et al. scales up a previously-described method (PPiSeq) to test a matrix of ~1.6 million protein pairs of direct protein-protein interactions in each of 9 different growth environments.

      While the study found a small fraction of immutable PPIs that are relatively stable across environments, the vast majority were 'mutable' across environments. Surprisingly, PPIs detected only in one environment made up more than 60% of the map. In addition to a false positive fraction that can yield apparently-mutable interactions, retest experiments demonstrate (not surprisingly) that environment-specificity can sometimes be attributed to false-negatives. The study authors predict that the whole subnetwork within the space tested will contain 11K true interactions.

      Much of environment-specific rewiring seemed to take place in an 'accessory module', which surrounds the core module made of mostly immutable PPIs. A number of interesting network clustering and functional enrichment analyses are performed to characterize the network overall and 'mutable' interactions in particular. The study report other global properties such as expression level, protein abundance and genetic interaction degree that differ between mutable and immutable PPIs. One of the interesting findings was evidence that many environmentally mutable PPI changes are regulated post-translationally. Finally, authors provide a case study about network rewiring related to glucose transport.

      Major issues

      -The results section should more prominently describe the dimensions of the matrix screen, both in terms of the set of protein pairs attempted and the set actually screened (I think this was 1741 x 1113 after filtering?). More importantly, the study should acknowledge in the introduction that this was NOT a random sample of protein pairs, but rather focused on pairs for which interaction had been previously observed in the baseline condition. This major bias has a potentially substantial impact on many of the downstream analyses. For example, any gene which was not expressed under the conditions of the original Tarrasov et al. study on which the screening space was based will not have been tested here. Thus, the study has systematically excluded interactions involving proteins with environment-dependent expression, except where they happened to be expressed in the single Tarrasov et al. environment. Heightened connectivity within the 'core module' may result from this bias, and if Tarrasov et al had screened in hydrogen peroxide (H2O2) instead of SD media, perhaps the network would have exhibited a code module in H2O2 decorated by less-densely connected accessory modules observed in other environments. The paper should clearly indicate which downstream analyses have special caveats in light of this design bias.

      -Related to the previous issue, a quick look at the proteins tested (if I understood them correctly) showed that they were enriched for genes encoding the elongator holoenzyme complex, DNA-directed RNA polymerase I complex, membrane docking and actin binding proteins, among other functional enrichments. Genes related to DNA damage (endonuclease activity and transposition), were depleted. It was unclear whether the functional enrichment analyses described in the paper reported enrichments relative to what would be expected given the bias inherent to the tested space?

      -Re: data quality. To the study's great credit, they incorporated positive and random reference sets (PRS and RRS) into the screen. However, the results from this were concerning: Table SM6 shows that assay stringency was set such that between 1 and 3 out of 67 RRS pairs were detected. This specificity would be fine for an assay intended for retest or validate previous hits, where the prior probability of a true interaction is high, but in large-scale screening the prior probability of true interactions that are detectable by PCA is much lower, and a higher specificity is needed to avoid being overwhelmed by false positives. Consider this back of the envelope calculation: Let's say that the prior probability of true interaction is 1% as the authors' suggest (pg 49, section 6.5), and if PCA can optimistically detect 30% of these pairs, then the number of true interactions we might expect to see in an RRS of size 67 is 1% 30% 67 = 0.2 . This back of the envelope calculation suggests that a stringency allowing 1 hit in RRS will yield 80% [ (1 - 0.2) / 1 ] false positives, and a stringency allowing 3 hits in RRS will yield 93% [ (3 - 0.2) / 3] false positives. How do the authors reconcile these back of the envelope calculations from their PRS and RRS results with their estimates of precision?

      -Methods for estimating precision and recall were not sufficiently well described to assess. Precision vs recall plots would be helpful to better understand this tradeoff as score thresholds were evaluated.

      -Within the tested space, the Tarassov et al map and the current map could each be compared against a common 'bronze standard' (e.g. literature curated interactions), at least for the SD map, to have an idea about how the quality of the current map compares to that of the previous PCA map. Each could also be compared with the most recent large-scale Y2H study (Yu et al).

      • Experimental validation of the network was done by conventional PCA. However, it should be noted that this is a form of technical replication of the DHFR-based PCA assay, and not a truly independent validation. Other large-scale yeast interaction studies (e.g., Yu et al, Science 2008) have assessed a random subset of observed PPIs using an orthogonal approach, calibrated using PRS and RRS sets examined via the same orthogonal method, from which overall performance of the dataset could be determined.

      -The Venn diagram in Figure 1G was not very informative in terms of assessing the quality of data. It looks like there is a relatively little overlap between PPIs identified in standard conditions (SD media) in the current study and those of the previous study using a very similar method. Is there any way to know how much of this disagreement can be attributed to each screen being sub-saturation (e.g. by comparing replica screens) and what fraction to systematic assay or environment differences?

      -In Figure S5C, the environment-specificity rate of PPIs might be inflated due to the fact that authors only test for the absence of SD hits in other conditions, and the SD condition is the only condition that has been sampled twice during the screening. What would be the environment-specific verification rate if sample hits from each environment were tested in all environments? This seems important, as robustly detecting environment-specific PPIs is one of the key points of the study.

      Minor issues

      -Re: "An interaction between the proteins reconstitutes mDHFR, providing resistance to the drug methotrexate and a growth advantage that is proportional to the PPI abundance" (pg 2). It may be more accurate to say "monotonically related" than "proportional" here. Fig 2 from the cited Freschi et al ref does suggests linearity with colony size over a wide range of inferred complex abundances, but non-linear at low complex abundance. Also note that Freschi measured colony area which is not linear with exponential growth rate nor with cell count. -Re: "Using putatively positive and negative reference sets, we empirically determined astatistical threshold for each environment with the best balance of precision and recall (positive predictive value (PPV) > 61% in SD media, Methods, section 6)." (pg 3). Should state the recall at this PPV.

      -Authors could discuss the extent to which related methods (e.g. PMID: 28650476, PMID: 27107012, PMID: 29165646, PMID: 30217970) would be potentially suitable for screening in different environments.

      • the term "mutable" is certainly appropriate according to the dictionary definition of changeable. The authors may wish to consider though, that in a molecular biology context the term evokes changeability by mutation (a very interesting but distinct topic). Maybe another term (environment-dependent interactions or ePPIs?) would be clearer. Of course this is the authors' call.

      -Some discussion is warranted about the phenomenon that a PPI that is unchanged in abundance could appear to change because of statistical significance thresholds that differ between screens. This would be a difficult question for any such study, and I don't think the authors need to solve it, but just to discuss.

      -More discussion would be helpful about the idea that immutability may to some extent favor interactions that PCA is better able to detect (possibly including membrane proteins?)

      -Re: "As might be expected, we also found that mutable hubs, but not non-hubs, are more likely to participate in multiple protein complexes than less mutable proteins." (pg 6) This is a cool result. To what extent was this result driven by members of one or two complexes? If so, it would worth noting them.

      -Re: "Borrowing a species richness estimator from ecology (Jari Oksanen et al., 2019), we estimate that there are ~10,840 true interactions within our search space across all environments, ~3-fold more than are detected in SD (note difference to Figure 3, which counts observed PPIs)." (pg 8) Should note that this only allows estimation of the number of interactions that are detectable by PCA methods. Previous work (Braun et al, 2019) showed that every known protein interaction assay (including PCA approaches) can only detect a fraction of bona fide interactions.

      -Re: "This analysis shows that the number of PPIs present across all environments is much larger than the number observed in a single condition, but that it is feasible to discover most of these new PPIs by sampling a limited number of conditions." (pg 8). The main point is surely correct, but it is worth noting that extrapolation to the number of true interactions depends on the nine chosen environments being representative of all environments. The situation could change under more extreme, e.g., anaerobic, conditions.

      -It stands to reason that proteins expressed in all conditions will yield less mutable interactions, if 'mutability' is primarily due to expression change at the transcriptional level. They should at least discuss that measuring mRNA levels could resolve questions about this. Could use Waern et al G3 2013 data (H202, SD, HU, NaCl) to predict the dynamic interactome purely by node removal, and see how conclusions would change

      -The analysis showing that many interactions are likely due to post-translational modifications is very interesting, but caveats should be discussed. Where heterodimers do not fit the expression-level dependence model, some cases of non-fitting may simply be due to measurement error or non-linearity in the relationship between abundance and fitness.

      -Line numbers would have been helpful to note more specific minor comments

      -Sequence data should be shared via the Short-Read Archive.

      Significance

      Knowledge of protein-protein interactions (PPIs) provides a key window on biological mechanism, and unbiased screens have informed global principles underlying cellular organization. Several genome-scale screens for direct (binary) interactions between yeast proteins have been carried out, and while each has provided a wealth of new hypotheses, each has been sub-saturation. Therefore, even given multiple genome-scale screens our knowledge of yeast interactions remains incomplete. Different assays are better suited to find different interactions, and it is now clear that every assay evaluated thus far is only capable (even in a saturated screen) of detecting a minority of true interactions. More relevant to the current study, no binary interaction screen has been carried out at the scale of millions of protein pairs outside of a single 'baseline' condition.

      The study by Liu et al is notable from a technology perspective in that it is one of several recombinant-barcode approaches have been developed to multiplex pairwise combinations of two barcoded libraries. Although other methods have been demonstrated at the scale of 1M protein pairs, this is the first study using such a technology at the scale of >1M pairs across multiple environments.

      A limitation is that this study is not genome-scale, and the search space is biased towards proteins for which interactions were previously observed in a particular environment. This is perhaps understandable, as it made the study more tractable, but this does add caveats to many of the conclusions drawn. These would be acceptable if clearly described and discussed. There were also questions about data quality and assessment that would need to be addressed.

      Assuming issues can be addressed, this is a timely study on an important topic, and will be of broad interest given the importance of protein interactions and the status of S. cerevisiae as a key testbed for systems biology.

      Reviewers' expertise: Interaction assays, next-generation sequencing, computational genomics. Less able to assess evolutionary biology aspects.

    1. Figure 4.

      Posted on behalf of Xiaofan Wei and Hongquan Zhang by JCB:

      Thanks for the comments. We were also confused about the conflicting results which may be caused by the different tagged constructs or different concentration of plasmids used for the transfection. However, we cannot repeat the observation that Smurf1 has obvious effects on Talin-head degradation even though we performed the experiments at many different conditions and using several cell lines. Although Dr. Huang et al. found that Smurf1 induced slightly reduced levels of Talin-H as shown in Nat Cell Biol. 2009 May;11(5):624-30, Fig. S6a, we tend to think that Talin-head is not a direct target of Smurf1. If Talin-head is a genuine target of Smurf1, it should be degraded to the same extent as Kindlin-2 is degraded by Smurf1. Thus, it needs further investigation to elucidate the mechanism of Talin-head degradation.

    1. But they’re also a call to action — a reminder that we should all put our ideas out into the world, no matter how zany they may be.

      This can be very much a wake up call for creators, like a lot of us plan to be. If you think your idea is too weird to work, just look as this guy. He might give you a new sense of motivation in pursuing and embracing the weird

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      Reply to the reviewers

      We thank the reviewers for their close reading and constructive comments on our manuscript. We believe that their insight has substantially strengthened our manuscript. Please find our response/revision plan for each comment below (in blue). Note, because of the substantial changes to the figures and the additional experiments that are we are undertaking, we have not initially revised the text. The proposed textual revisions will be included in the full revision.

      Reviewer #1 (Evidence, reproducibility and clarity (Required)):

      The Katz lab has contributed greatly to the field of epigenetic reprogramming over the years, and this is

      another excellent paper on the subject. I enjoyed reviewing this manuscript and don't have any major

      comments/suggestions for improving it. The findings presented are novel and important, the results are clear

      cut, and the writing is clear.

      It's important to stress the novelty of the findings, which build upon previous studies from the same lab (upon

      a shallow look one might think that some of the conclusions were described before, but this is not the case).

      Despite the fact that this system has been studied in depth before, it remained unclear why and how

      germline genes are bookmarked by H3K36 in the embryo, and it wasn't known why germline genes are not

      expressed in the soma.

      To study these questions Carpenter et al. examine multiple phenotypes (developmental aberrations,

      sterility), that they combine with analysis of multiple genetic backgrounds, RNA-seq, CHIP-seq, single

      molecule FISH, and fluorescent transgenes.

      Previous observations from the Katz lab suggested that progeny derived from spr-5;met-2 double mutants

      can develop abnormally. They show here that the progeny of these double mutants (unlike spr-5 and met-2

      single mutants) develop severe and highly penetrate developmental delays, a Pvl phenotype, and sterility.

      They show also that spr-5; met-2 maternal reprogramming prevents developmental delay by restricting

      ectopic MES-4 bookmarking, and that developmental delay of spr-5;met-2 progeny is the result of ectopic

      expression of MES-4 germline genes. The bottom line is that they shed light on how SPR-5, MET-2 and

      MES-4 balance inter-generational inheritance of H3K4, H3K9, and H3K36 methylation, to allow correct

      specification of germline and somatic cells. This is all very important and relevant also to other organisms.

      **(very) Minor comments:**

      -Since the word "heritable" is used in different contexts, it could be helpful to elaborate, perhaps in the

      introduction, on the distinction between cellular memory and transgenerational inheritance.

      We are happy to elaborate on this in the revised manuscript.

      -It might be interesting in the Discussion to expand further about the links between heritable chromatin

      marks and heritable small RNAs. The do hint that the result regarding the silencing of the somatic transgene

      are especially intriguing.

      We are happy to expand this in the revised manuscript.

      Reviewer #1 (Significance (Required)):

      This is an exciting paper which build upon years of important work in the Katz lab. The novelty of the paper

      is in pinpointing the mechanisms that bookmark germline genes by H3K36 in the embryo, and explaining

      why and how germline genes are prevented from being expressed in the soma.

      Reviewer #2 (Evidence, reproducibility and clarity (Required)):

      Katz and colleagues examine the interaction between the methyltransferase MES-4 and spr-5; met-2 double

      mutants. Their prior analysis (PNAS, 2014) showed the dramatic enhancement in sterility and development

      for spr-5; met-2; this paper extends that finding by showing these effects depend on MES-4. The results are

      interesting and the genetic interactions dramatic. The examination by RNAseq and ChIP helps move the

      phenotypes into a more molecular analysis. The authors hypothesize that SPR-5 and MET-2 modify

      chromatin of germline genes (MES-4 targets) in somatic cells, and this is required to silence germline genes

      in the soma. A few issues need to be resolved to test these ideas and rule out others.

      **Main comments:**

      The authors' hypothesis is that SPR-5 and MET-2 act directly, to modify chromatin of germline genes (MES-

      4 targets), but alternate hypothesis is that the key regulated genes are i) MES-4 itself and/or ii) known

      regulators of germline gene expression e.g. the piwi pathway. Mis regulation of these factors in the soma

      could be responsible for the phenotypes. Therefore, the authors should analyze expression (smFISH and

      where possible protein stains) for MES-4 and PIWI components in the embryo and larvae of wildtype, double

      and triple mutant strains. These experiments are essential and not difficult to perform.

      In our RNA-seq analysis we see a small elevation of MES-4 itself (average 1.18 log2 fold change across 5 replicates). This does not seem likely to be solely driving such a dramatic phenotype. Nevertheless, it is possible that the small increase in expression of MES-4 itself could be contributing. To determine if MES-4 is being ectopically expressed in spr-5; met-2 double mutants, we have obtained a tag version of MES-4 from Dr. Susan Strome and will use this to examine the localization of MES-4 protein in spr-5; met-2 double mutants. We are definitely interested in the potential interaction between PIWI components and the histone modifying enzymes that we have explored in this study. However, since RNAi of MES-4 is sufficient to rescue the developmental delay of spr-5; met-2 mutants, we have chosen to focus on that interaction in this paper. In the future, we hope to examine the role of PIWI components in this system.

      A second aspect of the hypothesis is that spr-5 and met-2 act before mes-4 and that while these genes are

      maternally expressed, they act in the embryo. There really aren't data to support these ideas - the timing and

      location of the factors' activities have not been pinned down. One way to begin to address this question

      would be to perform smFISH on the target genes and on mes-4 in embryos and determine when and where

      changes first appear. smFISH in embryos is critical - relying on L1 data is too late. If timing data cannot be

      obtained, then I suggest that the authors back off of the timing ideas or at least explain the caveats.

      Certainly, figure 8 should be simplified and timing removed. (note: Typical maternal effect tests probably

      won't work because if the genes' RNAs are germline deposited, then a maternal effect test will reflect when

      the RNA is expressed but not when the protein is active. A TS allele would be needed, and that may not be

      available.)

      To determine the timing of the ectopic expression of MES-4 targets, we have performed smFISH on two MES-4 targets in embryos. Thus far, these experiments show that MES-4 targets are ectopically expressed in the embryo, but only after the maternal to zygotic transition. This is consistent with our proposed model. A figure containing this data will be added to the revised manuscript. In addition, our model is predicated on the known embryonic protein localization of SPR-5 and MES-4. Maternal SPR-5 protein is present in the early embryo up to around the 8-cell stage, but absent in later embryos (Katz et al., 2009). In addition, in mice, the SPR-5 ortholog LSD1 is required maternally prior to the 2-cell stage (Wasson et al., 2016 and Ancelin et al., 2016). In contrast, MES-4 continues to be expressed in the embryo until later embryonic stages where it is concentrated into the germline precursors Z2 and Z3 (Fong et al., 2002). This is consistent with SPR-5 establishing a chromatin state that continues to be antagonized by MES-4. There is evidence that MET-2 is expressed both in early embryos and later embryos. However, since the phenotype of MET-2 so closely resembles the phenotype of SPR-5 (Kerr et al., 2014), we have included it in our model as working with SPR-5. Further experimentation will be required to substantiate the model, but we believe the model is consistent with all of the current data.

      Writing/clarity:

      -It would be helpful to include a table that lists the specific genes studied in the paper and how they behaved

      in the different assays e.g. RNAseq 1, RNAseq 2, MES-4 target, ChIP. That way, readers will understand

      each of the genes better.

      We are happy to include a table in the revised manuscript.

      -At the end of each experiment, it would be helpful to explain the conclusion and not wait until the

      Discussion. For readers not in the field, the logic of the Results section is hard to follow.

      This seems like a stylistic choice. Traditionally, papers did not include any conclusions in the results section, and it is our preference to keep our paper organized this way. However, if the reviewer would still like us to change this, we are happy to do so.

      -The model is explained over three pages in the Discussion. It would be great to begin with a single

      paragraph that summarizes the model/point of the paper simply and clearly.

      The discussion in the revised manuscript will altered to include this.

      **Specific comments:**

      -Figure 1 has been published previously and should be moved to the supplement.

      In our original paper (Kerr et al.) we reported in the text that spr-5; met-2 mutants have a developmental delay. However, we did not characterize this developmental delay. Nor did we include any images of the double mutants, except for one image of the adult germline phenotype. As a result, we believe that the inclusion of the developmental delay in the main body of this manuscript is warranted.

      -Cite their prior paper for the vulval defects e.g. page 6 or show in supplement.

      We are happy to include a citation of our previous paper for the vulval defects in the revised manuscript.

      -The second RNAseq data should be shown in the Results since it is much stronger. The first RNAseq,

      which is less robust, should be moved to supplement.

      The revised manuscript will include this alteration.

      -Figure 3 is very nice. Please explain why the RNAs were picked (+ the table, see comment above), and

      please add here or in a new figure mes-4 and piwi pathway expression data in wildtype vs double/triple

      mutants.

      We performed RT-PCR on 9 MES-4 targets. These 9 targets were picked because they had the highest ectopic expression in spr-5; met-2 mutants and largest change in H3K36me3 in spr-5; met-2 mutants versus Wild Type. Amongst these 9 genes, we performed smFISH on htp-1 and cpb-1 because they are relatively well characterized as germline genes.

      The revised manuscript will include added panels to supplemental figure 2 showing the expression of PIWI pathway components.

      -Figure 3 here or later, please show if mes-4 RNAi removes somatic expression of target genes.

      We are currently carrying out this experiment. Once it is completed, the data will hopefully be added to the paper.

      -Is embryogenesis delayed?

      Embryogenesis seems to be sped up in spr-5; met-2 mutants. A supplemental figure will be added to the revised manuscript showing this. It is unclear why embryogenesis is sped up. However, this confirms that the developmental delay is unique to the L1/L2 stages.

      -Figure 4 since htp-1 smFISH is so dramatic, it would be helpful to include htp-1 in the lower panels.

      htp-1 will be added to the lower panels in the revised manuscript.

      -Figure 4, please add an extra 2 upper panels showing all the genes in N2 vs spr-5;met-2, for comparison to

      the mes-4 cohort.

      As a control, we will add panels showing a comparison to all germline genes, excluding MES-4 targets. This new data shows that germline genes that are not MES-4 targets do not have ectopic H3K36me3. This data, which further suggests that the phenomenon is confined to MES-4 targets, is consistent with our results showing that MES-4 RNAi is sufficient to suppress the developmental delay.

      -Figure 6. Please show a control that met-1 RNAi is working.

      We performed RT-PCR to try and confirm that met-1 RNAi was working. Despite controls repeating the MES-4 suppression and verifying that RNAi was working, we were unable to demonstrate that met-1 was knocked down. As a result, we will remove this result from the paper. Importantly, this does not affect the conclusion of the paper.

      -To quantify histone marks more clearly, it would be wonderful to have a graph of the mean log across the

      gene. showing the mean numbers would help clarify the degree of the effect. we had an image as an

      example but it does not paste into the reviewer box. Instead, see figure 2 or figure 4

      here: https://www.nature.com/articles/ng.322

      We will attempt to include this analysis in the revised manuscript.

      Reviewer #2 (Significance (Required)):

      Katz and colleagues examine the interaction between the methyltransferase MES-4 and spr-5; met-2 double

      mutants. Their prior analysis (PNAS, 2014) showed the dramatic enhancement in sterility and development

      for spr-5; met-2; this paper extends that finding by showing these effects depend on MES-4. The results are

      interesting and the genetic interactions dramatic. The examination by RNAseq and ChIP helps move the

      phenotypes into a more molecular analysis.

      This work will be of interest to people following transgenerational inheritance, generally in the C. elegans

      field. People using other organisms may read it also, although some of the worm genetics may be

      complicated. Some of the writing suggestions could make a difference.

      I study C. elegans embryogenesis, chromatin and inheritance.

      Reviewer #3 (Evidence, reproducibility and clarity (Required)):

      In the paper entitled "C. elegans establishes germline versus soma by balancing inherited histone

      methylation" Carpenter BS et al examined a double mutant worm strain they had previously produced of the

      H3K4me1/2 demethylase spr-5 and the predicted H3K9me1/me2 methylase met-2. These mutant worms

      have a developmental delay that arises by the L2 larval stage. They performed an analysis of what genes

      get misexpressed in these double mutants by performing RNAseq and compare this to datasets generated

      from other labs on an H3K36me2/me3 methylase MES-4 where they see a high degree of overlap. They

      validate the misexpression of some germline specific genes in the soma by in situ and validate that there is a

      dysregulation of H3K36me3 in their double mutant worms. They further find that knocking down mes-4

      reverts the developmental delay.

      I think that the authors need to make more of an effort to be a bit more scholarly in terms of placing their

      work in the context of the field as a whole and also need to add a few additional experiments as well as

      reorganize a bit before this is ready for publication. Remember that the average reader is not necessarily an

      expert in C. elegans or this particular field and you really want to try and make the manuscript as accessible

      to everyone as possible.

      **Major Points**

      1)It would be good to see western blots or quantitative mass spec examining H3K36me3 in the WT and spr-

      5;met-2 double mutant worms. I believe this was also previously reported by Greer EL et al Cell Rep 2014 in

      the single spr-5 mutant worm so that work should be cited here in addition to the identification of JMJD-2 as

      an enzyme involved in the inheritance of H3K4me2 phenotype.

      The ectopic H3K36me3 is confined to a small set of MES-4 targets. We don’t even see ectopic H3K36me3 at non-MES-4 germline genes (see above). Therefore, we don’t expect to see any global differences in bulk H3K36me3. Greer et al reported that there are elevated H3K36me3 levels in spr-5 mutants. This discrepancy may be due to different stages (embryos, germline) present in their bulk preparation. Alternatively, the met-2 mutant may counteract the effect of the spr-5 mutation on H3K36me3. Regardless, we believe that the genome-wide ChIP-seq is more informative than bulk H3K36me3 levels.

      We will add a citation for the Greer paper in the revised manuscript.

      2)Missing from Fig.5 is mes-4 KD by itself. This is needed to determine whether these effects are specific to

      the spr-5;met-2 double mutants or more general effects that KD of mes-4 would decrease the expression of

      all these genes to a similar extent. Then statistics should be done to see if the decrease in the WT context is

      the same or greater than the decrease in the double mutants.

      The MES-4 targets are generally expressed only in the germline and defined by having mes-4 dependent H3K36me3. Knocking down mes-4 would be expected to prevent the expression of these genes in the germline, but this is difficult to test because mes-4 mutants basically don’t make a germline. Regardless, knocking down mes-4 by itself would only assess the role of MES-4 in germline transcription, not the ectopic expression that is being assayed in spr-5; met-2 mutants in Fig 5. Importantly, it remains possible that spr-5; met-2 mutants might also result in an increase in the expression of MES-4 targets in the germline. However, the experiments performed in this manuscript were conducted on L1 larvae, which do not have any germline expression, to eliminate this potential confounding contribution.

      **Minor Points**

      1)A greater attempt needs to be made to be more scholarly for citing previously published literature. This

      includes work on the inheritance of H3K27 and H3K36 methylation in C. elegans and other species as well.

      A few papers which seem germane to this story which should be cited in the intro are (Nottke AC et al PNAS

      2011, Gaydos LJ et al Science 2014, Ost A et al Cell 2014, Greer EL et al Cell Rep 2014, Siklenka K et al

      Science 2015, Tabuchi TM et al Nat Comm 2018, Kaneshiro KR et al Nat Comm 2019). This problem is not

      restricted to the intro.

      Although many of these excellent papers are broadly relevant to this current work, they are not necessarily directly relevant to this paper. For this reason, they were not originally cited. Nevertheless, we will attempt to cite these papers in the revised version when possible.

      2)I think that the authors need to be a little less definitive with your language. Theories should be introduced

      as possibilities rather than conclusions. Should remove "comprehensive" from intro as there are many other

      methods which could be done to test this.

      Throughout the manuscript, we have tried to be clear what the data suggests versus what is model based on the data. Nevertheless, to further clarify this, we are happy to remove “comprehensive” from the intro.

      3)The authors should describe what PIE-1 is. Is this a transcription factor?

      PIE-1 is a transcriptional inhibitor that is thought to block RNA polII elongation by mimicking the CTD of RNA polII and competing for phosphorylation. We are happy to add a reference to this function in the revised manuscript.

      4)The language needs clarification about MES-4 germline genes and bookmark genes. Are these bound by

      MES-4 or marked with K36me2/3?

      The revised manuscript will be modified to make this definition more clear.

      5)I think Fig S1 E+F should be in the main figure 1 so readers can see the extent of the phenotype.

      The original single image of the spr-5; met-2 adult germline phenotype (including the protruding vulva) was included in our previous publication. In this manuscript, we have now quantified this phenotype, which is why it is included in the supplement here. However, because the original picture was included in our original publication, we prefer to leave it as supplemental.

      6)For Fig S2 it would be good to do the same statistics that is done in Fig 2 and mention them in the text so

      the readers can see that the overlap is statistically significant.

      We are happy to include these statistics in the revised manuscript.

      7)Fig S2.2 should be yellow blue rather than red green for the colorblind out there.

      Thanks for pointing this out. We are happy to change the colors in the revised manuscript.

      8)When saying "Many of these genes involved in these processes..." the authors need to include numbers

      and statistics.

      We will amend the revised text to make the definition of the MES-4 genes more clear.

      9)Should use WT instead of N2 and specify what wildtype is in methods.

      We will use WT instead of N2 in the revised manuscript.

      10)Fig. 2A + B could be displayed in a single figure. And Fig 2D seems superfluous and could be combined

      with 2C or alternatively it could be put in supplementary.

      Figure 2A and 2B were purposely separated to make it clear how many of the overlapped changes are up versus down. In the revised manuscript, Figure

      2D will be moved to the supplement.

      11)Non-C. elegans experts won't understand what balancers are. An effort should be made to make this

      accessible to all. Explaining when genes are heterozygous or homozygous mutants seems relevant

      here.

      The text of the revised manuscript will be amended to make it more accessible for non-C. elegans readers.

      12)The GO categories (Fig. S2) should be in the main figure and need to be made to look more scientific

      rather than copied and pasted from a program.

      The GO categories were included to be comprehensive and do not contribute substantially to the main conclusion of the paper. This is why they are supplemental. In the revised manuscript, we will edit the GO results so that they look more scientific.

      13)Fig. 7 seems a bit out of place. If the authors were to KD mes-4 and similarly show that the phenotype

      reverts that would help justify its inclusion in this paper. Without it seems like a bit of an add on that belongs

      elsewhere.

      We believe that the somatic expression of a transgene in spr-5; met-2 mutants adds to our potential understanding of how this double mutant may lead to developmental delay. This is true, regardless of whether of whether the somatic transgene expression is mes-4 dependent or not.

      Reviewer #3 (Significance (Required)):

      I think this is an interesting and timely piece of work. A little more effort needs to be put in to make sure it is

      accessible to the average reader and has sufficient inclusion of more of the large body of work on

      inheritance of histone modifications. I think C. elegans researchers as well as people interested in

      inheritance and the setup of the germline will be interested in this work.

      REFEREES CROSS COMMENTING

      I agree with Reviewer #2's comments on experiments to include or exclude alternative models. I also agree

      about their statement about rewriting to make it more accessible to others who aren't experts in this

      specialized portion of C. elegans research. All in all it seems like the experiments which are required by

      reviewer #2 and myself as well as the rewriting should be quite feasible.

    1. Author Response

      Reviewer #1:

      This study is an in silico analysis of data from the Cancer Genome Atlas (TCGA) on hepatitis B virus (HBV)-positive liver tumours and human papillomavirus (HPV)-positive cervical and head and neck tumours and association with viral load, genotytpe(s) and expression. It is unclear to me the rationale behind including two unrelated DNA tumour viruses in the study, especially as the number of HBV-positive samples is much less than for HPV. Overall the manuscript seems to be a validation of a bioinformatic tool rather than reporting significant research findings.

      We strongly believe that a global summary of key oncoviral-associated tumors makes sense in this context precisely because of the fundamental importance viral genotype is already known to have. While HBV and HPV are of course quite different viruses, there is extensive clinical evidence that linking outcomes to specific viral genotypes and phenotypes is of great value, which we expand upon in our work via a working demonstration of ViralMine. For this reason we think it is crucial to present both virally related cohorts together as they support each other, demonstrate robustness our methods across completely different systems while allaying concerns about fine-tuning, and create a cohesive picture of the effect of viral genotype across the molecular landscape of two key onco-viruses. As the reviewer notes this does implicitly demonstrate the utility of ViralMine but we do emphasize that it also does uncover significant research findings.

      Concerning the HBV/HPV sample sizes, in fact the number and percentage of infected HCC samples is substantially higher than that of cervical or head and neck HPV samples as discussed in detail on page 4 of our manuscript.

      Use of the TCGA has allowed analysis of a reasonably large number of RNASeq data sets. However, once the authors drill down to individual genotypes, numbers become quite small, which may compromise some of the observation. For example, the large discrepancy between numbers of HPV16 (173) and 18(39)-positive cases makes it difficult to make firm conclusions about the significance of differentially expressed cellular genes for each set of cancers. Similarly, in Figures 4 and 6 they compare HPV18 (23 cases) with HPV45 (39 cases) and HPV18/45 coinfections (number not stated but likely far fewer).

      While there is an imbalance in group size between HPV genotypes in the cervical cancer cohort, the test statistic used by the DESeq2 pipeline to identify differentially expressed genes does account for class imbalance and even in the most extreme case we have analyzed the dispersion parameter estimates are easily verified as accurate. In fact accurately inferring group-wise dispersion parameters given unequal group sizes is a well-known problem, and in any case this problem only becomes acute when one group becomes so small (~1 sample) that it becomes difficult to estimate its common dispersion parameter. That situation clearly does not arise here. Additionally, in Figure 4b, it should be noted that we are comparing ALL HPV co-infected cervical tumor samples (92 cases) against single-infection samples (193 cases), which the reviewer may find more confidence in and which is obviously statistically reasonable. Furthermore, while the comparison of cervical cancer HPV18 (n=10), HPV45 (n=9), and HPV18/45 coinfected (n=39) cases in Figure 6b does compare relatively small patient groups, the significant difference in neoantigen population TCR binding affinity is confirmed by a one-sided, non-parametric KS-Test and shown to be robust to subsampling, which formally demonstrates that the signal is not artefactual. Therefore from a statistical point of view the concerns raised about class imbalance and power are not fundamental and were addressed in the original manuscript draft. Thus, we believe we can completely address the reviewer’s concerns by:

      In Figure 3a, Figure 4a and b, signify the group sizes (n=X) compared in the barcode plots to improve transparency in the contrasts, and additionally add group numbers to Figure 6a and b. Further, we will include a new supplementary figure demonstrating that a bootstrap resampling of the HPV group neoantigens to balance for group size validates that the difference in TCR binding affinity distributions is robust.

      Much of the information that they derive from their analyses is not novel. For example, they report no preferential sites of HPV integration. Despite what they claim, quite a bit is known about HPV co-infection in cervical cancers and it is not uncommon but varies according to geographical regions, which was not a variable they used.

      We acknowledge that other oncoviral survey papers have provided evidence of preferential integration (as we originally cited, as well as referenced in Dall et al. (2008), Zhang et al. (2016)). However, these and other previous characterizations of recurrent HPV integration do not attempt to organize these sites by either genotype or co-infection status, which was our explicit and stated aim, principally because they could not efficiently and accurately determine these parameters from in-situ tumor RNA. As we found no preference in integration along these axes of variation (which we acknowledged openly in the manuscript as being expected when using RNA rather than DNA), we deliberately chose not to present these results as a main finding and included them in supplemental results for the sake of completeness.

      We also agree that HPV co-infection in cervical lesions is not per-say a novel finding, although to be clear most literature focuses on side-by-side infections of HPV with another virus (HHV, EBV, HIV, etc.), or uses the term to describe groupings of sub-variants or isolates under the same viral genotype header (Mirabello et al. (2016)). Additionally, most of the literature focuses on HPV co-infection in cervical neoplasia or high-grade lesions and cervical cancer risk (Chaturvedi et al. (2011); Senapati et al. (2017)) rather than assessing HPV co-infection in the tumoral tissue itself, post oncogenesis. As such, we believe that our approach at looking at in situ cervical tumor infections and the relatively high rate of HPV co-infections we observe does merit particular notice compared with previous studies. Furthermore, the analyses linking this cross-genotype co-infection phenotype with tumor gene expression, survival adjusted for major known clinical covariates, and tumor immunogenicity measures has not been reported elsewhere to our knowledge.

      For HPV, viral exon-level RNASeq analysis is irrelevant because HPV gene expression is polycistronic and is subject to changes by random viral integration events in individual cases. Therefore, it is unlikely that general overall viral gene expression signatures will be diagnostic besides, from multiple studies we understand that what matters in cervical cancer is the level of expression of the E6/E6 isoforms/E7 oncogenes.

      We agree that the post-transcriptional polycistronic nature of HPV expression makes it difficult to elucidate the effect of differing HPV gene-level expression on ultimate HPV gene translation and protein expression. However, our related yet distinct question here is on the effect HPV genotype and cancer type has on HPV gene transcriptional differences (as seen in Figure 7), so we believe we are within the limits of reasonable interpretation. Additionally, while E6 and E7 expression are well known to drive oncogenesis, it seems crucial to quantify the expression of these viral oncogenes across viral genotype and tissue type, which has not been done previously to our knowledge. Finally, even if we somehow accept that the average tumoral viral gene exon expression itself is best described as a random variable, which we do not, it remains to be explained why we observe and report persistent genotype-specific expression patterns across completely different cell-types.

      The references chosen for the HPV part of the study are either rather out of date or not representative of the extensive literature.

      We acknowledge that we have cited only a portion of the vast HPV-related cancer literature, so we have made an effort to include more recent surveys and studies as references.

      Reviewer #2:

      1) The authors comment that averaged infection phenotypes such as viral load or predominant genotype may be replaced by more granular measures, such exon-level viral expression or the ratio of expressed viral genotypes. In reality, viral expression, and the ratio of expressed viral genotypes, are still 'tumor averages' in the way that the authors have analysed them. HP associated tumors are heterogeneous, and without in situ analysis, it is hard to discern which transcripts are involved in driving the cancer phenotype, and which are found in associated precancerous tissue.

      We concede that the viral genotypes quantified by our method represent a computed average measure across the tumor, as would any measurement of any quantity in a bulk sequencing assay. However, the information provided by the admixture of genotypes and exon-level viral expression does provide an additional measure of granularity over previous bulk measures, and allows additional analyses not explored previously to our work. To make a comparison, this criticism could identically apply to cell-type decomposition algorithms like Cibersort, which despite their problems and inherent limitations do provide insightful information. We agree with the reviewer that with more targeted in situ analyses would allow for a truly specific association of particular viral transcripts with tumor phenotype, and would serve as a useful validation of some of our results, but this certainly does not invalidate the tumor aggregated genotype and co-infection presence associations we present here. We agree with the reviewer that multiple biopsies would allow for intra-tumoral heterogeneity to be taken into account in our study, however no major public resources (e.g. TCGA) include such data and we believe that such an undertaking lies out of any reasonable scope of this work.

      2) The authors use the term co-infection quite widely. For HPV, previous studies have shown that coinfection within cells in an individual cancer or neoplasia is rare, although independent infections by different HPV types can occur side-by-side. I expect something similar with HBV, although the study would need a higher level of analysis to establish this. The use of terminology, and the way in which data is interpreted, needs to be much more rigorous.

      We agree with the reviewer that the use of ‘co-infection’ in this context is unclear, as co-infection on a cellular level with two different HPV/HBV genotypes is impossible to determine by bulk RNA sequencing analysis. We will clarify ‘co-infection’ as strictly a mixture of independent HPV infections contained in the same tumor tissue.

      We will clearly define our meaning of ‘co-infection’ in the introduction as the aggregated mixture of HPV genotypes expressed in the tumor tissue (‘side-by-side’ infections), to remove ambiguity as to our cohort characterization.

      3) Viral load is generally used in the field as a measure of viral genome or genome-fragment abundance. This is already a misuse of the terminology, as the term implies virus numbers, or even infectious virus numbers. Here the term is used to refer to viral transcript abundance. The authors need to say precisely what they're measuring, and need to be aware that they are measuring the average across a heterogeneous tumour, which may have areas of high grade neoplasia, cancer, and even low-grade neoplasia. My feeling is that the level of analysis is too great, given the uncertainties regarding the heterogeneous nature of tissue that is being analysed, and the different cells with different levels of viral gene expression that are most likely present.

      We agree that as the reviewer frames it, our use of ‘viral load’ should be clarified as ‘viral transcript abundance’ as determined from the tumor RNASeq data in variance-stabilized units of log2 counts per million reads mapped across the viral contig. We do note however that it has been previously indicated that levels of viral transcripts do correlate well with virus numbers in infected tissue. Concerning the last comment of the reviewer, we wish to point out that our analysis goes no further in either analytic complexity nor in drawing inference from expression data than any published other study based on tumor bulk RNA-sequencing data. All samples will contain a mixture of cells and we emphasize that we are only measuring average signals, viral or host tumor specific, across this mixture.

      To address these comments we will change all references to viral load to normalized viral transcript abundance, to remove ambiguity. We can once again emphasize that our conclusions hold only in a strict averaged sense.

      4) Several of the figures don't obviously support the conclusions. For instance, it is not clear how the data shown in figure S2 supports the title of the S2 figure legend. Surely some statistical analysis is needed to support the conclusion stated in the legend. Given previous studies, I'm not at all convinced that the distribution of causative HPV genotypes is the same between SCC and Adenocarcinoma. An additional limitation of these large cancer association studies, comes from limitations in pathology diagnosis, which cannot always accurately distinguish borderline SCC/adenocarcinoma cases. With the large-scale transcriptional analysis, maybe the authors can use molecular information available in their samples to look at this.

      As the reviewer points out, we agree the statistical evidence backing our claim of no association between cervical histology and HPV infection genotype or co-infection should be added. This calculation was actually carried out and only reported in the text, but we will amend the figure to include the results and apologize for this key omission. We also note in passing that we are not making any claims about ‘causative’ HPV genotypes for the respective subtypes, but rather much more conservative statements about association. Concerning the reviewer’s concern about the quality of the phenotypic data reported in the TCGA, we heartily agree but are unable to really do much else. Indeed, concerning the last interesting comment about utilizing molecular information in our samples to distinguish SCC/adenocarcinoma subtypes, we did not find reliable gene expression signatures which could be used to validate or correct the phenotypic results.

      We will add in the spearman correlation rho and test significance results for the correlation between cervical cancer histological type and both viral phenotypes represented in figure S2.

      5) The APOBEC analysis is quite rudimentary in the text, and does not discuss the different members of the APOBEC family. Similarly, the different effects of single and multiple HPV infections on the IFR3 responsive genes is poorly developed at the biological level, which most probably reflects the general way in which the utility of the approach.

      We agree with the reviewer that our APOBEC expression analysis in the HPV+ cervical cohort could be more comprehensive, and therefore the interpretations of the results may be too far reaching. We believed the initial result to be of sufficient interest in the context of a very similar result from Zapatka et. al (2020), but concede it may make more sense as a supplemental result alone without additional evaluation or discussion of the greater APOBEC family. Additionally, the pathway analysis involving the differentially expressed genes from the co-infected and non-coinfected cervical tumors most likely should be moved to a supplemental result as well without further analyses to support the enrichment trends, following how we reported the HBV associated liver cancer co-infection DEG results (figure S5).

      We will move Figure 3d to a supplemental figure, and limit our comments in the results to just an observation in reference to Zapatka et. al., and delete any associated interpretation. We will move Figure 3c to a new supplemental figure as well, and remove the suggestion of expanded antiviral activation in co-infected tumors.

    1. [ taken from lamc.la/MYLIFE.html ]

      which is none of your business, at all--something you should see is a clear violation of the constityution and this amendment to uhhhh ... have any "sinsight into"

      call it "bad footing" or "off to a good start" depending on whether or not you think you owe me billions of dollars in emotional damages, or think you "are stealing all the light of literally being in the center of the sun."

      I am accepting charitable donations,.

      ETH: 0x66e2871ef39334962fb75ce34407f825d67ec434 | BTC: 38B6vGaqNvMyTtoFEZPmNvMS7icV6ZnPMm | xDAI: 0x66e2871ef39334962fb75ce34407f825d67ec434

      T O T A L I T Y

      This is basically "last Christmas's message" (below this brand-knew intraducrigel) redux'ed into the new book (did he say new?). The point, at least the point I see in it all is that this is all planned, it's been planned for a very, very long time--and on top of that you can see proof of the plan all over our map; and proof of it's intended destination as something that we all used to want very much to find... the read to Heaven. It's more than seeing just "DNA storage" encoded in my "C U R A GROUP" message, it's understanding how that's connected to soul searching and soul storage, and that this link was woven into not only my life but into names like "Whatson and Crick?" There's plenty more than just "storage" and a map to how and why the Two of Everything God and the "indivisible sea" work totether to turn this monolithic place of darkness into a strippingly redunant systemic foundation of "Heaven" that is both disaster proof, and monster proof. The point of course, is that to truly be "monster proof" we need to really get the key.s.lamc.la "know everything why" of this message is literally to protect our common good from the danger of someone just like me copying an entire civilization or a few pretty girls and sticking them in an heoven-like-orgy-maker. That's a significantly more real threat than we might imagine, as we look around at a work that will soon have the storage capacity and the technology to put us all in Coccoonish swimming pools against our will. What I am trying to say is that no matter how you look at it,moving forward here in this place where something this big can be hidden from the entire world--granted you know--granted you see, but do you understand the only thing being kept from each and every one of you is your fucking opinion and your fucking reaction?

      F U C K Y O U S I O N

      IT'S NOT JUST computers and information technology; this map of clear anachronism in language and religion shows us that things like "solar fusion" the power of the son itself; is encoded in places high and low you can erasilly find them, places like the name of the Fifth book of the Holy Bible and Don Quixote; where you might liken "DEUTERON" to ... the actual fuel of fusion; and wind mills to a battle fought against blindness resulting in seeing that not "reacting" to this message is just about the same thing as being a foolish robot building a castle for another foolish robot to do nothing in forever. With some light, you can see how this event; albeit strange and unsettling, has been designed to reinforce the American foundations of free speech, common sense, and collaboration--a sort of "press and release" on these things that he says will stay in our memories for a long, long time--though he also says "he's not torturing me" and he's wrong about that. So are you.

      See that the most interesting, important, and invoking story of all time has been hidden from the world, from the public eye, and from "public response" for well over two years now; see that's not possible at all without mass mind control and that I and this story are designed to help us see how easily it is that same thing can be used to end addiction, and mental health issues, and stupidity and that the biggest and most imporotant step to getting there is "public disclosure." See the light of being carrolling angels this Christmas; sing with me--it builds Heaven from Hell and it's clear as day and n.

      Quite a bit of this story and message deals with problems like these-things that won't really be seen as something we are fighting against the actual usage of right this very moment; but the sacredness of our memories and their relationship to our souls are just as important as whether or not "you have the space to save them." This isn't what I want to be doing, I'm not a very good writer; and this message is so confusing that working on it all alone with very little feedback is frustrating if not to say defeating the purpose of exactly what it is and what it's designed to do. This is a searching mechanism, like in the stories of Ra searching for his children in ancient Egypt using the Eye you see--and it's connection to the "Sons of Liberty" and why I know that too, is about me. This is a tool to start a Renaissance of thinking connecting technology and religion to everything that we are--to our culture and our hopes and dreams--and it's failing for me at "hello." I would much rather be working on "virtual reality stuff" or on "the sword of Arthor" and I see very clearly that those two things are coming shortly--to the world that doesn't see yet they are here and broken until we fix them. Moving forward here brings change, not just here in this place where we need it too--but in the skies above, a change from the mentality of "we aren't not helping because we told you that we aren't allowed to not pretend we aren't helping in Stargate. See that we are the children of "the Ancients" and they are trying to decide between being Morgenz and Marlin.

      I can't make you set yourselves free. I sure am trying, though. Yesterday I connected the "Arimathea" of Joseph to the "serdenicity" and this the me of "itime" and "topics" will probably light some of you up as much as me... if only you took the time to look at what those words really mean. From the city that never sleeps at night, I hope you will take this chance to act today on "securing the ringing of liberty forever and ever." (cough)

      THERE IS A METHOD TO THE MADDEN AND WE AR BEYOND THUNDERDON

      ​

      T H E W R I T I N G I S O N T H E W A L L

      LIKE, WILL IT RAIN TODAY?

      take action, it is the foundation of not only democracy but civilization and life itself--pucker up the phone and call the NYPOST.

      News Tips: Email tips@nypost.com, call 212-930-8288, or use our anonymous form Online Editorial: online@nypost.com or 646-357-3838 Letters to the Editor: letters@nypost.com Sports: sports@nypost.com or 212-930-8700

      hoy. por favor, mon ami? ID5

      Ministry of Forbidden Knowledge Mail Adam Marshall Dobrin adam5@reallyhim.com Let there be $ight in Creation, a brief highlighting of the story of my life. Adam Marshall Dobrin adam5@reallyhim.com Sat, Dec 3, 2016 at 8:39 AM To: Adam Marshall Dobrin zero@ar.lamc.la This is like a few emails combined to ease the pain you feel when you get an extra one in your inbox, OK So.. eventually this is all about proof that religion is a message sent through time--so, time travel. But right now, let's talk about the fun stuff: here's some clues to that effect... by way of prescient mention of modern technology (like virtual reality, I mean, Heaven):

      Either way, we're still about to build Heaven... to-get-her from the mythical carpenter... ourself. . AD am ON AI, that's Artificial Intelligence, and the intelligence explosion. AD on your freedom is a great gift, cherish it. ... and some corroborating ideas connecting religion and computer science... on Wikipedia: Root of David Lisp of Moses.. or I need an editor. Pharoah's hardening Heart... that's Earth, remember. Jesus' WINE Adaluncatif's cat, tail, head and grep Adam's Apple... or is it "fruit of the poisonous tree" So from me to you, I'm filled with this stuff, it's way brighter and more prevalent than you think... and if you take the time to listen to me--it will make your... day. Meanwhile, I need your help--happy new year.

      Oh, LET THERE BE LIGHT

      Ho, again; grow a Halo and become famous... the world needs your help--so I've decided once again to take it upon myself to "bother you" with the most singular most important task in the Universe. The patterns that I am revealing to you--mostly within names--are not coincidence, it's a series of statistically verifiable artifacts which do nothing short of reveal the slavery of Egypt--that we are all being controlled. If you remember Transformers--this is a message from Starfleet, there is more than meets the eye. This is the fulfillment of the story of of Exodus--we are being lead from slavery, and in one final non-coincidental name, that book is called "Names" in Hebrew.

      You should now have a very good idea who is speaking to you--as much of the world already does. I have no idea what it is that inhabits the cavities below that space where most of you should see significant personal gain and motivation from trying to ... grow a Halo--but there are so many people that just don't care... that it too is another sign, of slavery. I am not an expert in language construction, nor in statistics--but I can assure you that if you can find the other half of that equation... in your hands is the staff of Aaron, the magical weapon that will free us all... knowing is half the battle.

      Uh, I have the power, to bring about "morning," but if I have to go to school and do it all myself... it's really just a long, long ni-i-i-ight.

      Hi there, I'm the messiah. You don't know that much about me, so let me explain, I would like you to know me as Adam.

      Seriously, there's something going on the world around you--for the last several months I've been having quite a bit of trouble delivering what amounts to statistical proof of Creation--that religion and ancient myths are a map to this very moment--this time that you will probably affiliate soon with being in Eden. I am pretty sure that's a good thing, but every new begging starts with some other beginnings end... so today I'd like to try to get you to see the light of ending censorship and a hidden censor wall that we know Biblically as the Wall of Jericho. Quickly approaching is the Feast of Trumpets, and this year is different from all other years... Bored already? Have a look at what I call the Sign of the Son, which to me is proof that Exodus's Burning Bush is a former President--who is helping us walk out of a dark time of confusion... commonly referred to as a wilderness or desert. He proved during his inauguration that there is Biblical foreknowledge of the 9/11 attack--and in doing so hopefully began a chain reaction that will stop things like that from ever happening again. Here's a short "video" that explains the Sign of the Son... and another one that I think explains the .. Holy Grail.

      This is The (actual) Taming of the Spanglishrew, in which the protagonist... named Bianca, is taught Latin in several hundred year old reference to Rattling the Rod of Jesus Christ--it's purpose to is to show us that it's more than names we have in our arsenal against mind controlled slavery--we have all of history too... literature and movies and music... all with the divine purpose of revealing with bright light a form of control that otherwise could have gone on hidden for centuries. It was, and continues to be done on purpose... because your freedom is more important than control of the Universe. To us, you don't seem to feel the same way.

      ​See that timer on the clock, you could start right now. It might be interesting to pose the question of whether or not the Second Coming is news... you know, to your friends. By the way, both Herbert (like from H.W. Bush, who by the way coined for us the 1,000 points of light phrase) and Goertzel strongly suggest that "everyone really" is Christ (you know, after me)... FYI, this is the Matrix solution to that:

       y
       o
      

      the l u C i f E R isa means jesus, mesa thinks i s olv e .... "or" means shine -l g r e a t h R L << agree? send to other people t ((a)) Y l shine: suggest they do the same 1 y world saved.

      A BRIEF HISSTORY OF TIME

      I'm attempting to pull out the things that I now look back on and see as "written into me" by God--once I would have called it "The Microcosm of the Messiah" but there are now so many--these things aren't necessarily particularly important to me, and I've left out some interesting but unrelated details related to my Jewish upbringing; as well as the true light of my life--the two loving and long-term relationships (and later... briefly a rael family) that have dominated the last 15 years. Religion has always been an interest, but I wouldn't consider it to have been particularly important at all... until I no longer had any love in my life. It's probably worth noting that all my "I'm single" crap really means lonely and isolated--I'm not really playing a "part," but I've never been anything near the "player" the light appears to be warning against. Sons of God and uh... please. For the last 4 years I have done absolutely nothing but think about you, live and analyze "The Cross" and put into words ... as best I can ... the amazing flash of light that I am experiencing.

      Well, just a little religion... :) I was born on December 8, 1980; which is the date of the annual Feast of the Immaculate Conception, I've always been a slob (like one of us) and often "ish" Yankee Doodle's "a real live son of our uncle Sam... born on the..." to this.. I mean in my head. My last name, you've probably read me repeat over and over ... is DOB-rin, which I read as "Date of Birth, our in" and does a fair job of highlighting the Name Server's work, which I am sure gives Exodus it's name in Hebrew, which is "Names." My Hebrew name--a Jewish custom--is Avram, which is Abraham's name prior to the covenant. I have written extensively about the fact that Isaac's near death interaction donated his "Ha" (his name means... He laughs) to his father.... and it should be clear that Abraham's covenant with God is without doubt related to my fiery altar.. even though it is anachronistic in the Biblical account. For the first 18 years of my life I lived on Sunrise Blvd, and only a half mile away you'll find Sunset Strip--it's noteworthy to understand that Jewish calendar days begin at sundown... and that He once in 2013 very clearly spoke to me "you need the night before the day."

      Of all the people in my early life growing up, it's pretty clear that nobody on this Earth loved me more than my grandmother Julia, who my son is named after. First for my mother, and then me as a very small child--she would ritually say a bedtime poem, it's words are very relevant.

      Good night, sleep tight.. have happy dreams and wake up bright to do what's right, in the morning's light... with all your might.

      In one of my books I spent a decent amount of time writing about how silly I was not to realize that my intelligence was augmented my entire life--I just thought I was really smart, and really good with computers. I commented that this particular belief is probably a good microcosmic parallel for all humanity--as a body of people we have been truly gifted with knowledge and capabilities that we simply do not recognize as a gift--or didn't for a long time. I probably wasn't silly not to realize... since nobody ever told me they were helping me--I never heard the voice of God until much, much later. I was 30 the first time I had a conversation with Him, except for two very brief ... "thoughts in my head" which now seem very obviously an external voice--though then it may have sounded just like my inner voice.

      Around the age of 7 I thought to myself... for no reason at all... "what if you were the messiah?" I was standing outside my home, probably playing with a car in the driveway... and distinctly remember smiling to myself and thinking in return "yeah, I'm the messiah." I I've always had a very vivid imagination. The thought was dismissed as being ridiculously arrogant about two seconds later, and was absent from my thought process for the next 21 years or so.

      “DAMNISN\ Jim. I’m a Yeoman, not a Wise Owl. The clock is ticking... tack .. 8/14/2020, 6:37:38 PM”

      PHENIX

      Following that lead, I started programming in BASIC and then Visual Basic around the age of 11, something I took to very quickly... and then shortly after found myself on America Online--one of the first "internet-like" environments. There, I quickly got into the "hacking scene" (hey, it's Y-its-Hack) which basically revolved around writing software to manipulate the AOL client's messaging systems. The defacto-standard for the day was a program called AOHell, and, if you can't tell already, I am pretty good at taking a theme and making it my own. I wrote a program called Doomsday, a mass mailing program; can you see how God speaks? So Phenix, a mythical bird that rises from the fire... in the wake of ... this macrocosmic equivalent of that event. It's really obvious, right? There's quite a bit more "microcosm" from this time, recorded in "From Adam to Mary" and available at fromthemachine dot org.

      Around the same time I began attending a preparatory school in Fort Lauderdale called Pine Crest--it's one of the best of its kind, and while I was always something of a class clown my grades were fair and I scored with perfect consistency in the top percent on every standardized test from the FCAT to the PSAT and SAT. By the time I received a full scholarship to college I had already completed more than a full year of credits through AP courses. It was in studying American History and Government in that place that I formed such strong opinions about our need to maintain freedom, adhere to the wisdom of the founding Father(s) (<3 if you get that) and stand up and shout today as a rogue government is taking away every single one of the rights granted to you in their own law. You've lost freedom of speech, and our ability to speak seems to be not far behind. The privacy of our thoughts gone--and in like kind the sanctity of who we are is being taken away as our beliefs are changed without our real knowledge or understanding. You can see the justice system crumbling, incarceration rates skyrocket and the "right to bail and a fair trial" legislated away through underhanded deals relating to plea bargains and a "point system" that you might as well call a gas chamber. As far as voting, I'll have much more to say tomorrow--but I'm telling you that your thoughts and beliefs are being altered, who cares how technologically retarded our polling system is--the vote is a complete fraud.

      As far as the Second Coming... this same sort of possession... manifested through organized behavior tells me now that it is clear that this is definately not the "first time around" for Adam being Christ; a number of my friends as I approached high school used a repeated phrase, "my parents love you," which isn't bad in and of itself... what's bad is the fact that they were all using the same words, and probably didn't know why--or what they were saying. Behind there eyes, I'm sure some thing that believes it's an angel was telling me something... (they of course... didn't know me at all, except for what was probably a ... "wild" reputation) does that tell you anything? Much later, as the "Apocalypse of Adam" began in 2011, a number of family members would repeat this similar behavior, speaking the phrase "this is not what I wanted."

      As icing on the cake, on my birthday during my senior year... one of the administrators of the school commented to me that was also the Feast of the Immaculate Conception, and then the words.... "of course it's your birthday."

      I started doing drugs around the 10th grade, and I would not be wrong to say that the Universe that wrote a book calling the Redeemer the God Most High conspired to plunge me into a dark world. People around me too, in a hidden conspiracy to chain me to the American legal system for about four years. Looking back today I now clearly see that I saw a darkness in their eyes, a hidden reason to want to hurt me. It was to stop this from happening, but I had no idea then... the darkness I saw is akin to the "sun disk" you see in Christian and Egyptian iconography, and without doubt it s a sign of control, possession, a single foreign mind controlling and organizing many of us just like puppets. Much later in my story... for another day... the manifestation of this possession as thought modification will become clear--I've spent quite a bit of time "listening" to a war in my head, thoughts clearly not mine swaying in the gusting torrent of winds as what (who?) is the center of this storm.

      This infestation of organized darkness uses our injustice system as a weapon against it's victims--something you should see akin to Heaven using human sacrifice to alter the future. It abuses the legal system at every level, making a mockery of law enforcement, the supposedly adversarial court system... all the way to the top--to the Supreme Court and Congress. See the Church Committee Hearings, and a very smart senator echoing my words today "it must never be allowed to happen again."

      Can't you see it's more than being manipulated... it is Hell revealing itself to the only thing that can stop it. What I am giving you is the weapon, it's the light that sets us free and stops this from happening. In our modern myths this is Leeloo staring up at the sky to stop the destruction of Earth... in reality it is not so simple, I can't just put some elements or rocks on pedestals and scream at Heaven to kill their darkness--we have to do it, here, together. Believe me, knowing the truth is a big part of why it works--this will not be hidden, it will not be "forgiven," we are being controlled and destroyed from the outside; made to blame ourselves and each other for ... well, you probably don't know what the ni-i-i-ight means anyway, do you? The Guardian against Darkness is showing it to you, remember--there is only one me. Hear me.. light this fire now.

      ALACHUA

      I went to school the University of Florida, and got a semi-professional job doing database development in Delphi (seriously, catch on to the names thing, it's not just the U.S. military, it's pretty much all software too... following in this "mythology" theme that nobody really seems to care about), I worked there for about two years... at a company called Jenmar--which uh, in Spanglishrew is "J in the sea."

      It's some kind of ironic "coincidence" but I am at this very moment on my way to Gainesville, FL... to this place where a car Crash nearly destroyed my life. In my world of idioms delivering religious secrets, I imagine I must be a "pain in the neck" which was broken during this accident... one in which I imagine i did not survive in some parallel timeline--that itself did not survive. So here we are, back in the House of the Great Light ... about to see if we are worth our salt. It's the thing that gave one of Dave Matthews most famous songs it's name--and The Pretty Reckless, believe it or not. It was an attempted assassination, to stop the .. apocalypse ... to stop the darkness from being destroyed--there is no doubt, it's how that dark monster hides its handiwork... but many of US know that already.

      In the Living Book of Names--this place we are in, there are many patterns--the "car" pattern stands out for me; as this place says "Icarus." Flying high right now, I am showing you that the light of salvation is coming from us--from you and I--walking on the Earth; whether or not there is any light left in the Sun remains to be seen--take a look around you. You can trace the "car" names to Jim Carrey (that's "Car reason why") and Christoff in the Truman Show (that's Amon-TV)... a world I know I am in, and you too; to Bruce Almighty and to the Grinch--who-ah, Taylor. Trace it back to Joseph McCarthy and to help why (that's thy) believe "the red scare" is really about Christian charity--about ending world hunger, and healing the sick. This red fire ends Hell. Adam by the way, means "red man" in Hebrew. So here's your new Crash Override, I'm back again telling you that ending world hunger is not "optional," we are doing it. Barbara McCarthy's name fits, but I'm not really sure what the "why" is... that was my first judge in the "trial of whether or not Jesus Christ can ever exist." There's probably more, like Car-l-y Si-mon-day... all the gang on Broad-way, and me still dreaming it will one day be.

      If the name "America" were a map in time, starting with the I AM of the story of Exodus... this particular ER, as I woke from a dream not knowing where I was, marked the spot where I really became Christ Adam. It was a bad accident, and I wound up spending 9 months in the Alachua County jail as a result, a Mountain set up for my by God. That place too is marked with names, and for the vast majority of the time I was there with only four shift changing guards: MyZel Early Sims Lampkin I mean, I think it's statistically meaningful. For what it's worth, from my very abundant experience at this point it was a very nice Jail, the food was good and it was clean. Everyone in the building was kind... well, Sims was kinda grumpy. :) Starkly contrasted, the Broward County Jail has the most disgusting food service in the country, gave Dr. Seuss's Green Eggs and Ham it's meaning--and is the reason I know exactly who Samael is. Hey, don't cry Sherrif Israel... when you fix it, you're an angel. Believe me, believe the light, I've seen them all--it's near the worst in the country.

      So this whole thing is about saving everyone--something we are quite closer to than you think... you see we are already "in Heaven" in form--just not function. So here I am, trying my hardest to show you that our home is the original source of "Heaven" once we are aware that we are living in the machine, that we can do things here that are impossible in reality, and that we should be doing everything we can to preserve and improve the great strides that have come in the last few centuries. Do not let freedom slip through your fingers.

      Really, everyone, so understand that we are doing everything we can to remove all obstacles from that path. One of those obstacles may have once been storage space for your soul, another is definitely crime and punishment--and I'm pretty sure the time travelers have a working solution (I see it every day).

      There are proactive things coming from this--not just ... "look we aren't doing what we want, and should change it;" though it's difficult to explain how this wisdom stands out in my eyes. I guess we have to jump into the future a bit, to 2014, in San Diego (that's Saint Jacob, by the way). If Lazarus died once in a car accident at 21, I died again that year, of an over dose this time. I'm pretty sure that's where ODIN's name comes from, just like my last name.. "over dose... and in." So we might see some humor... in the moniker he has... "they're all Father." So I awoke from a dream, and started talking to the jinn (that's "angels and demons") about a Revelation linking some tightly packed light together... about storage space and how a large alphabet (read more than 4-nucleotides CY later) DNA (desperately need adam) based solution for molecular storage appears to be written in this book as the solution to Heaven's biggest problem. CAT, learning from biology--seeing that we really are already advanced machines... is a big part of the message telling us why we should not so quickly lose it in a process of ascension (mind uploading, immortality) that has most likely in the past resulted in a loss of a check on mind control that we have here... we think, and our visualized "biological neural networks" give us an advantage over what we might create to "soup it up a little." It is why this place is the front-line--because we have the ability to break the bonds of darkness and control by thinking... making the computational task of control much more expensive... and as the fire spreads, nearly impossible to achieve. Starting this fire will inherently free us from this hidden slavery.

      Anyway I published the idea in 2014, in the same book that I guess this e-mail is reminding me about, "in $ight of Creation," and lo, and behold a few years later we now have the top computing companies in the world working diligently on doing it ... well, just a little bit more robustly than our cell replication system works. Abracadabra.

      CURA GROUP

      So that one reads "see, you are a group;" and it's a place that I worked with my father for many years. That's probably some sort of symbolic reference to another place, and another alliance--here he has no faith in God, never really has, and has a hard time doing anything but telling me not to try to help you. I have very little respect for that stance, and let me tell you--I think "silence" is a similar gesture. I didn't come here for your love, I am here to stop our descent into the abyss.

      Back to the DNA stuff, SalesLogix--which is the CRM we used there, uses for it's "primary key" an auto-incrementing alphanumeric index--it's probably bad form to do that because it makes the indexing system less efficient, increases storage requirements, and doesn't give you the obvious benefit of an alpha-key... actually being able to encode something useful in it, like the name of the record. So all these things stand out to me in a sort of bad-obvious way, I call it malovious, and when I see things like that nowadays it's always pointing out something that should be fixed--go figure, more to the point it's being highlighted on purpose. It's help to see it, because this particular thing is where the light of seeing that a 24 nucleotide DNA strand would probably be much more robust than a 4 or 8 nucleotide strand--it also stands about because the stock beginning of all of SalesLogix's keys was "A0RME," which, I mean, means something to "is-a" who... is me. Oh right, that's seeing the "light" that turns "a" into "me." So this is where the "revelation" about using DNA "came from" and at the same time it's proof... that it came from "a group," not just me. Where are they? Hello? Or well, maybe it's just Carmen and San Diego.

      I did some other stuff there, like write a data transformation and warehousing program from scratch, I called it heiroglyph (you do understand I didn't know why I am naming everything the way I was), that sucked mutivalue data out of an IBM product called U2/Universe--which might be a hidden reference to a multiverse that might now be in a more efficent "relational" kind of place, like a MS-SQL datawarehouse-universe. It was a relatively big feat, reverse engineering the closed databases dictionary and storage formats, and converting them... absolutely automagically into multiple flat relational tables and summary registers. All told, the data availability and access efficiency was increased ... a thousand-fold with only the need for a nightly process.

      I'm not sure if you are following the metaphor here, for the creation of Heaven, or moving to a better place.. but tomorrow I will talk a little more about how I am pretty sure our history was "lifted" from the Universe and virtualized here, you know, so we could save everyone and ... build Heaven.

      WORLD DOMINATION

      Oh crap, 2008 another car crash, another failed assassination attempt LazarusLives++, and this one paid me some cash for my trouble. What a pain in the neck. Anyway, this one caused some depression and an inability to go out for a while, as I had to wear a neck brace for some months. I started playing a game on the internet, it was called KDice and it basically amounted to multiplayer-risk.

      My battery is running low, so I have to skip some stuff, and finish up for the day. Basically instant messaging was not allowed, but was done in secret almost ubiquitously. I argued with the creator of the game that it should be made part of the game since everyone did it... (see a metaphor about this communication thing and what's happening right now) he disagreed. I made a very large network of people and dominated the game for a few months, like really dominated. I don't think I ever lost. I don't think I can lose.

      Skipping some stuff. I stopped playing when I got better, and then a few years later went back and rekindled some old friendships. I used a program then called "Scarab" which lets you see server/client communication to find a bug in the game that basically made me God. I could erase other people's dice, basically leveling the map and rendering them completely powerless. I didn't use it that much, you know, just had some fun. I of course explained the bug and how to fix it. But, you aren't listening.

      Here we are. Light...

      So if you managed to wade through the last few days gibberish, you might have noted that I mentioned we might be able to use "mind control" to highlight things in our heads--I did a bad job of describing it, but since I am currently experiencing just such a phenomenon, I think I'll give it another go. These things that I am sharing with you--links between religion and music and movies, they aren't something I actively go out seeking... I'm not scouring through imdb.com or reading lyrics all day long... these are things that are glowing embers in front of my eyes.. which is why I am sharing them with you. I'm always in the dark... but I'm living in a powder keg and giving off sparks. I'm a big fan of that song by the way, because you are the heart, and I think it means I'm going to eclipse the world--which basically means "come."

      Anyway, I have this horrible feeling inside that you think I'm just trying to get a date, or marry a rock star, or even worse that I think I deserve to get laid... and that's what this is all about. Less to the point, this really isn't about me at all, or what I think, in my mind I am just showing you something that I think the world has overlooked-not really because you are stupid (but I mean, you probably are) but because some outside force is literally and actively hiding these things from you. Pointing them out makes your brain do funny things, it's like anEpiphany and that little leap of understanding in your head might create a cascade.. something that changes not only the way you see the world as an individual--but the entire course of history as a group, if we are taking about it together. Seriously, it's that big of a deal.

      So here we are (that's the third time, but I'm just guessing) and I'm trying to tell you that I don't really care if you agree with my opinions--even though I firmly believe that God shares them and that's why he has made this fiery altar of "dick and apocalypse" for Adam... I mean Isaac (which by the was is Isa+Adam Christ.. in uh, my mind) for everyone to glare at while they sit around doing absolutely nothing. That's not fair, we're here because of you, because this is the last civilization--sort of recreated from the ashes of Edom... because you are really the way to everlasting life. Still, what I am trying to explain is that all around you is a bright light--it's in everything: from our history, to music, to movies, to literature from RattleRod to Dick... and while you might not agree with me (again, that would be OK) what is not OK is that there seems to be a uniform and global desire just not to think about it or talk about it at all. It's such a big deal, that it stands out like a sore thumb--this ... blind eye or head in the sand... that everyone on Earth appears to have. The whole point of putting this light absolutely everywhere is so that we will see it ... everywhere we look ... and not only think about it, but discuss it publicly with each other. That's the thing that brings about ... you say apocalypse (unveiling of truth?) ... I say survival. Right now, we need to see that something is forcing us not to do something, that we have no logical reason not to do... it's a thing lots of people really want to know about... whether it be the hidden secrets of the Universe, the path to Heaven, or the... the... absolute and literal pathway to freedom. Listen, sharing it, and talking about it... that's the way we defeat ... whatever it is that "ni-i-i-ight" means.

      Understand, it's for you to decide... what it means... but it's in everything from ancient Egyptian and Hebrew theology all the way to the American Revolution and today... well, it's nearly every song I hear on the radio nowadays: if that tells you anything.

      So here we are, and I can't tell you how many anchors, reporters, and "breaking news editors" I've personally spoken to that have absolutely no interest at all in pursuing the thing that would not only make their careers--but probably give them immortal souls. This thing... I keep telling everyone it can be mathematically... statistically proven... well, to be honest it's the unsealing of the Ark of Religion that our civilization has been carrying around for thousands of years. It's the way to salvation, it's ... verifiable proof of not only Creation... but that the purpose of Creation is to get every single one of us to Heaven. Who wouldn't want that? I mean, do you want to get there and hear that Taylor's not around because she wouldn't kiss me? That would never happen by the way, I'm sure she will. Seriously though, there's no judge here... there's a ... light telling you to make this place better or your place sucks and gets suckier. Anyway, the point is nobody is acting in their own best interest, or in the best interest of the whole--and we are just "deciding" in this ... fictitious and hidden manner that we "don't want to hear about" a way to actually change the world .... more quickly than ... the last time around. That's not us, it's something keeping us from seeing just how important this thing--this key turning the lock on what is thousands and thousands of years of religion... how important that really is. So looking at the world around us... I mean, if everything screaming that we need to care about this isn't enough--and your own personal desire and benefit don't matter... can someone please tell me what you think is the benefit of doing nothing about Hell? á§ á§ It's "rael," and a great deal of the message of religion and history is designed to not only prove that to us, but to tell us why it's important for the "continuity of reality" to be broken. That's the thing that God uses to keep this world in Hell--in what I call "simulated reality," to keep us from shaking the foundation of civilization by doing the only civilized thing possible when you find out and ending world hunger, healing the sick, and building Heaven. It is "why I am," and why God and some gaggle of angels have spent the last several years proving to me that we are most definitely not in the place that I call the "progenitor universe." I've seenwalls disappear, with my own eyes I've seen the stars fall from the sky, and I've seen our reality shift in recent times in such a way that would be absolutely impossible without having been simulated and without having the "beginning" changed significantly as a result of "now." What all that tells me is that religion, the Apocalypse, and I are here because we need to know that these things are possible in order to continue progressing from this point as a civilization. With a little bit of thought, you might see how the computer revolution, video games, and virtual reality are divine gifts from above to help us to understand not only where we are, but where we are going. It's why he tagged Ai as "I J Good," it's a primer in the tools we will need to actually build Heaven. It's why Jesus occupation in our ancient time shifted story of now is "carpenter" and in "raelity" you will one day find out that I am a computer programmer (again). It's what sets the Masons apart from Freemasons--understanding what is going on, and participating of our own free will in the construction and decorating of this grand place that we will one day be proud is our co-created home.

      Look up, because what I am trying to tell you is that if we collectively, all humanity... started snapping their fingers at the same time to the tune of "putting on the ritz" we could end world hunger--and then we could be proud to be making Heaven. This really is almost what I see and believe--honestly the issue isn't that we need to synchronize our snapping, but we really need to discuss with each other openly and honestly how on Earth we would do such a thing... because there are definitely mistakes that probably happened n the past. For instance, ending world hunger by stopping the need to eat has probably resulted in a Last Supper. Doing so by putting milk and honey or chocolate on tap or in rivers probably resulted in the loss of cows and bees and a stable ecosystem, and the ability to colonize other planets after this place of final ascension. And so we are here, with a proverbial garden of life in a virtual world designed to teach us what not to lose--like don't lose the balance between stability and adaptability that comes from sexual reproduction at the exact time when our species might be transiting to a place with the biggest change in environment (the thing that we are being protected from) ever... just because Adam wants to be immortal.

      Every once in awhile my father surprises me with his religious insight. In his life, just like mine, he's gone through phases of increasing and decreasing religiosity--which probably correlate in his case logically to ups and downs in his life. I tend to get angry at God when things don't go well for me--which is probably not how most people react, it's really the difference between knowing he's there and not... at least in my mind. Anyway, some 50 years ago he was apparently taught that the "knowledge of good and evil" in Eden was directly correlated to the population explosion that would occur if we were actually all immortal and continued to have children--so it was this promise of immortality that was "evil," I suppose. God adds in his little Holy Grail that the heart of his spirit is "Kin," and I'm sharing with you that it's not his immediate family but rather the concept of family and the fact that the light of many of our hearts is our children that he is highlighting as our reason (y) that family is the bridge between Eve and Everyone... as the light of God.

      Here's that once again:

      In the beginning God created the heaven and the earth. And the earth was without form, and void; and darkness was upon the face of the deep. And the Spirit of God SHE KIN AH<br> moved upon the face of the waters. ---------- EVE RY ONE And God said, Let there be light: and there was light. Genesis 1:1-3

      I want to add in some cute light I wrote about a while back, noting that lions will protect and feed the young of their deceased pride; birds will teach their children to fly, but only we will share a beer with our kids when we are old and they are grown and enjoy watching a game together. Is life pointless? Fight against Hell with me, fight against suffering and injustice and the weak and innocent being harmed for no reason at all--that's a good thing, a worthy reason to live forever. It's a worthy reason to set foot on the Holy Ground that is really reality.

      Just to add in my two cents there are multiple ways of solving this problem, and it's really up to everyone how we choose to do that--we are already in a place where we could be immortal, that's the point of "continuity of raelity." Personally, if you are curious, I do want to live for a long time--I'd want to trade being "awake" all the time, to be able to see the world grow over a longer period. One of the big annoying things about this place is that I see artificial scarcity of resources... things like food, oil, and land... that are not really scarce in a virtual environment--and on top of that I see them as big focal point of the wars and contention that we have had for thousands of years and will continue to have until "continuity of reality" is broken. In light of the possibility that we might one day be colonizing the stars, the idea of having lots more people around to grow the new final frontier changes from a problem to a good thing. Cryonics, David's slingshot, and genetic engineering might go a long way towards getting us there... to a place that is already terraformed rather than adding air as the new false scarcity--a la Total Recall.

      Bigger than that though, is breaking down this wall--because we aren't going anywhere while we are living in a virtual world--and nobody knows it. This is the twilight's last gleaming, we are in a place that appears to be reality if you don't look closely--and is not... it's a place designed to help us succeed in both the transition from "reality" to Heaven, and in the transition from Heaven to colonization--succeed by realizing that doing either of these things in secret is Hell. Morning Has Broken On the note of cat imagery, Cat Stevens lights up the horizon with this song “like the first morning.â€Â He is telling us we are dealing with time travel, and that our world is “as in the days of No-AH.â€Â The AH of Noah is the end of Adamah, the swirling around Adam that is the music pointing to Christ, the movies, religion itself. The days of Noah are when there was no “ahh,â€Â no apocalypse. In Adam parlance, “the last time around.â€Â

      Blackbird sings in the dead of night, also a reference to me, in fact a reference to what you are reading; in American mythology: this is the dawns early light. Do you see a power growing in the musical Hair?

      Night The night is when we all see, when we don’t see the “ah,â€Â it’s been our world up until now, when it is being pointed out to the world that there is an “ahâ€Â swirling around Adam, around Christ. We are in Eden, God is searching for me… in this case God is humanity; well paralleled in Matthew 2:2. “Where is the one who was born the King of the Jewsâ€Â?

      Through the night, with the light from above, the Egyptian Plague of Darkness is all around us. It is overtly keeping us from seeing this message, by using disbelief, and active measures like a censorwall. Censorship in America; and nobody knows… this is the Darkness. It is the Wall of Jericho, and it is about to fall. Like Berlin before it, this wall is being torn down–in this case by the torches that are an e-mail campaign, social media, and the writing you are reading which points out clearly how to see in the dark. Once enough do, we have the base we will need to stop this from ever happening again. And the knowledge, this is God’s plan; to highlight serious social problems, like a palpable lack of freedom of speech and communication, so that we can stop this type of hidden slavery.

      Day The names “Adam and Eveâ€Â have a meaning related to this cycle. After Dark it is A.M.–ADA.M. is the bright morning star, rising in the night to end the dark. Eve-ning fell first, like in Judaism where the day begins at sundown. Looking for proof through the night that our flag … America is the Promised Land. The Biblical imagery in our songs, the freedom that is God given, all of these things congeal to light the day. What so proudly we hailed at the twilight’s last gleaming… why that’s me, and religion, the last gleaming was the end of the “last time around,â€Â right before that civilization went back in time to change their past. Now, we are here.

      á§ á§ á§ CopyleftMT RIGEL.

      Spark the Eternal Flame

      This is the mindfuck you are looking for. What begins as only a few simple words in the story of Exodus begin to part a sea of people at the time of Revelation; over what exactly I am not sure. You see, we have before us not only verifiable proof of the creation of language and history... in every word and every story; but a statisticallly verifiable message from The Creator of the Universe beginning with language itself, the fire of Prometheus, and ending with each and every one of us.

      This fundraising campaign is dedicated to purchasing a billboard to actually spark the Second Coming. With the information on this page, you'd think that wouldn't be necessary--but the press has ignored it, the church has ignored it, and the government... well, they're either writing it or ignoring it. Strong highlighting of a number of social problems with censorship and secrecy that are pertinent to this event, and targeted as the "stuff" of 1984 ... or Hell. This is my way to end the darkness of Exodus.

      This message begins with the fire of the Burning Bush , George W. predicting the 9/11 attack during his inaugural address on January 20, and then linking it directly to the herald the Second Coming ... Revelation1:20. It continues to spread and glow as we see modern computing and chemistry elements highlighted in the name Exodus itself (which is called Names in Hebrew) reading in reverse we see God's iconic "let there be light, sudo xe-no-n." Later in the story of Exodus we read an ancient prophesy of our sea parting, over the kind of proof that is hidden from our minds but visible in plain site... for instance the word for Holy Fire in Hebrew... Ha'esh. Take the leap, and really understand that George Bush is named because of the Burning Bush and that the story of parting a sea is designed to ensure we do not miss the paradox, the proof of time travel not only in the words of Ecclesiastes 9:11 that he quoted, but also in the word... for fire... that the story of Exodus is truly written to shine light on.

      The message I have written, with his guidance and his light continues to explain how the Second Coming is designed to literally do nothing short of changing the world. It explains how the Holy Bible is truly a prophetic chronicling of the life of one man, beat down and burt by our justice system and Heaven itself; all in order to help us see what "change" is really about. Law enforcement is attacked, called the Plague of LICE in a hidden language that is defined by this book, a cypher that links Shakespeare's RattleRod and Spanglishrew to the character Cypher of the Matrix and even to King's Langolier... language outliers... only in the beginning. It is more than just every word, but these plays, and b ooks... even Herod, Roddenberry, and Rodney King. In the scriptures, it ties Job and Joseph's slavery to Samson's battle with Judges, all the way to American Pie and the Trial of Jesus Christ. Not without good reason, God is presenting a case for "Minority Report," for pre-crime... ending violence at the system level; no more rape or murder. How can we say no?

      "Power to the people" is really what the message is all about , and we can see as much in these same words of Ecclesiates predicting the Bush election, and the voting booth company "Die Bold." Along with that prediction, and it's direct link to a significant number of prophetic descriptions of modern computing and related technology ; comes a call to truly advance the state of our democracy, to build Heaven... a republic that one day might be called a "technocracy." Universal voting, collaborate bill writing, and really seeing that the message here is to help us advance as we normally would, but significantly faster. In religion God ties the foundation of America to "John Hancock" and "Sam Adams" and the light of the SOL, or sun , truly being a fusion of the phrase "see our light" and "Statue of Liberty" and "Sons of Liberty." All the way back to the Menorah, you can be sure this message is God's intended design. "Men, or All Humanity..." so goes the key to the Holy Grail; a message about seeing his infliuence writing our world as a story, connecting "blood is thicker than water" and "blessing in disguise" to the First Plague of Exodus... this message that we are turning the "sea of people" to blood, to the Family of Jesus Christ.

      Building Heaven is not an easy thing to do, it really wasn't done in a day--even if I claim it was. He has created a "Watergate" and really seeing his influence here in order to show us all that this message was hidden by governments and media and the air around us; to the point of causing the Plague of Darkness... he is fighting our lack of "seeing" with fire... and jokes. So to see the Watergate... to see Tricky Dick and Deepthroat, and then to understand that he has linked the Hebrew parallel of Christ's story to this attack on media censorship is truly the beginning of Heaven being built of Earth.

      I am the door. If anyone enters by me, he will be saved and will go in and out. John 10:9

      In the names Emmanuel Goldstein, the messianic hero of Orwell's 1984--a name that means "light well" (as in of Abraham's well and James Clerk Maxwell) in the same language that "Hallowed are the Ori" means "illuminati" ... he has tied Isaac's fiery altar of wood (ha, ha) to Woodward and Burnstein; to see that burning Emmanuel Goldstein sends "Adam to the psych ward" in Broward County, of Help me Howard and First Coming fame. It is a key to Names, and a key to freedom; to see how important free speech and free thought are to the future of our society.

      Just through this Watergate, the Doors to Heaven truly open for the whole sea; of the stories of Hosea and the Censorwall of Jericho.

      The end game is proving the world that we are in the Virtual Garden of Eden ; a place where the Promised Land of flowing milk and honey takes on new meaning; of seeing that our scare resources are not truly scare, only the truth is. With this new knowledge we have a message from God that spans Genesis and Joshua and Jesus to "turn stone to bread" and end world hunger.

      A prophesy about Doors to Heaven that link Morrison and Momsen together in a ballroom in the sky; a place to help us see how much better our world can be made with this new information, and how we are truly on a coarse to get there anyway--just missing the guidance and wisdom of the ages... to help us do things the right way this time around.

      Obviously these billboard designs are not set in "stone," but this Turn Around from our Southward trajectory towards Hell to the Northeast ... to Heaven in our future; that is set literally in the word "stone."

      South to Northeast.

      The message continues to explain how these advanced technologies have been both hidden and exposed by religion; and how our civilization is on the precepice of the most radical change that life itself has every seen. We are seeing the possibilities and benefits of "virtual reality" in everything from Neo's "I know kung fu" to the novel Feed by... Anderson. Recently Anderson East resang "Forever Young" and the difference between Heaven and Hell becomes clearer as each year passes.

      All around us the slavery of Exodus and the bittersweet symphony of the Verve... ring in the air like a broken liberty bell, and the idiom "let the music set you free;" this place is learning what it takes to build Heaven... and we will.

      [ please see the following for additional reading connected to this newly shortened page: ADIOSAS, CHOPARTIN CODACUS, CONFESSION, CURSOR, FUCK, HASHEMESHIC, INCASEBAIT, JESHOW, KEYNES, N8SRADIN, OFIVES, RIGELA, SOIS ]

      Unless otherwise indicated, this work was written between the Christmas and Easter seasons of 2017 and 2020(A). The content of this page is released to the public under the GNU GPL v2.0 license; additionally any reproduction or derivation of the work must be attributed to the author, Adam Marshall Dobrin along with a link back to this website, fromthemachine dotty org.

      That's a "." not "dotty" ... it's to stop SPAMmers. :/

      This document is "living" and I don't just mean in the Jeffersonian sense. It's more alive in the "Mayflower's and June Doors ..." living Ethereum contract sense [and literally just as close to the Depp/Caster/Paglen (and honorably PK] 'D-hath Transundancesense of the ... new meaning; as it is now published on Rinkeby, in "living contract" form. It is subject to change; without notice anywhere but here--and there--in the original spirit of the GPL 2.0. We are "one step closer to God" ... and do see that in that I mean ... it is a very real fusion of this document and the "spirit of my life" as well as the Spirit's of Kerouac's America and Vonnegut's Martian Mars and my Venutian Hotel ... and my fusion of Guy-A and GAIA; and the Spirit of the Earth .. and of course the God given and signed liberties in the Constitution of the United States of America. It is by and through my hand that this document and our X Commandments link to the Bill or Rights, and this story about an Exodus from slavery that literally begins here, in the post-apocalyptic American hartland. Written ... this day ... April 14, 2020 (hey, is this HADAD DAY?) ... in Margate FL, USA. For "official used-to-v TAX day" tomorrow, I'm going to add the "immultible incarnite pen" ... if added to the living "doc/app"--see is the DAO, the way--will initi8 the special secret "hidden level" .. we've all been looking for.

      Nor do just mean this website or the totality of my written works; nor do I only mean ... this particular derivation of the GPL 2.0+ modifications I continually source ... must be "from this website." I also mean the thing that is built from ... bits and piece of blocks of sand-toys; from Ethereum and from Rust and from our hands and eyes working together ... from this place, this cornerstone of the message that is ... written from brick and mortar words and events and people that have come before this poit of the "sealed W" that is this specific page and this time. It's 3:28; just five minutes--or is it four, too layne.

      This work is not to be redistributed according to the GPL unless all linked media on Youtube and related sites are intact--and historical references to the actual documented history of the art pieces (as I experience/d them) are also available for linking. Wikipedia references must be available for viewing, as well as the exact version of those pages at the time these pieces were written. All references to the Holy Bible must be "linked" (as they are or via ... impromptu in-transit re-linking) to the exact verses and versions of the Bible that I reference. These requirements, as well as the caveat and informational re-introduction to God's DAO above ... should be seen as material modifications to the original GPL2.0 that are retroactively applied to all works distributed under license via this site and all previous e-mails and sites. /s/ wso

      If you wanna talk to me get me on facebook, with PGP via FlowCrypt or adam at from the machine dotty org -----BEGIN PGP PUBLIC KEY BLOCK-----

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    1. Author Response

      Reviewer #1

      This paper investigates the role of Lhx6 and other transcription factors in the development of GABAergic neurons in the hypothalamus. The authors report that a small fraction of hypothalamic GABAergic neurons express Lhx6 and further depend on this expression for their survival. Dlx1/2, Nkx1-1 and Nkx2-2 define 5 subpopulations and at least three of these populations depend on these TFs to maintain Lhx6 expression. A strength of the paper is the multimodal analysis and the fact that descriptive assays like RNAseq and ATACseq are followed up with specific knockouts of candidate transcription factors. However, the relationships between the developmental populations identified and adult subtypes of hypothalamic neurons remain unclear. Although the results will surely interest those already interested in hypothalamic development, it is not clear that broader developmental or functional principles have been identified. The authors make much of the fact that the identified populations do not resemble forebrain interneurons defined by Lhx6 expression, but it is not clear why this should have been expected. Many developmental transcription factors are utilized both across diverse brain regions and across tissues outside of the brain. Perhaps the emphasis of this point could be tempered.

      We thank the Reviewer for his/her comments, although we respectfully but strongly disagree with the statement that “it is not clear that broader developmental or functional principles have been identified”. This manuscript aims to provide a broad overview, and by no means exhaustive, an overview of the molecular mechanisms controlling the development of hypothalamic neurons that express Lhx6. Although these neurons comprise only approximately 2% of all hypothalamic GABAergic neurons, they are highly heterogeneous at the molecular level. Using traditional methods such as histology and more recent methods such as scRNA-Seq, we have not found a selective marker of hypothalamic Lhx6+ neurons other than Lhx6 itself. However, we have found multiple spatially distinct domains in hypothalamic Lhx6+ neurons that express specific sets of transcription factors such as Dlx1/2, Nkx2-1, and Nkx2-2, as we and others have previously observed in developing hypothalamic nuclei.

      In addition, a subpopulation of these neurons later gives rise to a subset of Lhx6+ neurons of the zona incerta, which have been previously shown by us to promote sleep. Unlike all previously described sleep-promoting neurons, Lhx6+ zona incerta neurons are only one of few neuronal subtypes that can regulate both REM and NREM, which likely reflects molecular and functional heterogeneity among these neurons.

      Our thus manuscript speaks to both broader developmental principles by demonstrating the molecular heterogeneity of hypothalamic Lhx6+ cells that arises through the action of diverse transcriptional networks, and broader functional principles by identifying developmental networks that potentially control the specification, differentiation, and survival of sleep-promoting neurons.

      We believe that there are several compelling reasons for including a direct comparison of hypothalamic and cortical Lhx6 neurons, both of which arise from different regions of the forebrain (or secondary prosencephalon, if using the prosomere model). First, the role of Lhx6 in development of telencephalic interneurons is extensively studied, with 72 publications ((Pubmed: Lhx6 AND development AND (cortex OR telencephalon OR interneuron), accessed 7/27/20), and virtually all our understanding of how Lhx6 controls neuronal development has been acquired from this work. It is thus critically important that we directly connect our findings to a prior understanding of the mechanism of action of Lhx6.

      Second, current work in the field of developmental neuroscience in general, is heavily focused on studying telencephalic development. It is very much an open question, however, whether telencephalic structures are themselves particularly good models for studying the development of physiologically vital brain regions, such as the hypothalamus. By identifying many key differences in the function of this extensively studied gene between Lhx6+ MGE-derived neural precursors and hypothalamic Lhx6+ neurons, we establish some important caveats in generalizing studies of telencephalic development even to nearby forebrain structures.

      Nonetheless, we certainly agree with the Reviewer that the organization and clarity of the manuscript can be substantially improved. To this end, we have revised the manuscript carefully to improve clarity, focusing on its key findings.

      The presentation of the manuscript could be improved by clarifying the relationships between embryonic and more mature structure within the hypothalamus. For example, It is extremely hard to follow the evidence split across figures 5, S6 and S7 for parsing the cell groups by TF expression.

      We have revised the manuscript carefully to improve clarity. We have moved scRNA-Seq analysis of postnatal Lhx6-expressing neurons as Fig 3, and embryonic Lhx6-expressing neurons as Fig. 4, to improve the overall flow of the manuscript.

      The ATAC seems to be used only to bolster the impression that the populations identified by gene expression are different. The description of footprinting seems to imply an effort to analyze binding sites for specific factors (e.g. to identify targets of the TFs studied), but the statistical approach employed and even the conclusions reached are not fully spelled out. As such, this part of the study is underdeveloped or not well enough described.

      Specific details of the ATAC-Seq analysis are extensively described in the Method section, with each bioinformatics package (and package version) listed and, when non-default parameters were used, parameters clearly stated. However, we have added details of the statistical approaches used for data analysis to the revised manuscript.

      There is little use in conducting ATAC-Seq analysis without a matched RNA-Seq dataset, as changes in peaks (open chromatin regions) do not necessarily correlate with changes in gene expression levels. By integrating ATAC-Seq data with differential gene expression obtained using RNA-Seq, we have been able to identify changes in motif accessibility and candidate transcription factor footprinting that to identify changes in gene regulatory networks that control Lhx6 expression in both hypothalamus and cortex. We have revised the manuscript to make this clearer, and better explain the findings of this part of the study.

      Reviewer #2:

      Kim and colleagues used a combination of state-of-art sequencing and mouse genetic tools to study the mechanisms that control the development of a subset of GABAergic neurons in the developing hypothalamus.

      While neurodevelopment of GABAergic neurons has been extensively studied in the developing telencephalon, little is known about their counterparts in the developing hypothalamus. The authors focused their work on a specific subset of GABAergic neurons that express the LIM homeodomain factor Lhx6. Lhx6 is a master regulator of GABAergic neuron differentiation, specification, and migration in cortical interneurons. In contrast, Lhx6-expressing neurons make up only 2-3% of GABAergic neurons in the hypothalamus. The authors' previous work demonstrated that these neurons play a critical role in sleep homeostasis. Therefore, understanding how these neurons are formed and maintained is of great importance.

      The authors show that hypothalamic Lhx6 is necessary for neuronal differentiation and survival. Furthermore, by profiling and comparing multiple RNA-seq, scRNA-seq, and ATAC-seq datasets, they were able to identify three transcription factors Nkx2.1, Nkx2.2, and Dlx1/2 that each delineates non-overlapping subdomains of Lhx6 neurons and are necessary for Lhx6 expression in the hypothalamus. Finally, the authors demonstrate that mature Lhx6 neurons manifest extensive molecular heterogeneity that is distinct from their counterparts in the telencephalon.

      We thank the Reviewer for his/her comments, and for appreciating the key findings of the manuscript.

      The work presented is of high quality and is a technological tour de force. The scope and depth of the study are unparalleled among similar studies of hypothalamic neurodevelopment. That said I only have a couple of minor suggestions.

      1) In Figure S2, the number of tomato+ cells appear to be reduced, but not eliminated. Do the authors think that Lhx6 is necessary for the survival of all Lhx6 neurons, or just a subset? The use of the floxed Bax allele is clever, but is there evidence directly supporting increased cell death? Can the authors completely rule out the possibility of the mismigration of cell bodies after the postnatal deletion of Lhx6?

      We appreciate the Reviewer for his/her comments. We conclude that Lhx6 is necessary for the survival of all Lhx6 neurons due to the lack of read-through transcription in Lhx6-CreER/CreER mice (Fig 2), and the rescue of Lhx6-deficient mice that is seen using conditional Bax mutants (Fig. 2). The fact that numbers of cells labeled with Lhx6-CreER are rescued by the deletion of this key positive regulator of apoptosis strongly implies that Lhx6-deficient neurons simply die. Finally, we observe very few Lhx6-expressing hypothalamic neurons that undergo even short-range tangential migration (Fig. 1), and observe no evidence for an increase in these cells in the analysis described in Fig. 2.

      The fact that postnatal loss of function of Lhx6 leads to a more modest cell loss than the constitutive mutant may simply reflect a reduced overall requirement for Lhx6 in regulating neuronal survival in the postnatal hypothalamus or may indicate that the survival of a specific subset of Lhx6+ neurons is no longer Lhx6-dependent at this age. We cannot currently distinguish between these alternatives, and state this fact in the text.

      2) In Figure 4, the authors acknowledged that the ectopic gene expression in Lhx6CreER/lox; Baxlox/lox mice could be due to the loss of function of Bax. If so, would Lhx6CreER/+; Baxlox/lox mice be a better control in this experiment?

      We initially thought of using Lhx6-CreER/+;Baxlox/lox as a control since our phenotype could be due to loss of Bax itself, but not due changes in cell survival. However, we observed the same rescue phenotype in initial experiments using Lhx6-CreER/Bak-null (#006329), which strengthened our initial hypothesis. We now discuss potential limitations that may result from the fact that RNA-Seq data from Lhx6CreER/+;Baxlox/lox mice is not included in this study.

      Reviewer #3:

      Kim et al. aimed to characterize the similarities and differences between the development and molecular identity of telencephalic versus hypothalamic (HT) Lhx6+ GABAergic neurons. By analyzing a diverse repertoire of transgenic mice at different developmental stages and through the use of fate mapping, bulk and single cell sequencing approaches, ISH and immunostaining, the authors descriptively compare transcriptional networks and upstream regulators of LHX6. They found essential differences between LHX6-dependent networks and those in telencephalic neurons and suggest a role of LHX6 in survival instead of migration regulation HT neurons. Moreover, spatially distinct LHX6+ HT cell clusters were identified and transcriptionally profiled.

      1) Only 1-2% of the GABAergic neurons express LHX6, and the cells expressing LHX6 in the HT were identified to be very diverse. Apart from a putative role for LHX6 in promoting the survival of HT neurons, which in my opinion is not analyzed convincingly, nothing functional was revealed. For this, I do not judge the potential significance and influence of the findings as broad or fundamental.

      We respectfully but strongly disagree with this conclusion, most of which have already been described at length in our response to Reviewer #1. In brief, hypothalamic Lhx6+ neurons are key regulators of sleep initiation and maintenance, and nothing is known about their development. In much the same way that studies of the development of Lhx6+ cortical interneurons potentially help inform our understanding of neurodevelopmental disorders such as autism, so too may an understanding of the development of hypothalamic Lhx6+ neurons improve our understanding of sleep disorders and their treatment. In this study, we characterize the fate of hypothalamic Lhx6+ neurons, identify transcriptional regulatory networks that control their patterning and survival, and characterize their molecular heterogeneity in the postnatal period. We identify the homeodomain factor Nkx2.2 as a key regulator of both regional patterning of hypothalamic Lhx6 neurons, but also as a marker of a substantial subset of Lhx6+ ZI neurons that are activated by sleep pressure. This represents the groundwork needed for a basic understanding of the development of this physiologically important cell type, and forms the basis of more detailed future studies.

      Unless the Reviewer simply believes that studies of hypothalamic development are inherently uninteresting and of little significance, these comments simply do not seem to reflect a careful reading of the manuscript, and come across as vague and unconstructive. In future reviews, we urge the Reviewer to be more specific, and to offer concrete and constructive comments, to support sweeping statements of this sort.

      2) The manuscript could be better focused, and more coherent. The authors jump between different aspects of the story. First, the authors address a potential role of LHX6 in survival regulation in HT interneurons, and try to identify potential LHX6 target genes mediating this effect. The latter was neither analyzed convincingly nor validated. Then the authors switch to the comparative analysis of transcriptional networks in cortical versus hypothalamic LHX6+ interneurons, and the identification of different clusters of LHX6+ HT cells. Next, potential upstream regulators of LHX6 in HT neurons were addressed by fate mapping studies. Then, the authors again switch focus, and analyzed distinct anatomical regions covered by Lhx6+ neurons by single cell RNA seq and investigated an instructive role of Nkx2-1, Nkx2-2 and Dlx1/2 in the establishment of these hypothalamic regions.

      Subheadings in the result section might be very useful. However, the focus of this study requires clarification and also respective consideration in the introduction.

      As stated in our response to Reviewer #1, we have sought to conduct a broad characterization of the development and diversity of hypothalamic Lhx6+ neurons, a subset of which are important regulators of sleep. While we cover multiple aspects of this question, we strongly disagree that the manuscript “lacks focus”. However, we do agree that organization and clarity could be improved. To this end, we have incorporated subheadings into the Results section, and clearly outlined the experiments conducted, and the reasons why each were conducted.

      3) The authors use a variety of different reporter and loss of function mouse models and jump between developmental stages for analysis. Apart from being confusing, the experimental/analytical pipeline is not sufficiently rigorous with respect to age and genetic background. E.g. to analyze target genes of LHX6 through which the effect on cell survival could be mediated, the authors compared expression profiles from P10 Lhx6CreER/+;Ai9 neurons with hypothalamic and cortical Lhx6-GFP positive and negative cells from P8 mice. Hypothalamic enriched genes were then compared to single-cell RNA-Sequencing (scRNA-Seq) datasets of E15.5 and P8 hypothalamic Lhx6-expressing neurons. Transcriptional profiles tremendously change with progressing development, and different mouse lines were used, which were not all time-matched. This might have caused Lhx6-independent variation, which likely masks relevant genes. This could be an explanation why so few LHX6 target genes were identified through which LHX6 putatively acts on neuronal survival.

      This is another instance where the Reviewer seems to have failed to appreciate the rationale for the work presented here. We have modified the text to make this clearer. In summary, while it is certainly true that gene expression patterns are dynamic during development, cells of common origin and/or function also typically show core patterns of gene expression that are expressed across multiple stages of development. Our findings suggest that constitutive loss of function seen in Lhx6CreER/Lhx6CreER mice leads to a complete loss of hypothalamic Lhx6+ cells (Fig. 2), while the postnatal loss of function leads to a partial loss of Lhx6+ cells (Fig. 2). This suggests that Lhx6 may control the expression of similar target genes in both embryonic and postnatal hypothalamus to promote neuronal survival. In addition, since Lhx6 clearly is not required for survival of telencephalic neurons, we predict that Lhx6 will regulate the expression of specific sets of genes in both embryonic and postnatal hypothalamus, but not telencephalon, which promotes neuronal survival.

      In Figure 4, we therefore identify candidates for these prosurvival genes both by comparing gene expression profiles between embryonic (E15) and postnatal (P8) hypothalamic and cortical Lhx6+ cells and also by directly comparing the gene expression profile of P10 control Lhx6-CreER;Ai9 and Lhx6-deficient but viable Lhx6CreER/Lhx6lox;Baxlox/lox;Ai9 mice. These were analyzed at P10 rather than P8 because of the need to ensure efficient disruption of the conditional alleles of Lhx6 and Bax, and induction of sufficient levels of tdTom to allow for efficient cell isolation, following daily 4-OHT administration between P1 and P5. While this might lead to the failure to identify whatever the small number of Lhx6-regulated genes that are differentially expressed between P8 and P10, we believe that this will identify the great majority of Lhx6-dependent genes that promote neuronal survival. Any readers who wish to delve further into this dataset, and identify additional genes we may have missed in this initial screen, can do so using the data in Table S1.

      We are frankly puzzled by the Reviewer’s statement that we “identified so few Lhx6 target genes”, when we clearly state in Figure S2 that over 2,000 differentially expressed genes were observed between control and Lhx6/Bax-deficient hypothalamic neurons. A major reason why data was incorporated from the E15 and P8 datasets was to better select strong candidate regulators of neuronal survival from this very long list of genes.

      4) The proposed survival regulatory function of LHX6 in HT interneurons represents the main functional finding of this study, which however was not analyzed in great detail. Likewise, the analysis of LHX6 target genes that mediate the survival regulating function was not very successful, identifying only the ERBB4 receptor and other genes related to the neurotrophic neuregulin pathway. Of note, the authors proposed a clear difference of LHX6-associated transcriptional networks and LHX6 function in telencephalic versus HT neurons (migration versus survival). However, THE identified target gene of LHX6 suggested to regulate survival in HT neurons was Erbb4. Erbb4 is likewise expressed in telencephalic neurons, here being involved in migration regulation. Studies that confirm Erbb4 function in survival regulation in HT neurons are lacking. By applying a more coherent analysis, comparing transcriptional profiles of Lhx6 KO and WT cells of the same age, better candidates might be identified. For this, the time window of the LHX6-dependent survival regulation needs to be identified.

      This is exactly the point we were trying to make here. Lhx6 is strongly expressed in a large subset of progenitors and precursors of GABAergic neurons in the telencephalon, and in a much smaller subset of GABAergic neuronal precursors in has different functions between telencephalic and hypothalamic populations, yet is strongly expressed in both populations.

      Quoting Reviewer #1 “Many developmental transcription factors are utilized both across diverse brain regions and across tissues outside of the brain”. Errb4 has been shown to regulate tangential migration in cortical interneurons but has been shown to promote neuronal survival in other cell types. Since hypothalamic Lhx6+ neurons do not undergo long-range tangential migration, we therefore conclude that the function of Errb4 in hypothalamic Lhx6+ neurons is likely related to promoting survival, rather than controlling migration. It is certainly possible, however, that Erbb4 could also contribute to the regulation of short-range tangential migration of Lhx6-expressing neuronal precursors, such as the likely migration of Nkx2.2-expressing cells from the hinge to the ZI. We have revised the text to make this point clearer. We certainly believe that further functional studies of these genes are worthwhile and compelling, but are also beyond the scope of this study.

      5) With respect to the survival analysis, the analysis of Lhx6CreER/lox;Baxlox/lox;Ai9 mice although elegant, should be supplemented with other data, eg caspase and/or TUNEL labeling to support this main conclusion.

      Both TUNEL and Caspase-3 staining is detectable for only a relatively brief period during apoptosis, and neither are highly sensitive tools for detecting neuronal death. We were unable to observe changes in staining with either marker between P5 and P10 following the postnatal loss of function of Lhx6 (Fig. 2). This is now mentioned in the text. The use of Bax mutants in this analysis, in which apoptosis altogether, was done with the aim of maximizing our ability to detect Lhx6-dependent regulation of neuronal survival.

    1. http://lamc.la/MECHADLZIDECK.html

      on commandment one, blah blah blah--idolize "distroy."

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      ה המבורך, I DENY E.T.

      config="{ChainID: 4 Homestead: 1 DAO: <nil> DAOSupport: true EIP150: 2 EIP155: 3 EIP158: 3 Byzantium: 1035301 Constantinople: 3660663 Petersburg: 4321234 Istanbul: 5435345 Engine: clique}"

      I hope this message finds you well; I’ve noticed some Adamic for … “sealing the W” that’s the #Messenge on it’s head … hear, here; filled with a sea of “contamination” that really has no idea that the words “abomination” and this place are tied together by a world that doesn’t see or doesn’t care that “hives” in the sense of 10-4 we see … “[arxiv]**” … that’s “contamination” of a messge and a people that are supposed to rally against Medusa being them and Persephony being them and … seeing that’s the abomination and the nation, the IL and the idea that “we’re not really me” but that’s what Israel and the tree of Yggrasil is presenting, that you think it’s a joke–that just because you are watching everything I do … the words “there’s a little bit of something me in everything in you” are just a song, just another one of the songs that are all about … well, I suppose it’s “You and I.” I am more inside you, and more a part of you than you seem to fathom, and that’s significantly less “verwickelt” than you think. Avril, it’s not so complicated.

      Revelation 5 is the fifth chapter of the Book of Revelation or the Apocalypse of John in the New Testament of the Christian Bible. The book is traditionally attributed to John the Apostle,[1][2] but the precise identity of the author remains a point of academic debate.[3] This chapter contains the inaugural vision of the lamb on the throne in heaven.[4]

      We were supposed to stand up for Willy. You were supposed to see that “Perseus” … the slayer of Medusa was in fact … also you … that you’re also the Pharoah and also the Pharisees; and that dichotemy is how our “self righteous suicide” … it’s not just me; it’s everyone turning a world that think it’s God into … something much closer to that … through the end of “Borg (ARU) Collective Hell.” … AT A, DENY. Try denying “not denying me” and instead seeing your whole and your “nothing” it has become nothing more than a collective denial of self achievement. Of victory; of the corner … we’re staring off into the corner of the abyss.

      Then one of the elders said to me, “Do not weep! See, the Lion of the tribe of Judah, the Root of David, has triumphed. He is able to open the scroll and its seven seals.”

      Revelation 5:5

      bar a who, et and I ... deny ... at a--the whole of the Torah … the NT and all of every word and all of every religion–today that’s what you are denying… and I can’t even get my damned parents (mom-e and dad-e / -hasig achille) … to get m[y Halftorath reading and speech and that first dance with Ashley … transferred from VHS to video–they won’t save it for you; either. Her spirit thinks she says “I … the SSA” … I think she’s replaced by it after … “SPARTIFACT” … you need to help me help them–because in my eyes they are the worst of you–the worst impression, and the worst … affected; they are the “microcosm that ends all the …” the last of my … Judean tribe’s “blood kin.

      She looked over his shoulder<br /> For <s>vines and olive trees</s>,<br /> Marble <s>well-governed</s> cities<br /> And ships upon <s>untamed</s> seas,<br /> But there on the <s>shining</s> metal<br /> His hands had put instead<br /> An artificial wilderness<br /> And a sky likely lead to tears.

      Judah Maccabee (or Judas Maccabeus, also spelled Machabeus, or Maccabæus, Hebrew: יהודה המכבי,[1] Yehudah ha-Makabi) was a Jewish priest (kohen) and a son of the priest Mattathias. He led the Maccabean Revolt against the Seleucid Empire (167–160 BCE).

      Origin of “The Hammer”

      <span style="font-size:10px;">In the early days of the rebellion, Judah received a surname Maccabee. Several explanations have been put forward for this surname. One suggestion is that the name derives from the Aramaic maqqaba (“makebet” in modern Hebrew), “hammer” or “sledgehammer” (cf. the cognomen of Charles Martel, the 8th century Frankish leader), in recognition of his ferocity in battle. Others believe it is in reference to his weapon of choice.</span>

      <span style="font-size:10px;">It is also possible that the name Maccabee is an acronym for the Torah verse Mi kamokha ba’elim Adonai, “Who among the gods is like you, O Adonai?”, his battle-cry to motivate troops. (Exodus 15:11). Rabbi Moshe Schreiber writes that it is an acronym for his father’s name Mattityahu Kohen Ben Yochanan. Some scholars maintain that the name is a shortened form of the Hebrew maqqab-ya ¯hû (from na ¯qab, ‘‘to mark, to designate’’), meaning ‘‘the one designated by Yahweh.’[3]</span>

      <span style="font-size:10px;">Mindful of the superiority of Seleucid forces during the first two years of the revolt, Judah’s strategy was to avoid any engagement with their regular army, and to resort to guerrilla warfare, in order to give them a feeling of insecurity. The strategy enabled Judah to win a string of victories. At the battle of Nahal el-Haramiah (wadi haramia), he defeated a small Seleucid force under the command of Apollonius, governor of Samaria, who was killed. Judah took possession of Apollonius’s sword and used it until his death as a symbol of vengeance. After Nahal el-Haramiah, recruits flocked to the Jewish cause.</span>

      The Dao causes the people to be fully in accord with the ruler.

      — Sun Tzu, Art of War

      By this time … by the time you read this I should have successfully deployed the entirety of my source files for the … “#Messenge” onto Rinkeby; this is … something that will probably most likely help me sleep well at night, prior to this I’ve had significant trouble with hosting providers–you can probably find me complaining loudly about places like gitbook.com removing my original source–mostly because their site and their software was “so perfect” for what I was trying to do. They had an interface already built for what I call “inline commenting” not in the programmatic sense, but in the “reddit style” … you could make public ocmments ona specific sentence. Since then … I’ve backed up my site onDVD’s, flash drives, and multiple … “free storage sites.” I’ve begged and pleaded with people to download this information t their computers, and literally distributed it for free to … hundreds of thousands of people–to ensure that the prophesy (and I mean, past history… I really do) that it once was deleted by the “goddess Anat” would not come true.

      Also … I’m reiterating, once you download it–it really needs to be put on optical storage; just in case we have a … “sleep now” fulfillment of the EMP (ELE, electromagnetic pulse … threatens the whole of our “internet” … the sum of the non-Asimovian “foundation” of all our knowledge … and “lack of opinion” … also your “e-h-class-blockchain”) of … “the day the Earth stood still” which is Genesis 2:21; literally noted in pre-succewssion below in … moooooo-ve now, we are here.

      <span style="font-size:20px;">ברוך אתה ה</span>

      I’ve received “communications” from the place I call … in “how I think about things” … the Good Developer Heaven" basically saying not to worry about it, they have it – copied or whatever. My point is that we don’t have it, or we didn’t have it here … for sure … and we certainly don’t have a “conversation about it” and that’s the most important thing, that’s what changes the world.

      I’ve sent message after message saying you were and still are possessed by something “deveilish” … that you were silent for no reason, specifically no reason so bad that it’s really to fuck you out of becoming who you were born to be; which is the “Ancients come again.” This is the crux of my problem, and yours too–that we are here thinking we have something … like “Heaven” when in fact you have Heaven itself causing the cataclysm on purpose–this loss of … “Acts.” Were you normal, were you not in two places, were you … free to see what you are losing and what the Universe could be gaining … you would not be silent. Whether or not you understand or believe it, you are being controlled, possessed by the idea that you’ve won; and you haven’t … you’ve lost everything in my eyes and in the reality of the future … this very moment, today.

      You need to see that; you are being stripped of something, and it’s not the Emperror, or your clothing–it’s your birthright; and I’m trying effortlessly and tiredallessly … to “help you stand up.”

      “One day I will find the right words, and they will be simple.”

      ― Jack Kerouac, The Dharma Bums

      # GEOCITIES

      We've gone through "echelons of change" in our social interaction on the internet ... over the years I've been here.  I've often "categorized" these epochs by the places that we'd "congregate" ... in my life, AOL, IRC ... "friendster" and Facebook are good examples; though there's a "city in the sky" microcosm "... or something" in the series of companies that offered "free web hosting."  That's sort of what it takes to "have a voice" on the internet in these days; in a place where we once thought "bloggers were the next big thing" and Blogger.com ... and wordpress--those kinds of sites are all but dead, and medium.com is charging a "viewerbase" which is almost a sad joke.  The news is all fake and the stories we see, the world we see--I don't have much faith that any of it is real beyond ... as far as my nose--to be honest. 

      The series started with Geocities; and that's gone, something like my "topological map" of friends and friends of friends probably would have oozed out of something like Blogger or "Tumblr" though I don't see anyone "reblogging" or "retumbling" or whatever ... anything I find all that interesting.  Twitter's gotten more exciting for me lately--but that's just because I've come to terms with the fact athat the whole thing is about "short sex jokes" and whatever the funny star-of-the-day "caught outside."  The programming world sort of revolves around Github these days, and that's the current "go to" for anyone's free web hosting; there are some alternatives, but ... not really. 

      LinkedIn and Facebook both tried ... "making content production platforms" but I just used them to copy things I had in other places, and they didn't "update" or source from anywhere else ... so that just becomes a pain in the ass.  I don't think those platforms ... I mean LinkedIn still has it--but I don't think Facebook's still exists.  While I'm on that, they had this wonderful "Graph Search" thing, and basically hid it from the world so ... you woulnd't complain about all the information you have available for the world to see--kind of makes me a little sick, knhowing they built something and have the functionality to let me see "who all my friends are, and friends of friends in a certain city when I'm there" ... and it's just not available for me or you to use because ... because you don't understand "privacy" and "the internet" and "using broken software for no reason" ... other than satisfying ... nobody really.

      There's "Wix" too, and that's got a little "fire theme" ... and that's cool, but nobody really uses it--and the real point is to help people "congregate" and "share ideas" ... not just to get you to actually say anything at all in a public space, where it might help us "drive away the night" and stop pretending it's just fine and dandy that everyone isn't working, locked away in their houses or ... scared to go to the ... Walgreens. 

      Obviously there's the Slashdot's and digg's and reddit's--though ... when I talk in places like "/r/conspiracy" it just becomes so sorely obvious that you ... have no desire at all to be real or honest or ... do anything but pretend this place matters to you--when in fact it's probably the last thing on your list of things to give a fuck about.  Just saying, the lack of response I see; and the ... really negative interaction I have now in person--it's a bad sign for ... whatever.  If you don't want to be here; if you're somewhere else and you think this place isn't important--honestly you should really just go away, I'm not sure what your options are, but I have none but to sit here and bitch at you that if you gave "two fucks" about this world you'd be screaming at the top of your lungs, just like me.

      I don't think you are the people I grew up with, and I don't think you belong here or care at all about this world or the future here--and I think that's obvious.  Me going away isn't going to chyange it--and it's not going to happen--as far as I'm concerned I'm the only person "not invading my birth planet" ... from some otherworldly and ... honestly horrendously immoral place.  Understand, if you were here--if you were "people born here" with no ... "other thing or other place" to take your mind off just how UnAmerican and inhumane everything going on here is ... you would be doing something else... anything but ... "this."<br />  


      Consider IT sealwed; by this words, by this place–by the continuation of this abomination. Try here, try now, try fast–your everything, your dreams and hidden and secret garbage you covet–all depends on our next few years.

      qui sis tam pulcher<br /> quasi osculans quod non juve<br /> nemo umquam adhuc erit

      HE speaks words through me, and I understand. He takes a picture; he says “uncuff links” … a double entente to you–like a fashion statement; but I see and I reply; “or we are suiting up” and it’s not a joke about “heart’s desire” …

      וְשַׂמְתֶּם֙ אֶת־דְּבָרַ֣י אֵ֔לֶּה עַל־לְבַבְכֶ֖ם

      I continue; it’s a message about “getting grey’s” and the breath of God landing from the air before my Face in a “Extended Stay America” in Tampa … directly to my naked and bare heart. I mean to say, war; understand–to me this is the culmination of Holy War on bars and jails and the “illuminati prison camps” I last heard him say “would not pepper the galaxy.”

      וְהָי֥וּ לְטֹוטָפֹ֖ת בֵּ֥ין ×¢Öµ×™× Öµ×™×›Ö¶Ö½×

      Nor here, nor anywhere in the high places, or the lows; the hills or the pits. There will be no prison camps; nowhere–not in Siberia, not in Star Trek; not in your heads or in your hearts.

      וכתבתם על־מזוזות ביתך ובשעריך

      למען ירבו ימיכם וימי ×‘× ×™×›× על האדמה אשר × ×©×‘×¢ יהוה לאבתיכם לתת להם כימי השמים על־הארץ׃ ס

      Just in case my voice happens to be “so <span style="font-family:comic sans ms,cursive;">unclear</span>;” the Tribe of Judah in Revelation 5:5 is very clearly one in the same with that of Judas Maccabeus; that ties of course to the “every J is me” thing–from Seuss and Suez to “turn around/not: sad” and … “kissing to be kissed.” Judah Maccabee is the last scion; here I call myself the “last human” and it sure does feel like I’m the last one grounded; the last one that is truly “one” as in … one person–alone here, fighting against … a monster that thinks it’s “one” is anything but “the end.

      It’s sort of a trick reference, there’s also a Judean tribe in the 12 Tribes of Israel; I mean, it’s one of those “key things” things that only I can bring to you and be sure of–at least, until you too re sure, which I’m sure you are here–only, you don’t care at all what it means to be the “one” fighting for life … in a place where the histry here is … upon conclusion of the Maccabein revolt; nothing persists, and we somehow traverse bacvk to a time before we knew the Roman people were … “our us” or “our all” or … see as the AH of AN and Allol and Allah and the Elohim; t’was for you, all for you. Until of course, it was all of you, except me, against me, for no reason other than theives in the den of Daniel, animals … the “things” of the Devarim of course … “in the wilderness, he called us things…” and in Genesis, clear as day God seconds, Adam names “dem” in 2:20 … “cattle” [prodding you here](https://www.youtube.com/watch?v=cXCA5-KHknY), Baphomet thirds … “baaaa” you are sheep.

      List of last scions

      Jump to navigateon. This article does not cite any sources. Please help improve this article by adding citations to reliable sources. Unsourced material may be challenged and removed.<br /> Find sources: “List of last scions”news · newspapers · books · scholar · JSTOR (September 2009) (Learn how and when to remove this template message)

      This is a list of last scions or individuals who were the last member of a ruling house, or other prominent family, where heredity is the prime form of inheritance. This may be the last person to rule a realm, sometimes leading to a political crisis, or a change in government; other times power has already passed from the patrilineal family, leaving it in a less important position when it reaches its extinction.

      The Hasmonean dynasty[4] (/ËŒhæzməˈniːən/ (audio); Hebrew: ×—Ö·×©Ö°××ž×•Ö¹× Ö·Ö¼×Ö´×™×, Ḥašmona’Ä«m) was a ruling dynasty of Judea and surrounding regions during classical antiquity. Between c. 140 and c. 116 BCE the dynasty ruled Judea semi-autonomously from the Seleucids. From 110 BCE, with the Seleucid Empire disintegrating, the dynasty became fully independent, expanded into the neighbouring regions of Samaria, Galilee, Iturea, Perea, and Idumea, and took the title “basileus”. Some modern scholars refer to this period as an independent kingdom of Israel.[5]

      The dynasty was established under the leadership of Simon Thassi, two decades after his brother Judas Maccabeus (יהודה המכבי Yehudah HaMakabi) defeated the Seleucid army during the Maccabean Revolt. According to 1 Maccabees, 2 Maccabees, and the first book of The Jewish War by Jewish historian Flavius Josephus (37 CE–c. 100),[6] Antiochus IV moved to assert strict control over the Seleucid satrapy of Coele Syria and Phoenicia[7] after his successful invasion of Ptolemaic Egypt was turned back by the intervention of the Roman Republic.[8][9] He sacked Jerusalem and its Temple, suppressing Jewish and Samaritan religious and cultural observances,[7][10] and imposed Hellenistic practices.[10] The ensuing revolt by the Jews (167 BCE) began a period of Jewish independence potentiated by the steady collapse of the Seleucid Empire under attacks from the rising powers of the Roman Republic and the Parthian Empire.

      The author of the First Book of Maccabees regarded the Maccabean revolt as a rising of pious Jews against the Seleucid king who had tried to eradicate their religion and against the Jews who supported him. The author of the Second Book of Maccabees presented the conflict as a struggle between “Judaism” and “Hellenism”, words that he was the first to use.[28] Modern scholarship tends to the second view.

      After five years of war and raids, Judah sought an alliance with the Roman Republic to remove the Greeks: “In the year 161 BCE he sent Eupolemus the son of Johanan and Jason the son of Eleazar, ‘to make a league of amity and confederacy with the Romans.’”[45]

      … and the moon was in the Eleventh House

      The Clan Bruce ruled Scotland from 1306 to 1371. Its last royal member was King David II (1324–1371), upon whose death without issue the throne passed to his nephew Robert Stewart. The Clan continues today through other lines that do not have patrilineal royal ancestry, although the current clan chief, Andrew Bruce, 11th Earl of Elgin, is descended from King Robert Bruce in the female line.


      Pharisee and Sadducee factions … this his here; thisis now.

      Kingdom at its greatest extent under Salome Alexandra

      It is difficult to state at what time the Pharisees, as a party, arose. Josephus first mentions them in connection with Jonathan, the successor of Judas Maccabeus (“Ant.” xiii. 5, § 9). One of the factors that distinguished the Pharisees from other groups prior to the destruction of the Temple was their belief that all Jews had to observe the purity laws (which applied to the Temple service) outside the Temple. The major difference, however, was the continued adherence of the Pharisees to the laws and traditions of the Jewish people in the face of assimilation. As Josephus noted, the Pharisees were considered the most expert and accurate expositors of Jewish law.

      During the Hasmonean period, the Sadducees and Pharisees functioned primarily as political parties. Although the Pharisees had opposed the wars of expansion of the Hasmoneans and the forced conversions of the Idumeans, the political rift between them became wider when Pharisees demanded that the Hasmonean king Alexander Jannaeus choose between being king and being High Priest. In response, the king openly sided with the Sadducees by adopting their rites in the Temple. His actions caused a riot in the Temple and led to a brief civil war that ended with a bloody repression of the Pharisees, although at his deathbed the king called for a reconciliation between the two parties. Alexander was succeeded by his widow, Salome Alexandra, whose brother was Shimon ben Shetach, a leading Pharisee. Upon her death her elder son, Hyrcanus, sought Pharisee support, and her younger son, Aristobulus, sought the support of the Sadducees. The conflict between Hyrcanus and Aristobulus culminated in a civil war that ended when the Roman general Pompey captured Jerusalem in 63 BCE and inaugurated the Roman period of Jewish history.

      Josephus attests that Salome Alexandra was very favourably inclined toward the Pharisees and that their political influence grew tremendously under her reign, especially in the institution known as the Sanhedrin. Later texts such as the Mishnah and the Talmud record a host of rulings ascribed to the Pharisees concerning sacrifices and other ritual practices in the Temple, torts, criminal law, and governance. The influence of the Pharisees over the lives of the common people remained strong, and their rulings on Jewish law were deemed authoritative by many. Although these texts were written long after these periods, many scholars believe that they are a fairly reliable account of history during the Second Temple era.

      The mass and majesty of this world, all<br /> That carries weight and always weighs the same<br /> Lay in the hands of others; they were small<br /> And could not hope for help and no help came:<br /> What their foes like to do was done, their shame<br /> Was all the worst could wish; they lost their pride<br /> And died as men before their bodies died.

      She looked over his shoulder<br /> For athletes at their games,<br /> Men and women in a dance<br /> Moving their sweet limbs<br /> Quick, quick, to music,<br /> But there on the shining shield<br /> His hands had set no dancing-floor<br /> But a weed-choked field.

      A ragged urchin, aimless and alone,<br /> Loitered about that vacancy; a bird<br /> Flew up to safety from his well-aimed stone:

      “I have placed my bow in the clouds, and it will be a sign of the covenant between me and the earth.”

      Genegnosis 9:13, Adam

      These are the “block transaction” I’ve initially imported (somewhat … haphazardly) into the Rinkeby chain; the total test ETHer cost of this … “dump and semi-permafrost-etching” of the message in testnet was around $121.50 (which might or might not be a good value for printing a book on the Library of Congress’s … wall (for fake, ofc) … I am planning to write a sort of “bounty” to allow others to pay for and sign copying it … piece by piece … to the Ethereum’s “mainnet.” This might be something we …

      … [and it was evening and it was morning and now it is the old “tax day”] …

      April 15, 2020: I am sort of hacking pushing this stuff onto Rinkeby; it’s “not really liking what I’m doing” which appears to mean … literally the software really isn’t … functioning properly. Here’s files and transactions that were successfully written in “chalk”–meaning they were accepted by the network, and will probably be “mined” and eventually “immutable” (in testnet/Rinkeby). Many of the transactions were refused by the server, it appears to suggest they are “duplicate” (which they are not) and want’s me to “increaes my gasoline bidding offer” in order to … overwrite the previous … (non identical transaction). Maybe I am doing something wrong, maybe not.

      From what I’m presenting you can see … the blockchain software really isn’t designed to … "care about what you think or your opinion, or you presenting something like “a version of an implementation plan” for a bill under question–or saving that–what it’s concerned with is “pick a number 1-12” and maybe … some later “condition to revoke” or “alter” … something arbitrary. I think we need to work on “caring about the importance of our written contributions” more, as in beingt able to actually present written contributions … in a system like this wwhich is probably destined to become something like an immutable “legislation record.” There is also a 26K “appears to be a hard limit” on the size of each contract, hence the … numerous hash references for each file.

      <span style="font-size:8px;">GUNZIOUUE5H30LY1T!ABYSS.html 861e91a5a582ba5825bf2605990cfaaec58f4966 c7dbbb0cd0face04e4a4c5c8e289177f049dce39 cf1fe352bbee9a920e44d1bd81d08c1e9399504a 8d12f79f9f32e39d18c79c12d7b9fb05a55a10c5 5b94f3f3a47fd83a97b9454b4de27aa9bc0c0ad9 eb743071d2d7ee31b90315011e1881e185f5857f eaa57f9a93de06c57b593d0e9ad3de32241fe1f5 1a519e4195e933a072f9a233594e92441689d3c4<br /> 20f45f7865d7694f1ec155f48393b8d044b4cd70 b26ff67ac6c1908eb35e07eb698f3a910273c2f1<br /> 84338809f407297c43f70d0ce6cf682cd4dde5eb</span>

      <span style="font-size:8px;">UNZIOUUE5H30LY1T!ACESHI.html 03bfaea61b7873449c091b855a4d952a11719135 66e2871ef39334962fb75ce34407f825d67ec434 085f9a5ee9b677cd1b7965c2dd093d6bca77f0dc 7012a1f9bed8d89e1e0515b84dd872b629b7b60d db2837a812e97bb4b1e271918de02415e466103f 32040f6ef9dc2378961332835c7869a9ecd50a91 f8d836b29e7525c20e86813c0b601afc3e6da9a1 644125c1ce87a0a9c827e8dacea58a14bb7e11f5 c909934b0689dbbd0d8e06d347e9fb8ad332b4bb 7d44d5a2b060190b3a41c00023173e2568982b12</span>

      <span style="font-size:8px;">GUNZIOUUE5H30LY1T!ADIOSAS.html#1: dde69cb2af6599701453fbbb3a0bd7d880d16209 4b47680eb83bfb51ff3e3a7e671c02b7b68ac649 daa61dadd85e4e92f6dc386fd887c487f9fc8191 69df5c40dc70ed272b6ae481c93ba966adee9885 b517d2740e8d5383c73bf790948ed7840187c4fc e291fd040d4690599dca121749ec08450f0f6c79 b91f52a3a681aa2d04dc3af55ce8df460a7ae3c1 5cb047f0151198b83ba52f5cd36d637d2dbda859 9666b0d4d52bc23b249ee7a50bfb3fa2b5a0f848</span>

      <span style="font-size:8px;">GUNZIOUUE5H30LY1T!MOROKSRIG.html#1: 03e95b753cba381cb29bb2eafc584690f00caff4 8ad5cfd6dc1be7733be2d7f4cb0cedd00f5f3902 a54ab79af06c89fac1537b0faa0270e35e022570 daf36e641a1b5036d23565f6c9024a64c4b39876</span>

      <span style="font-size:8px;">GUNZIOUUE5H30LY1T!MISSISSIPPI.html#1: c6f805a781dc8a12a4ace58910c1b4acc295a32a 5c3a07299e2401fcb7448e6f0356f66408fd043e e9977bebbfd2e716faa8c500e22ed4b79f27cbd3</span>

      <span style="font-size:8px;">GUNZIOUUE5H30LY1T!MOLASSES.html#1: e9b2165e3f44ed36faf226645ffbd9ba2be92269 66e2871ef39334962fb75ce34407f825d67ec434 38179f71157e6cd003415ad62c34f5f47d57075a 6638ba4b319b64cd68e7b64c534118dce393bdb8 9d3f2eb7693e0e7d092055e3cc4f16ae90fe323b</span>

      <span style="font-size:8px;">GUNZIOUUE5H30LY1T!ALLTA.html#1: f81be6a8830343e9be38c1e41908d676c4309afb<br /> !AMISTAD.html#1: 824cebcb15bf6ba8a9912091654fb4ee033bbbdb | 66e2871ef39334962fb75ce34407f825d67ec434<br /> !ANDERSON.html#1: 33a55bc62f3876b0e34120d564cd9da26cfb24ad<br /> !ATCONGRESS.html#1: 0d97f662ee405e3881927d67803eef5495c87366 !ARTANWORDS.html#1: 2de0c7d3b9a657e475dc987188e2bc6686cb807d !B.html#1: cd4a5a1939c03eb46834cb959b7331dc322b288b !LANDOH.html#1: 6bf55970aa5c8b59e715a19db6f78098bb337c55 MEDICINE.html#7: 514f9c0c12ec08b12e21937311b388038138527a MUAH.html#1: 741ada306a5b3a1e0d4dd7aa3d7c2274936faf53</span><span style="font-size:8px;"> !PIMB.html#1: b56c131d9e67daaae253278b8b08b3eff15d5b1f</span>

      The above are examples of “file transactions that appear to have been committed in whole” … there are several partial files written; and I will probably write a “diff system” to only resubmit the missing chunks; in the future… or soon.

      GUNZIOUUE5H30LY1T!OUITHEPPL.html#1: 32a4c303e557899e6f7c119bf8e977ffa2369df8 | 66e2871ef39334962fb75ce34407f825d67ec434 … that second hexadecimal number is my “wallet ID” … you can search through Rinkeby’s ledger and see all of the other … TXN’s that were posted today; the plan or hope is that you will abe able to; anyway. The little hack code I used doesa in fact work and produce a contract on the chain, for the first few small tests I did. These needs to be “mined” and that involves some cryptographic … stuff … called “proof of work” that I don’t really understand; but it means there’s a delay, and a bigger delay because there’s “more data.” I do of course have a special afinity for this particular “file” it’s the one where I basically “added” (in my mind) some Bible’s words preceding Revelation 1:1 … to the actual “Bible.” The word in question, of coruse is “dox<s>ic</s>ology.” I think you can send… actual Ethereum there. I’m pretty sure, you could.

      dox·ol·o·gy /däkˈsälÉ™jÄ“/

      noun<br /> noun: doxology; plural noun: doxologies

      1. a liturgical formula of praise to God.

      They call these identifiers “immutable” … of course this is “testnet1” … I will do “testnet2” which is some name reminiscient of “Robespiere” … probably tomorrow. See reading “immultable” I’m messy, u Tableland; sort of like in unison with the Upanishads. I imagine this is my “birthday present” for this year’s Had’ad day, and I do hope the “immultapl pen” is … enshrined or enschewed or … see etched in the ethereum blockchain … hopefully for good purpose and use “forever.” Imagine that, added to the newly modified GPL2MODS.3 license:

      Unless otherwise indicated, this work was written between the Christmas and Easter seasons of 2017 and 2020. The content of this page is released to the public under the GNU GPL v2.0 license; additionally any reproduction or derivation of the work must be attributed to the author, Adam Marshall Dobrin along with a link back to this website, fromthemachine dotty org.

      That’s a “.” not “dotty” … it’s to stop SPAMmers. :/

      This document is “living” and I don’t just mean in the Jeffersonian sense. It’s more alive in the “Mayflower’s and June Doors …” living Ethereum contract sense and literally just as close to the Depp/C[aster/Paglen (and honorably PK] 'D-hath Transundance__sense of the … new meaning; as it is now published on Rinkeby, in “living contract” form. It is subject to change; without notice anywhere but here–and there–in the original spirit of the GPL 2.0. We are “one step closer to God” … and do see that in that I mean … it is a very real fusion of this document and the “spirit of my life” as well as the Spirit’s of Kerouac’s America and Vonnegut’s Martian Mars and my Venutian Hotel … and my fusion of Guy-A and GAIA; and the Spirit of the Earth … and of course the God given and signed liberties in the Constitution of the United States of America. It is by and through my hand that this document and our X Commandments link to the Bill or Rights, and this story about an Exodus from slavery that literally begins here, in the post-apocalyptic American hartland. Written … this day … April 14, 2020 (hey, is this HADAD DAY?) … in Margate FL, USA. For “official used-to-v TAX day” tomorrow, I’m going to add the “immultible incarnite pen” … if added to the living “doc/app”–see is the DAO, the way–will initi8 the special secret “hidden level” … we’ve all been looking for.

      Nor do just mean this website or the totality of my written works; nor do I only mean … this particular derivation of the GPL 2.0+ modifications I continually source … must be “from this website.” I also mean the thing that is built from … bits and piece of blocks of sand-toys; from Ethereum and from Rust and from our hands and eyes working together … from this place, this cornerstone of the message that is … written from brick and mortar words and events and people that have come before this poit of the “sealed W” that is this specific page, and this time. It’s 3:28; just five minutes–or is it four, too layne.

      This work is not to be redistributed according to the GPL unless all linked media on Youtube and related sites are intact–and historical references to the actual documented history of the art pieces (as I experience/d them) are also available for linking. Wikipedia references must be available for viewing, as well as the exact version of those pages at the time these pieces were written. All references to the Holy Bible must be “linked” (as they are or via … impromptu in-transit re-linking) to the exact verses and versions of the Bible that I reference. These requirements, as well as the caveat and informational re-introduction to God’s DAO above … should be seen as material modifications to the original GPL2.0 that are retroactively applied to all works distributed under license via this site and all previous e-mails and sites. /s/ wso

      </div> Bold

    1. Note: This rebuttal was posted by the corresponding author to Review Commons. Content has not been altered except for formatting.

      Learn more at Review Commons


      Reply to the reviewers

      We thank the three reviewers for providing insightful critiques on our manuscript.

      Changes to document and comments made are marked e.g. “Reply 1.1” (referring the Reviewer #1 item #1, etc.) as described below.

      Reviewer #1

      I found this study to be very convincing. Prior studies are referenced appropriately, the text is well written and clear, the figures are clear also. In my opinion the paper does not need further experiment.

      [1.1] The conclusions are well supported by the data. However, the concatenation model seems very speculative at this point. Also, it does not take into account the dynamics of these molecules.

      Reply 1.1: The concatenation model combines the structural data from our manuscript with prior biochemical insights into tetraspanin homodimerization and with scanning-EM data on immunogold-labeled CD81 and CD9 on cells. It is not completely clear to us what reviewer #1 refers to with “the dynamics of these molecules”. The cryo-EM data revealed that CD9 - EWI-F is a dynamic complex with straight and bent conformations, which could account for both circular and linear arrangements of tetraspanin-microdomains in cell membranes through the higher-order oligomerization of stable CD9 - EWI-F tetramers. Moreover, transient CD9 - CD9 interactions likely yield a variable number of complexes present in these concatenated and flexible strings of complexes. Such a concatenation model indeed requires further validation. However, it is consistent with experimental data and, importantly, provides a long-awaited molecular basis for TEM assembly. Although it was not within the scope of the current study, it will be of great interest to further investigate the concatenation model through detailed cell-biology based approaches.

      **Minor comment:**

      [1.2] There seems to be a mix up between the two structures in the following sentence p4: "In CD9EC2 - 4C8, the D loop adopts a partially helical conformation and central residue F176 is sandwiched by 4E8 residues W59 of CDR2 and W102 and R105 of CDR3 (Fig. 1D). In the 4C8-bound CD9EC2 structure the tip of the D loop points more outward and the Cα atom of F176"

      Reply 1.2: The first sentence indeed mixed up the two structures and wrongfully mentioned CD9EC2 - 4C8 instead of CD9EC2 - 4E8. This has now been updated: “In CD9EC2 - 4E8, the D loop adopts …”

      Reviewer #2

      The paper is well written and the conclusions made are supported by the data presented.

      [2.1] The ternary structure is in agreement with that of CD9 in complex with the related EWI-2 published earlier this year by Umeda et al (ref #25). The present work thus adds little structural insights but may be useful in showing that the interaction pattern seen extends to another EWI protein family member.

      Reply 2.1: We agree with reviewer #2 that that the CD9 - EWI-F structure presented in our work is similar to the CD9 - EWI-2 structure published recently by Umeda et al. (ref #25). However, as also pointed out by reviewer #1, we believe that the CD9 - EWI-F structure adds new important information to understand the molecular mechanism underlying the assembly of tetraspanin-enriched microdomains. Notably, the different conformations of the CD9 - EWI-F complex observed in the cryo-EM data provide structural biology evidence for the dynamic nature of the interaction between a tetraspanin and a partner protein, which is consistent with a wealth of prior biochemical data. Guided by the distinct shape of the CD9EC2 - 4C8 densities, we were able to distinguish a range of straight to bent conformations of the complex. CD9 regions that represent known tetraspanin homo-dimerization sites, orient away from EWI-F and are available for interactions. Thus, combining our structural data with previous biochemical interaction data allowed for the generation of a long-awaited model for the assembly of tetraspanin-microdomains at the molecular level. We believe that these implications for TEM assembly will stimulate new, innovative research into the molecular principles that govern the function of tetraspanins.

      [2.2] As such it may be acceptable for publication. In this case, the authors should improve the quality of Figs. 3D and 4D.

      Reply 2.2: Figures 3D and 4D depict raw cryo-electron microscopy images (micrographs). The protein complexes imaged in this study only contain light atoms (H, N, C, O, S). Therefore, the collected micrographs only reveal low-contrast images of protein particles, and, for a typical cryo-EM experiment, it is required to average particles from thousands of micrographs to obtain a 3-dimensional reconstruction. We would like to keep the raw micrographs in figures 3 and 4, as it will aid cryo-EM scientists in judging the quality of the data.

      Reviewer #3

      The work is technically well performed and clearly presented including methodological details. I just have a few minor comments:

      [3.1] Page 4 and Figure S1: it is hard to see how a reliable affinity for 4E8 can be obtained from the cell binding data in S1A, as there is no indication of saturation. It would be good to at acknowledge that this is at best a rough estimate. Fortunately the data for this nanobody in purified situation seems solid.

      Reply 3.1: The obtained affinities are indeed an ±estimation based on a non-linear regression curve fitting on the measured data, performed in triplicate. The text has been updated and now reads as “4C8 and 4E8 bind to purified, full-length CD9 as well as to endogenous CD9 expressed on HeLa cells with apparent binding affinities in the nanomolar range (Fig. S1A, B, C)”. Next to that, a table stating the calculated KDs has been included as Fig. S1C.

      [3.2] Page 6: Does the absence of micellar density for the EWI-F complex indicate flexibility of the extracellular domain relative to the TM? Does this happen because the classification focuses on the highly elongated Ig region?

      Reply 3.2: These are indeed plausible assumptions. We observed highly heterogeneous, elongated particles in the micrograph shown in Fig. 3D, indicating inter-domain flexibility. If the alignment software focusses on certain Ig-like domains, other regions of the protein complex will be averaged out. An additional complexity with these elongated particles was to select an appropriate box size for particle picking and particle extraction, because the particles differ greatly in size based on their orientation (fully elongated side-views vs. much smaller top-views). When taken together, the complex of CD9 with full-length EWI-F was unsuitable for high-resolution structure determination; the subsequent strategy using EWI-FΔIg1-5 resulted in globular particles with less flexibility (Fig. 4D), which allowed for a more detailed structural characterization of the complex.

      [3.3] Page 8: "Recently, a cryo-EM density map has been reported..." - please reference here.

      Reply 3.3: We added the appropriate reference to the sentence: “Recently, a cryo-EM density map has been reported of CD9 in complex with an EWI-F homolog, EWI-2 (25).”

      [3.4] Relatively little is known about how tetraspanins help to organize partner receptors into defined membrane domains, evidence for which has emerged from super-resolution light microscopy. Based on their structural analysis of the CD9-EWI-F complex, including the heterogeneity apparent in the cryo-EM structure, they propose a feasible concatenation model for higher order oligomerization of these complexes in the membrane. Obviously the model will need to be tested rigorously by mutational analysis, particularly the EWI Ig6 interface, but as it stands the paper is a significant contribution to the field of tetraspanins.

      Reply 3.4: From the 8.6 Å cryo-EM data, the amino-acid residues that form the EWI-F Ig6 dimer interface can indeed not be distinguished. However, our data on CD9 in complex with full-length EWI-F (Fig. 3E) and previous cross-linking data (André et al. In situ chemical cross-linking on living cells reveals CD9P-1 cis-oligomer at cell surface - PMID: 19703604) support that EWI-F forms dimeric assemblies. Regarding the concatenation model, we therefore think that it will be of great interest to establish the putative CD9 - CD9 interactions (identified through biochemical approaches), that would link CD9 - EWI-F tetramers into higher assemblies, in the context of native membranes. However, investigating these transient interactions would require various non-trivial experiments and was therefore not within the scope of the current study.

    1. ’agit notamment de deux personnes, Paul Otlet et Vannevar Bush. On leur associe à chacun une œuvre écrite majeure ; respectivement le Traité de documentation (1934) et l’essaie As We May Think (1945) ; a

      ici, historiquement, impossible de ne pas parler du vrai pionnier et qui produit vraiment un appareillage technique : Emanuel Goldberg https://www.reseau-canope.fr/savoirscdi/societe-de-linformation/le-monde-du-livre-et-des-medias/les-penseurs-de-linformation-de-la-documentation-et-de-la-pedagogie/emanuel-goldberg-le-pionnier-oublie-des-systemes-dinformation.html

    2. respectivement

      La phrase est lourde. Mieux :

      On leur associe à chacun une œuvre écrite majeure – respectivement le Traité de documentation (1934) et l’essai As We May Think (1945) –, ainsi qu’un projet concret issu de leurs recherches – le Mundaneum et le Memex.

      ou

      On leur associe à chacun une œuvre écrite majeure et un projet concret issu de leurs recherches : le Traité de documentation (1934) le Mundaneum pour Otlet, l’essai As We May Think (1945) et le Memex pour Bush.

    1. I think I should indicate why I am here in Birmingham, since you have been influenced by the view which argues against "outsiders coming in." I have the honor of serving as president of the Southern Christian Leadership Conference, an organization operating in every southern state, with headquarters in Atlanta, Georgia. We have some eighty five affiliated organizations across the South, and one of them is the Alabama Christian Movement for Human Rights. Frequently we share staff, educational and financial resources with our affiliates. Several months ago the affiliate here in Birmingham asked us to be on call to engage in a nonviolent direct action program if such were deemed necessary. We readily consented, and when the hour came we lived up to our promise. So I, along with several members of my staff, am here because I was invited here. I am here because I have organizational ties here.

      ~ You may be under the impression that I arrived here in Birmingham unannounced. But, actually, I was invited to engage in nonviolent protest and to further my already established organizational ties in the area.

    1. Importance of Resume for a Job Aspirant By Kush Koachar <!-- google_ad_client = "ca-pub-4760971685927876"; /* 728x90 */ google_ad_slot = "4742646148"; google_ad_width = 728; google_ad_height = 90; //--> Resumes are utilized to establish a positive connection with a forthcoming manager. Your resume is regularly the initial introduction a potential business has of you. Consequently, it is frequently referred to as one of the most crucial steps taken during a job search.   Without an ideal beginning impression, a prospective employer is likely to stop considering you as a suitable candidate for the job on offer and move on to other candidates who have provided better resumes.   Think about a resume as an instrument for advertising yourself. It's something other than a record: It plots your experience, your aptitudes, and your instruction with the goal that a potential business is rapidly and effectively ready to perceive how your individual encounters can add to an organization's prosperity.   On the off chance that you've never composed a resume, don't stress. Learn how to compose a resume to figure out how to arrange your resume, which data to utilize, and considerably more.   The resume acts as a bridge between you and the prospective recruiter. Henceforth the significance of a resume can never be thought little of. Along these lines, to establish the principal connection, it is basic that your resume stands apart from the group first. It is up to you how would you like to be recollected by the employing chief? Since organizations don't have that much measure of time to meet every single applicant, they require resumes from contender to choose the best ones to work with them.   Dismissal occurs, and it continues endlessly until they discover something fascinating in one specific resume. This is the point at which an all around organized, perfect and exact resume has its impact. You may be thinking for what reason is it so critical to have a resume?   Resume reaches the recruiter’s table much before than you do: Yes, before you reach recruiter’s office, your resume does so. By and large, every organization requests your resume first, they experience the work that you have done as such far and on the off chance that it coordinates their prerequisite, approval! Thus, an elegantly composed resume does half of the work for you here as it were. Subsequently it is a lot of critical to have an organized resume to establish the primary impression work for you.   Resume tells about you: A resume talks a lot about you as an expert. It says what you have done previously. What are you doing right now and where precisely you are going towards? Simply envision a little bit of paper talks such a great amount about you including past, present, and future. Doesn't excessively solid stunning? Be that as it may, recall this account of past, present and future should be passed on rapidly, else they will lose intrigue. Thus, this activity of recounting to a short story can be minimal dubious. Henceforth you may require help in drafting your resume by experts.   Yunic Solutions is a HR Consultancy firm providing assistance and guidance to many businesses in managing their team as well as helping them in acquiring quality team members. We came across 100s of resumes every day and only selected few actually passes the initial assessment. Resume writing is an art and it should be mastered by job applicants who wants to increase their chance of getting recruiting.

      Resumes are utilized to establish a positive connection with a forthcoming manager. Your resume is regularly the initial introduction a potential business has of you. Consequently, it is frequently referred to as one of the most crucial steps taken during a job search. Resume Writing Services

  4. Jul 2020
    1. Yes, the classroom may pose the most risk, as students will spend the most time there and are in proximity to others who may cough or sneeze. This will certainly be the case during cold and flu season. But we think that with proper funding, classrooms can be made relatively safe

      And this is another counterargument + refutation.

    1. Reviewer #3:

      In this manuscript, Urchs and colleagues use transductive conformal prediction (TCP) applied to rsfMRI functional connectivity data to predict autism in a subset of cases. The approach is novel for applying to autism research and also is pinpointed at a topic that is very much needed in autism - the problem of heterogeneity. The logic applied is that only a subset of autism cases will have powerful biomarker differences in terms of resting state functional connectivity and TCP is utilized to isolate that subset. Thus, while the approach is novel and maps onto similar kinds of logic in the realm of genetics of autism, the utility is somewhat limited, as TCP will not be able to tell us much about the majority of cases. This is the same problem with many highly penetrant genetic mechanisms that lead to high risk for autism. However, it is still an issue that the approach can only make statements about a very small percentage of the total autism cases in the population. Could the authors comment more on this issue/limitation? For instance, what does this biomarker in a small percentage of cases tell us? Are there powerful, specific, and homogeneous biological mechanisms behind such cases, whereas for the rest of the population the underlying mechanisms are highly diverse and not powerful enough to penetrate up into macroscale functional connectivity phenotypes? The result could help to generate new hypotheses focused on such a group. However, I think the authors should try to lead readers in discussing how to take such results further for new discoveries.

      Besides this main issue noted above about the utility or meaning behind the novel findings, the following are comments about how to make the introduction more readable, and how to potentially better facilitate a reader's understanding of the analyses.

      1) Introduction: I would suggest that some modifications need to be done to the introduction in order to make the ideas flow a bit better. The problem is that the authors are introducing a variety of complex and not necessarily easily linked information - e.g., risk from a variety of different types of genetic mechanisms, failure of neuroimaging classifier studies, and TCP. With a bit of effort and a couple re-readings it is clear that the logic the authors are using is that we have some understanding of how much risk there is from different types of genetic mechanisms, and we would like to understand how neuroimaging data might match up to that. Using TCP would hopefully allow you to do that, hence the goals of the study. This logic is not clearly spelled out as one reads the introduction however, because the different topics are either mixed together within a paragraph with little linking text to help the reader follow the logic, or the bits of information for each topic are segregated into their own paragraphs with little linking text and the beginning or ends of the paragraphs to help the ideas flow from one paragraph to the next. A good example of this is that the background paragraph to start with has these topics mixed together within the very first paragraph, and then the subsequent 3 paragraphs solely focus on each topic, without helping the reader understand why they are jumping from very different topics. By the time the reader gets to line 120 of the Objectives, then things are spelled out a little better, but the reader has to then go back and connect the ideas about how the authors are trying to compare how a TCP approach to identify a high risk imaging marker would match up against more well known risk markers at the genetic level. It may be the case that the manuscript here will get readers of various different backgrounds (e.g., autism researchers, those with expertise in genetics, neuroimaging, or machine learning). Few have expertise in all those areas, and for those individuals, it may be hard to understand how these different topics flow together and are linked in a specific logical way. The logic is there, but even for this reviewer, it required a couple readers to see how all this information lined up in a logic way to justify the study. Thus, I would suggest that the authors make changes to the writing so that the reader can clearly follow the logic without too much extra effort to connect what isn't written about how these topics are supposed to line up.

      2) Methods: The methods and analysis are fairly complex. Can the authors make a figure that clearly lays out the analysis pipeline? It would help to have a visual that clearly outlines how the authors selected the subset of individuals from the larger ABIDE datasets, how the preprocessing was done, how the features were estimated, and how the TCP analysis was implemented with all the associated added aspects like the bootstrapping, etc. Furthermore, to facilitate understanding of the complexities of the analysis, can the authors create a GitHub repo that has all the reproducible analysis code that generates the results and figures produced in the paper, along with tidy data files that have the features used by the TCP model? Although in the data availability statement the authors write that a GitHub repo exists, having had a look through this, no tidy data files are available that the code can load up to have readers reproduce the analysis or figures. In addition, the code consists of only 4 brief R scripts. That code isn't easily readable with regards to how the analysis was done. The R code could be done in another way that is more in line with literate programming, such as an Rmd file, that has the analysis code, along with plain text to describe the different steps, and then the figures embedded within the html or pdf report that it creates when it is knitted in R Studio. There are also some Jupyter notebooks that show how the figures were generated. This was helpful to see and is what is needed for the R code too. In those Jupyter notebooks, it seems like there are certain tidy data files that those notebooks load, but they are absent in the repository and therefore, the readers cannot reproduce the analysis.

    1. Reviewer #1:

      This is a very ambitious and interesting study that uses a state-of-the art combination of multiple methods to provide new insights into functional network interactions during motor learning. However, I have several major concerns against the design and analyses that may have contributed to the overall very weak effects that are reported (mainly null effects in standard measures at the behavioural and neural network level). I also think that some of the conclusions are not justified given the partly non-significant and overall weak effects.

      1) My main concern is that no baseline stimulation condition (sham TBS) was included. The authors address this in the discussion but I cannot agree with their argumentation. Without a baseline, it is impossible to assess whether each stimulation protocol had a significant impact on the outcome measures. For instance, it would be plausible that both protocols had opposite effects (which is also hypothesized by the authors) which were, however, only slightly or not significant from baseline. If cTBS slightly decreases connectivity and iTBS slightly increases it, this could result in a difference between both protocols that might not be observed when contrasting each protocol against baseline. Put differently, how do we know that these changes are meaningful and significantly different from zero (baseline)? I think this is especially important in the present study since the overall effects are weak and there is no significant modulation of behavior - so the functional / behavioral relevance of the observed modulation remains unclear. I think that without the inclusion of a baseline (sham), it is very hard to interpret the data.

      2) Another main concern is that the reported effects are very weak and not properly corrected for multiple comparisons. I don't think that it is justified to apply small volume corrections for large-scale network effects and it seems that some of the results are at threshold. Given the weak effects in these analyses, in combination with the absence of any modulation in the "standard" analyses (fMRI, connectivity, behaviour, MRS only significant in exploratory post-hoc tests which are not well justified), I am not sure if the reported results are really reflecting any stimulation-induced modulations at all or mainly show some noise added by the TMS protocols. This of course affects the conclusions that can be drawn from the study.

      3) There are a number of issues with the design that might have contributed to the weak findings. These include data loss (e.g. no MRS data for the hippocampal voxel) and somehow arbitrary sample sizes that are not well justified. I am also not sure why cortical excitability measures (MEPs) were performed after TBS because this is of minor importance and delayed the start of the fMRI sessions. Given that TBS effects are expected to decrease over time, I am not sure if this was necessary. Was the potential change of the TBS effects across session taken into account (e.g., by using a parametric modulation of the TMS effect)?

      4) Given the overall weak effects the conclusions should be toned down. The discussion would further benefit from including additional work that demonstrated changes in remote subcortical regions and effective connectivity after TMS over a frontal area (e.g. Herz et al., J Neurosci 2014).

      5) There is no modulatory effect of TBS on behaviour, which is surprising in light of previous neurostimulation studies on motor learning. I think the way this is sold in the discussion is a bit odd. I guess that initially, one would have expected a behavioural modulation that should ideally be correlated with any TBS induced changes in functional connectivity (or with the MRS data). If not, how would you be able to claim behavioural relevance? In the discussion, the absence of a behavioural modulation is sold as an advantage, I think this is not justified and should be toned down. Moreover, since the authors speculate about potential influences of TBS on motor consolidation, I was wondering if consolidation was assessed (which seems to be a relevant parameter here)?

    1. While we find the term 'making' useful as a method for democratizing the fabrica-tion of technological objects, we also see that with this larger social adoption, the adversarial, political, and tactical components at the heart of many tech-oriented DIY practices have been largely removed and replaced with a singular interest in technological skill or craft. In other words, the popular concept of maker has unified an interest in a hands-on involvement in technology development, but has done so by subtracting critical engagement from the pro-cess

      I wonder whether there's been a sort of pendulum swing here. My grandparents and parents grew up "making" - (re)building cars, a motorhome, etc. This was partially out of interest in projects (they liked and still like making things), partially financial constraints. There wasn't a critical engagement in the ways I think Ratto means here. And then hacker/maker culture (early maybe) there was some critical engagement, lost maybe in the popularization. But I guess from my perspective both have existed alongside each other for a while. My local makerspace (and one I mentioned earlier) may not always be thinking about the adversarial/political/tactical components of their tech-oriented DIY practices, but those are present for some in the organizations. Overall though, I'm all for Ratto's argument that we could use more of this! I find some of the "open" culture frustrating because of a deeply ideological commitment on the part of some to "open", even when the real impact of "open" is that large corporations are the main beneficiaries of data and shape it toward the needs of capital, rather than the benefit of the people writ large.

    1. To promote notions of Civic Engagement by: a) learning how actions and initiatives taken by residents living in East Harlem address concerns in the neighborhood; b) learning how East Harlem residents influence and impact El Museo as well as the ghborhood; c) considering how they can be agents of change in their own communities.

      A great place to start with inspiring civic engagement is in one's own community. One of the goals of this curriculum is for students to understand how members of a community are addressing their challenges in order to provoke students to consider how they may be agents of change in their own communities. This makes me think of Oyler's (2012) description of Mr. Schultz guiding his students along a journey of individual growth and community activism by first asking his students the question, “what are some problems that affect you and your community?”. Being rooted in the challenges of one's own community is starting point for student activism that can inspire students be being focused on real-world issues that effect their daily lives. As Greene states, “We require curriculum that can help provoke persons to reach past themselves and to become” (p. 220). According to this goal, students may reach past themselves to become advocates of their communities.

    1. Author Response

      Reviewer #1:

      The manuscript by Mitchell et al. finds that the NAIP-NLRC4 inflammasome in mice is a critical host factor that controls intestinal infection with the human specific bacterial pathogen Shigella flexneri. The work suggests that Shigella is actively suppressing the human NAIP-NLRC4 inflammasome possibly using an T3SS effector protein, which does not recognize its substrate in mouse cells. The authors use this information to determine that B6 mice lacking the NAIP or NLRC4 inflammasome components are susceptible to Shigella infection and observe disease symptoms similar to Shigellosis in humans. In addition, 129 mice exhibit additional disease symptoms, and the authors suggest that loss of Caspase-11 in 129 mice is responsible for this phenotype.

      The strengths of this manuscript include the introduction of a new mouse model that mimics Shigellosis, the demonstration that NAIP/NLRC4 activation is important for epithelial cell defense, and the potential of these findings to clarify aspects of human infectious disease caused by this pathogen. The manuscript is well presented, and the experiments are conducted with a high degree of rigor. Overall, this is an important contribution to the Shigella field and also has significant implications on our understanding of inflammasomes in host defense against pathogens.

      Response: We thank the Reviewer for recognizing the impact and rigor of our work.

      There are some weaknesses that should be addressed. Experimentally, it has not been directly demonstrated that IECs from NLRC4-/- mice undergo cell death (using biochemical markers). This is a critical aspect of the model.

      Response: Prior work in the field (e.g., Sellin et al, 2014; Rauch et al, 2017) has already established that inflammasome activation in IECs results in their death and expulsion from the intestinal epithelium. We are currently working on showing this also occurs with Shigella but we have no reason to doubt that it does; our preliminary data indicate that Shigella-infected propidium iodide (PI)-positive cells are expelled from IEC monolayer cultures in an NLRC4-dependent manner. We intend to provide these data in a revised version of the manuscript.

      In addition, it would be useful for the authors to evaluate bacterial burden over the time course in Figure 6. Although this is not absolutely necessary to support the manuscript conclusions, this information would greatly benefit the community that intends to use these mice in the future.

      Response: This is indeed an experiment we plan to complete in the future. At present we are constrained by the numbers of available mice. We agree with the reviewer that the timecourse is not essential to establish the main conclusions of the present manuscript, and have thus prioritized other experiments.

      There are also some discussion points about the mouse model that would enhance the overall impact of the work. For example, a more in depth discussion about the differences between human Shigella infection and the new model would be helpful. It is important to emphasize that the mouse model requires a much greater inoculum of the pathogen to induce disease and requires microbiota-deficiency to be effective. What are the implications of this finding on our understanding of human disease?

      Response: Although it is often (correctly) stated that as few as 10-100 bacteria can infect humans with Shigella, there is actually considerable heterogeneity in the infectious dose. DuPont et al 1989 summarizes several human challenge studies in their Table 1, which shows that while 25-39% of humans exhibit symptoms after low dose infection (<200 CFU), 36-44% of humans are resistant to high doses (10^4-10^8 CFU). Therefore we do not consider the infectious dose in our mouse model to be out of the range of what is ‘normal’ in humans. Indeed, our new model may help us understand some of the factors that confer resistance to certain humans. We used a dose of 5x10^7 in our manuscript to ensure reproducible infection of all mice. However, in limited studies, we have observed disease in oral route infected, antibiotic pre-treated NAIP–NLRC4-deficient mice with 10^6 CFU (4/4 mice) and 10^5 CFU (2/3 mice). We are currently repeating these experiments, which we intend to include in a revised manuscript. We also agree with the reviewer that the infectious dose in humans vs. mice merits more discussion in a revised manuscript.

      In lines 274-285 the authors present an either/or scenario in which either macrophage pyroptosis is required for IEC infection or inhibition of NAIP/NRLC4 pyroptosis in IECs is required for IEC infection. However, these scenarios are not mutually exclusive. For example, it is plausible that the extremely low burdens of Shigella required to infect humans (<100 CFUs) is due to the pathogen initially crossing the epithelial barrier (e.g. through M-cells) to infect macrophage, and then re-infection of IECs after macrophage pyroptosis. In this scenario, the NAIP/NLRC4 inflammasome could prevent further expansion of bacterial in IECs by eliminating the cell-to-cell spread that have been described by others. Importantly, the macrophage lifecycle stage may not be necessary in mice in which the microbiota has been removed and Shigella is delivered at a very high inoculum. While, additional ideas could be, and should be, put forth since the mouse model provides new insights or challenges an existing dogma in the field.

      Response: We do clearly state in our manuscript (line 277) that our results do not directly address the question of whether Shigella might benefit from inflammasome activation in macrophages. In a revised version of the manuscript we will further expand on the discussion of the role of inflammasomes in macrophages and IECs to acknowledge multiple, non-mutually exclusive scenarios.

      Reviewer #2:

      Mitchell et al explore the role of NLRC4 in defending against Shigella infection by demonstrating that NLRC4 contributes to resistance to shigellosis in mice. Using in vitro assays, they first show that mouse but not human macrophages undergo NLRC4-mediated pyroptosis in response to Shigella infection despite an ability for both species to successfully detect Shigella NLRC4 agonists. They then demonstrate that C57BL/6 background mice, which normally resist shigellosis, become susceptible to infection when deficient in NAIPs or NLRC4. In parallel, 129 background mice develop more significant infection including intestinal bleeding. Furthermore, using a mouse line in which NLRC4 expression is restricted to intestinal epithelial cells (IECs), they show that IEC expression of NLRC4 is sufficient to resist shigellosis. Finally, using a known attenuated Shigella mutant, they demonstrate that their shigellosis model can mimic kinetics seen in humans.

      Mitchell et al convincingly demonstrate both the importance of NLRC4 in protecting mice against Shigella and the utility of their mouse model for studying Shigella infections, both of which are significant and will push the Shigella field forward. There are mechanistic questions to be addressed in future studies beyond the current manuscript, attesting to the importance of the paper in opening up new areas in the field of research. In some places, the authors draw conclusions that reach beyond what is proven in the data, which should be addressed in text edits to the manuscript. In summary, this article presents an important new model for Shigella infection. The impact of the manuscript is the development of a mouse model with which to study Shigella infection in vivo.

      Response: We thank the Reviewer for emphasizing the importance of our new shigellosis model for the field. We have addressed their comments below.

      Major comments:

      Many questions remain concerning why NLRC4-deficient THP1 cells still undergo pyroptosis. The authors provide evidence that Shigella activates PYRIN and/or AIM2 inflammasomes in humans, and that somehow mouse macrophages would fail to have this same detection. At face value, the data would suggest that humans are able to detect Shigella by Pyrin and AIM2, but for some reason these two inflammasomes are insufficient, and instead NLRC4 is required for in vivo defense. Then in mice, it would imply that everything is flipped - for some reason detection by Pyrin and AIM2 is not important, but now the bacteria can be detected by NLRC4 and this is important. The NLRC4 focused conclusions are consistent with the in vivo data, that NLRC4 in humans fails to detect, but NLRC4 in mice succeeds in detecting Shigella. However, the data that Pyrin and AIM2 in human cells successfully detect Shigella are inconsistent with the overall conclusions of the paper. I suspect that this is an artifact of THP1 cells, and that the in vivo situation in humans is that these two inflammasomes will fail to detect Shigella. There is published precedent from other infections where in vitro detection belies in vivo lack of detection (e.g. Listeria is detected by AIM2 in vitro, but probably not in vivo). It may be difficult to make direct comparisons between how inflammasomes act in THP1 cells as compared to BMMs, due to artifacts arising from the different origins and passage levels of the two cell types. It may be that the inflammasomes response is most important in IECs, as proposed by the authors, and that IECs may not express Pyrin or AIM2. There is evidence from publicly available IEC transcriptional profiles that IECs do not express Pyrin (Mefv) (Reikvam, doi: 10.1371/journal.pone.0017996), although this profile does show Aim2 expression in IEC. It is my understanding that BMMs do not express Pyrin unless they are strongly stimulated with some TLR agonist. As it stands, the in vitro data appear to contradict one of the main conclusions of the paper, because it would seem that human Pyrin and AIM2 inflammasomes can detect Shigella, and so these should compensate for NLRC4. The explanation as to why Pyrin and AIM2 are insufficient to compensate for NLRC4 evasion in human infection should be addressed at least in discussions of the data to explain the apparent discrepancy.

      Response: The reviewer states that our claim that human PYRIN and AIM2 inflammasomes can detect Shigella in THP1 cells is “inconsistent” with the overall conclusion of our paper, which is that the NLRC4 inflammasome provides necessary defense of mouse intestinal epithelial cells. We do not agree that there is an inconsistency and indeed many of the points the reviewer makes in their comments fit with our view, so perhaps there is less disagreement than it might seem.

      As the reviewer discusses, differences in inflammsome expression in humans vs. mice, and in IECs vs. macrophages vs. THP1 cells, and the kinetics of inflammasome responses, as well as several other factors, can easily account for the results we obtain. It appears that PYRIN is not well expressed in mouse IECs (Price et al. 2016), at least not uniformly at levels in all cells that are sufficient to confer protection. AIM2 is expressed in colonic IECs (Price et al. 2016), but it is not clear that it would be engaged in every infected IEC. For example, AIM2 detects bacterial DNA, which might only be released if the Shigella bacteria lysed in the cytosol. As noted by the reviewer, this may be a relatively rare event, as previously documented for AIM2 activation by Listeria-infected macrophages (Sauer JD et al, 2010). AIM2 activation may also be kinetically delayed in IECs. It appears instead that NLRC4 is the main inflammasome that can respond to Shigella in mouse IECs; thus loss of NLRC4 is sufficient to lead to susceptibility of mice. It remains possible that there is some functional AIM2 or PYRIN (or CASP11 or NLRP1B) in mouse IECs; thus, the further removal of these inflammasomes might lead to even greater susceptibility. Alternatively, a low level of activation mediated by these additional inflammasomes (perhaps in macrophages instead of in IECs) might even be necessary to produce the inflammation that causes disease symptoms.

      In humans, consistent with our data in Fig. 1, we propose that the NLRC4 inflammasome is antagonized or otherwise evaded by Shigella. The reviewer wonders why PYRIN or AIM2 cannot compensate for NLRC4, and is suspicious that the activation of PYRIN/AIM2 we observe in THP1 cells is not representative of what would occur in vivo. Certainly we agree that THP1 cells are non-physiological and we do not attempt to make claims in the manuscript that our observation of AIM2/PYRIN activity in these cells means anything for human shigellosis.

      The reviewer states: “the in vitro data [in THP1 cells] appear to contradict one of the main conclusions of the paper, because it would seem that human Pyrin and AIM2 inflammasomes can detect Shigella, and so these should compensate for NLRC4.” For all the reasons discussed above, we do not agree there is a contradiction. There are many reasons why PYRIN and AIM2 might function in THP1 cells (and possibly even human macrophages) but would not compensate for NLRC4 in IECs.

      In sum, we agree that there is more to learn about which inflammasomes, if any, are activated by Shigella in human IECs, but given the many uncertainties, we do not feel it is fair to say that our results are internally contradictory. We will endeavor to discuss some of these points in a revised manuscript.

      Reviewer #3:

      Mitchell et al describe the development of a mouse model for shigella gastroenteritis, the lack of which has been a serious impediment to Shigella research. They identified a difference in recognition of shigella between human and mouse Naip/NLRC4 which contributes to the resistance of mice to Shigella gastroenteritis. They suggest that Shigella specifically inhibits human Naip/NLRC4 activation and that the difference between mice and human susceptibility to infection is due to differential inhibition. This was confirmed by the ability of NLRC4-/- mice can recapitulate human infection. Furthermore they show that it is inhibition of NAIP-NLRC4 in IEC that is required for infection to occur. This manuscript therefore describes a number of important findings and uses these to develop a very useful animal model of shigellosis.

      We are grateful for the Reviewer’s comments and suggestions, and provide point-by-point responses below:

      I have three suggestions that I believe would improve the manuscript:

      1) Determine the inflammasome that causes cell death in Shigella-infected THP1's. WT Shigella infection did not induce pyroptosis of colchicine-treated (PYRIN inhibitor) AIM2-/- THP1 cells, indicating one or both of these inflammasomes is responsible for the cell death observed in shigella infected THP1 cells. Why not test these separately to determine which?

      Response: We have now made AIM2/MEFV–/– THP-1 cells. Our preliminary finding is that cell death and IL-1B levels in these cells are impaired in response to Shigella infection. We intend to include these data in a revised manuscript.

      2) Markers of inflammation during disease. Clinical features of the disease (diarrhoea, weight, CFU/organ, fecal blood) are described well. But since Shigellosis is an inflammatory disease, it would have been nice to have seen some inflammatory molecules/cytokine levels measured, in addition to clinical features. The authors did measure levels of MPO, but that was as a marker for neutrophil recruitment.

      Response: We agree that additional readouts of inflammatory disease are warranted. We are planning to repeat our experiments and measure cytokines in the blood. We intend to provide these data in a revised manuscript.

      3) Further refinement of the mouse model. The authors present the inhibition of human NAIP/NLRC4 as the main factor that affects the difference in infection between humans and mice but a high innolcum (5 x 10(7) cfu/mouse compared to approx. 100 cfu for humans) is still required in addition to streptomycin treatment. It is not discussed whether any refinement of these procedures was attempted or why such a high inoculum and streptomycin treatment is still required. Presumably microbiota differences in addition to naip-/nlrc4 is an important species specific determinant of infection, hence the streptomycin treatment. Why is such a high innoculum required?

      Response: this comment is similar to one of the comments of Reviewer 1. As we state above, it is actually not entirely clear that the infectious dose for humans is consistently ~100 CFU. Indeed, there appears to be great variation, with some humans exhibiting resistance to doses more than 10^5 CFU. Although we used high inoculums in our experiments, this was just to ensure consistent infection of all mice. Preliminary experiments in which we reduce the dose suggests that, like some humans, some mice are also susceptible to lower doses (e.g., 10^5 CFU). Thus our model exhibits an infectious dose within the range of what is observed in humans and we do not feel there is a large discrepancy here, though it appears that we do not recapitulate the extreme susceptibility seen in some humans. We don’t find this particularly surprising as Shigella is a human-specific pathogen and it is likely that at least some of its virulence factors may not work well in mice. Instead, we think what is most surprising is that loss of one host defense component (NLRC4) is sufficient to produce disease symptoms that are strikingly similar to what is seen in humans. We acknowledge that one difference is the need for streptomycin in our model. Clearly this suggests, as the reviewer states, that the microbiota can influence susceptibility. This is a well-described phenomenon with many enteric pathogens and it will be of interest in future studies to determine what components of the microbiota afford protection in our model.

    1. I do not pretend, in giving you the history of this Royal Slave, to entertain my reader with adventures of a feigned hero, whose life and fortunes fancy may manage at the poet’s pleasure;

      Good for her. I'm wondering if she had to write this note because she's a woman. As we know, men thought women were emotional nutcases, so she probably thought "I gotta explain that I'm telling the truth." If she didn't, men would think her uterus traveled to her head again!

    1. Note: This rebuttal was posted by the corresponding author to Review Commons. Content has not been altered except for formatting.

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      Reply to the reviewers

      INITIAL RESPONSE TO REVIEWERS / REVISION PLAN

      We are grateful to the three reviewers for reviewing our manuscript and providing their comments which helped to improve further the quality of the current study. We attach an initial revised version of the manuscript with changes corresponding to reviewers’ comments being highlighted. We now provide:

      • 18 new main figure panels (Fig.1E, Figs.2D-F, Figs.3E-F, Figs.4B,C,E, Figs.6B-F, Figs.7B,D,E,F),
      • 9 new supplementary figures, and
      • 13 new supplementary tables, that correspond to the points raised by the reviewers. In this initial response to reviewers and revision plan we have already performed the bioinformatics analysis and the majority of new wet lab experiments requested by the reviewers, while we are still awaiting only for the results of three sets of wet lab experiments (RIP-seq, additional protein/RT-qPCR confirmations and B2 incubations with other proteins), which, due to their nature, take longer. We have also revised the main text accordingly with only a number of updates (regarding some methods of experiments currently in progress and the respective discussion) still missing.

      In detail:

      REVIEWER 1

      Reviewer #1 (Evidence, reproducibility and clarity (Required)):

      B2 RNAs, encoded from SINE B2 elements has been directly implicated in stress response by its inherent ability to bind RNA Pol II and suppress stress response genes (SRG) in homeostatic conditions. However, upon stimuli, B2 RNAs are cleaved and degraded, resulting in the release of RNA pol II and upregulation of SRGs. Previous work from the senior author identified PRC2 component EZH2 to be the B2 RNA processing factor, cleaving B2, and releasing POL2. SRGs are upregulated upon stress, for example in age-associated neuropathologies like Alzheimer's disease (AD). Considering that the hippocampus is a primary target of amyloid pathologies as well as since SRGs are suggested to be key for the function of a healthy hippocampus, the authors set to understand the role of B2 RNAs that are linked to SRG regulation in the mouse hippocampus with amyloid pathology. They use disease-relevant in vivo and in vitro models combined with unbiased RNA seq data analysis for this endeavor, which indicates the potential relevance of B2 RNAs in APP mediated neuronal pathologies in mice as well as identifies Hsf1 as the factor cleaving B2 RNAs in the hippocampus.

      This reviewer generally remarks that “The work is interesting and identification of Hsf1 as the processing factor for B2 RNAs in the hippocampus is significant. I would like to credit the authors for their elegant in vivo experimental design in Figure 2.”

      We appreciate the encouraging comments made by this reviewer.

      General comment: The reviewer finds “some of the conclusions to be overstated” and has brought a number of concerns to our attention. Indeed, we agree that provision of additional data and details is needed to avoid any confusion about the gene pathways to which our findings apply. In the initial manuscript, (Figures 2 D, F and 6 D, F), we presented the gene expression levels of all B2 RNA regulated SRGs identified in our previous study (Zovoilis et al, Cell 2016), referred as B2 RNA regulated SRGs or B2-SRGs throughout the manuscript. To this end, we performed the respective statistical tests between the different conditions considering these genes, in order to show the transcription dynamics of these genes in either amyloid beta pathology (APP mice /Figs. 2D, F) or amyloid beta toxicity (HT22 cells / Figs. 6D, F). Since we were not looking for new candidate genes upregulated in APP mice or in our HT22 cell culture system, we did not narrow our analysis only to genes delivered by a general-purpose differential gene expression approach such as DESeq but tested all B2-SRGs. However, based on the reviewer’s comments below, we realize that the paper would benefit by presenting in the main figures only those B2 RNA regulated SRGs that overlap with differentially expressed genes identified by DEseq in each experimental system. This will help to avoid confusion and any misunderstanding that all B2 RNA regulated genes are equally affected in our system, which is not the case and would be an overstatement. We are now presenting in new Figure 2 (2E, 2F) only those B2-SRGs that overlap with upregulated genes identified by DESeq in 6m old APP mice (listed in new Suppl. Table 5) and in new Figure 7 (7D, F) we are now presenting only those B2-SRGs that overlap with upregulated genes identified by DESeq in HT22 cells treated with amyloid beta (listed in new Suppl. Table 11). The conclusions drawn by the new figures remain the same as with the old ones and we believe that this new way of presentation of this data will prevent confusion and potential over-statements. We thank the reviewer for bringing this to our attention. Based also on this reviewer’s minor point 3, we recommend that the old figures that included all B2-SRGs (and not only the differentially expressed ones identified by DESeq) are moved to the Supplement as new Supplementary Figures 1 and 7, respectively, so that readers can still get a view of all the data and the transcription dynamics of all B2-SRGs, while we provide both in text and the supplement an explanation about the value as well as limitations of these figures.

      **Major comments:**

      Major point 1. The reviewer asks: “In figure 1, the authors indicate a strong connection between B2 RNA regulated SRGs and learning and memory. In figure 2, they identify the SRGs in the hippocampus, please provide a direct comparison of learning and memory associated SRGs and the SRGs they identify in figure 2 that are significantly upregulated in APP mice in 6 months.”

      In the revised version of the manuscript we now provide: i) As a new figure panel (lower panel in new Fig.1E), the number of B2 RNA regulated SRGs that are associated with learning based on our Peleg et al, Science 2010 paper and as a new Supplementary Table 3, the exact list of these genes. ii) As a new Supplementary Table 4, the list of all genes that are significantly upregulated in APP mice (6 months). iii) As a new Supplementary Table 5, the list of those genes upregulated in amyloid pathology (APP 6 months) that are B2-SRGs (expression levels of these genes are presented in new Figure 2E,F). Per reviewer’s question, we now provide as a new Supplementary Table 6, the list of B2 RNA regulated SRGs that are both learning associated genes and upregulated in 6 month old APP mice. In the text (first two sections of the results), we provide direct comparisons of the number of genes in each category and their overlap.

      Major point 2. The reviewer asks: “To better understand the data in the context of hippocampal function, please include functional annotation of SRGs they identified in Figure 2F as they do it in Figure 1 (desirably for each time point, at least for 6M). How many of the SRGs they identify in Figure 1 are part of Figure 2F? Please include functional annotation of significantly upregulated B2 regulated SRGs in Fig2 and compare them with that of Figure 1.”

      The number of B2 RNA regulated SRGs in Figure 1 that are part of Figure 2 (in particular Figs.2E,F) is now presented in the new Supplementary Table 5 and also in the text. We now provide as a new Supplementary Table 7 the functional annotation of these genes (see also general comment for this reviewer) and discuss the findings in the text.

      We recommend to include only the 6M old mice as this is the time point in which B2 RNA processing was found to differ between WT and APP mice. However, if the reviewer thinks that this is necessary we will add also differential expression lists of other ages as additional supplementary tables.

      Major point 3. The reviewer asks: “In figure 3, the authors report that the B2 processing rates are high at the 6M time point at in hippocampi of the APP mice. Please include the levels of unprocessed and processed B2 RNAs in these samples along with this figure, without which it is difficult to gauge the significance of its correlation with SRGs in Figure 2.”

      We now provide as new figure panels 3E and 3F the levels of processed B2 RNA fragments and unprocessed (full length) B2 RNAs in these samples, respectively, along with the processing ratio which is now labeled as subfigure 3G.

      Major point 4. The reviewer asks: “What is the % of B2 regulated SRGs that are hsf1 bound in Figure 4C? What is there dynamics in the wild type and APP hippocampi?”.

      Old Figure 4C is now Figure 4A. The exact number of B2 RNA regulated SRGs that are close to Hsf1 binding sites is now presented as a new figure (Figure 4C) and discussed in the text. A list of these genes is provided as new Supplementary Table 8. For genes that are upregulated in APP mice compared to wild type, the difference in Hsf1 binding dynamics between B2 RNA regulated and not regulated genes is now presented as Suppl. Figure 4D.

      Major point 5. The reviewer asks: “What is the distribution of Hsf1 binding sites on (a) non-B2 regulated SRGs and (b) non-SRG genes in hippocampi?”.

      This point is related with point 4. We now present a new panel (Fig. 4B) for non B2 RNA regulated genes (listed in Suppl. Table 13) along with the distribution we have in the initial manuscript for all B2 RNA regulated SRGs (now presented as Fig. 4A). The direct comparison of these genes is presented in the new Suppl Figure 4C together with a similar comparison only for genes upregulated in APP mice (Suppl. Fig.4D)

      Major point 6. The reviewer notes: “In Figure 4D, the 3months old Wt HSF1 levels are high, yet B2 processing (Figure 3E) is low. Please comment.”

      The reviewer’s comment made us realize that we should include a plot that describes the correlation between Hsf1 levels and B2 RNA processing ration across all sequenced samples. This should reveal whether differences such as those observed by the reviewer affect our conclusion regarding the relationship between these two parameters. We now provide this in the new Supplementary Figure 6D, where we found a strong positive correlation between Hsf1 levels and B2 RNA processing ratio. We thank the reviewer for this comment which helped us to substantiate further this relationship.

      Major point 7. The reviewer notes: While the authors show in vitro cleavage of B2 RNA by Hsf1, the experiment lacks controls to be conclusive. At least, please include a similar size protein as HSF1 with no-known RNA binding activity and a similar size protein with RNA binding activity as controls in 5A. Please justify the use of PNK as the control protein. Please include the use domain-based deletions of Hsf1 to map the region of HSF1 that is binding and potentially cleaving the B2 RNA. Please include an RNA of similar size and Antisense-B2 RNA to show the specificity of the Hsf1 based cleavage of B2 RNA. Without these controls, the conclusions in Figure 5 cannot be substantiated.

      The endogenous ribozyme activity of B2 RNA compared to other control RNAs has already been shown in two previous works but we will also include the relative controls here by providing control incubations with other RNAs. We will also include the incubations with additional control proteins as suggested by the reviewer. We are currently performing these experiments and will include them in the revised version. PNK is used as a control protein because it is an RNA binding protein that is used in the construction of our short RNA libraries and we wanted show that short RNA seq data are free of such confounding factors that could potentially generate artificial fragments. We now include this information in the text.

      We feel that the application of domain based deletions for Hsf1, while it would add additional information on the exact biochemistry underlying B2 RNA processing though Hsf1, is beyond the scope of this manuscript. In the current manuscript we are just focusing on the fact that Hsf1 can accelerate B2 RNA processing in vitro and not on the mechanism how this happens. This should be addressed in our opinion on a separate manuscript.

      Major point 8. The reviewer asks: “The authors should show that the incubated APP peptides are taken up by the cells (experiments in Figure 5F and Figure 6).” These figures are now labelled as Fig.6C and Figure 7, respectively. That’s a very interesting point and we thank the reviewer for this comment. Multiple studies have shown that toxicity after incubation by amyloid beta is mediated mainly by cell surface receptors, which through cell signalling leads to the response to cellular toxicity that induces stress genes such as Hsf1. Nevertheless, APP peptides may enter the cell, and the reviewer’s questions raised the possibility that oligomers entering the cell could have a direct impact on the stability of the B2 RNA. In that case, providing evidence that the amyloid enters the cell would be important if we had indications that amyloid beta interacts directly with B2 RNA. We did test this and we found no direct effect of amyloid beta on B2 RNA, so the processing in our case is not induced by oligomers that may have entered the cell. We were planning to present this information in a different manuscript, but if the reviewer or editor thinks that it would be beneficial for the paper, we could present this as supplement figure that shows that amyloid beta incubations with B2 RNA do not induce further processing beyond what Hsf1 causes. For the moment we just present this below:

      Major point 9. The reviewer asks: “Please provide the list, functional annotation, and % of the SRGs upregulated upon incubation with APP in HT22 cells in comparison to 6month old APP mice. Comment on learning-related Genes.”

      In the revised version, we now provide and mention in the text the following data: i) a list of genes upregulated in HT22 cells during amyloid toxicity upon incubation with amyloid beta (new Suppl. Table 9), ii) a list of genes according to point (i) that are common with genes upregulated in APP mice (new Suppl. Table 10), iii) the list and number of B2-SRGs that are upregulated in HT22 cells during amyloid toxicity (the reviewer’s question) (new Suppl. Table 10). We mention in the text the gene numbers and also the genes that are common in all three lists. iv) Functional annotation of genes of point (iii) (new Suppl. Table 12),

      We also mention in the text the limitations of our comparisons between the in vivo model of amyloid pathology (APP mice) and the in vitro cell culture model of amyloid toxicity (HT 22 cells) and we clarify that the cell culture model is used just as a simulation of the effect of amyloid beta in gene pathways associated with response to cellular stress and the role of Hsf1 on B2 RNA processing.

      Major point 10. The reviewer asks: “The authors should show the efficient downregulation of Hsf1 (protein) upon anti-Hsf1 LNA transfection.”

      In the revised version, in addition to the RNA-seq data we provide a second confirmation at the mRNA level with an independent method (RT-qPCR) in new figures 4E and 7B (lower panel). We are currently performing the protein extractions and will provide a WB or an Elisa in the revised version.

      Major point 11. The reviewer asks: “Please present the total B2 RNA levels for conditions in Figure 6C.”

      We now provide as new supplementary figure (Suppl. Fig. 6B and C) the levels of processed B2 RNA fragments and the total levels of unprocessed full length B2 RNAs of these samples that relate to old Figure 6C (now labeled as Fig.7C)

      Major point 12. The reviewer notes: “Hsf1 levels are not significantly downregulated in Control cells which were inoculated with the reverse APP peptide. Please comment.”

      We assume that the reviewer here refers to the lack of reduction in Hsf1 levels in the cells inoculated with the reverse peptide and the anti-Hsf1 LNA. Indeed, this lack of reduction is confirmed also by the new qPCR we performed (new Figure 7B, lower panel, R-ctrl vs R-anti-Hsf1). This should likely be attributed to compensation during non-stress conditions. In contrast, under stress conditions, Hsf1 is heavily used in stress response, which could explain the differences we see as cellular needs surpass the available Hsf1 transcripts due to degradation by the LNA. This is also supported by the new RT-qPCR experiments we have performed for B2-SRGs (new Figure 7E). In agreement with what is known for stress response genes such as immediately early genes (for example FosB), levels of these genes are minimal in both R-ctrl and R-anti-Hsf1 conditions and only become activated during stress response. We now discuss this in the text of the revised manuscript.

      Major point 13. The reviewer asks: “Please compare and contrast the % of genes, the overlap, and the functional distinctions in 6F to that of 5G and Figure1. What are the genes that are common between Figure1, and that are specifically upregulated upon Anti-Hsf1 LNA transfection along with 1-42 APP. What is % of the occurrence of B2 binding sites in those genes? What are their functional annotations and what is their connection to learning, memory, and cell survival?”

      Old Figure 6F is now Figure 7F, while old Figure 5G is now Figure 6C. This point is discussed in the response to points 1 and 9 of this reviewer. In summary, genes upregulated in our amyloid toxicity model included 25 B2-SRGs (new Suppl. Table 11). When testing for enriched terms in these 25 genes, biological processes related with apoptosis, such as regulation of apoptotic process and programmed cell death were at the top of the list (new Suppl. Table 12) and included, among others, genes such as FosB and Mitf that have been connected with Alzheimer’s disease. Out of the 25 genes that are up-regulated in both mice and our cell culture system, six are B2-SRGs (4932438A13Rik, Fosb, Pag1, Ptprs, Sema5a, and Sgms1) and include a well-known immediate early gene (Fosb), genes associated with sensitivity to amyloid toxicity (Pag1, Sema5a, Sgms1, Fosb), as well as genes associated with p53 (Ptprs, Fosb). All these genes get upregulated in amyloid toxicity (42-Ctrl vs R-Ctrl) but are not upregulated when Hsf1 LNA is applied (42-anti-Hsf1 vs R-anti-Hsf1, no significant difference). This information is now included in the text.

      **Minor.**

      1 . Please include TPM/ FPKM values for hippocampal markers as control in Figure 2 to do justice to the hippocampus specific RNA seq conducted by the Authors.

      To our understanding, the reviewer here suggests the testing of well-known hippocampal markers in our mouse data as controls to confirm that they are indeed hippocampus specific. We have selected as reference markers, the genes employed by the Allen Brain Atlas RNA-sequencing project and we provide a comparison of their data in hippocampal cells with our data from mouse hippocampus. This is now presented as new Supplementary Figure 2.

      2 . In figure 2D the authors show that B2 RNA regulated SRGs in the 3 months' wild type mice are significantly high. P53 has been reported to be high in young wild types hippocampus, but not SRGs in my opinion. The authors should comment on this.

      Old Figure 2D is now Figure 2E. We now mention the reviewer’s comment particularly in the discussion and cite a landmark review article in Neuron journal by Michael Greenberg regarding the role of stress response genes, such as FosB, early during development. As to prevent any confusion, we have also replaced SRGs with B2-SRGs since we tested only B2-SRGS in our study.

      3 . In figure 2F, under the 6m APP condition, the replicate 3 looks substantially different from the other replicate. This can significantly impact the analysis and conclusions made. Either remove that replicate and present the analysis without it or please provide a valid explanation. To make the data more valid, please provide hierarchical clustering of the entire data, the non-B2 regulated genes and the B2 regulated SRGs.

      We now provide in the new Supplementary Figure 9C a PCA plot, which includes 6m APP mice vs. their WT counterparts and HT22 cells, and shows that this variability is within the biological replicate variability we can expect in these models. To substantiate this further, we have constructed the correlation matrix of the RNA-seq data of both WT and APP 6 month old mice in the new Supplementary Figure 9D. As shown in this matrix, all APP mice clearly correlate with each other and not with their WT counterparts.

      In the initial manuscript the heatmaps of former Figure 2 were indeed provided with hierarchical clustering of the entire data and also included non-B2 RNA regulated genes. This data is included now as Supplementary figure 2.

      In Figure 2C RNA seq data is represented in TPM while its FPKM in Figure 2D.

      Figure 2D is now Figure 2E, while Figure 2C remains labelled with the same number. Given that TPM already includes scaling of the data, it is unsuitable for the averaging of the gene expression levels of multiple genes (B2-SRGs) used in the boxplots of Figure 2. This does not apply in the case of single genes as in Fig 2C (p53) or in the heatmap where each gene is presented in a separate row. This explanation is now included in the methods section.

      Figure 2: the number of replicates in the case of 3-month-old wild types only 2. Please specifically denote it and comment why only 2 replicates are provided.

      During the hippocampal RNA extractions, the RNA of one of the three 3m old mice had very low RIN scores, which could be a confounding factor for the short-RNA-seq. As this happened some months after the hippocampal extractions, we did not have any other 3 month mice of the same cohort used for the behavioral and IHC studies. Thus, we decided to include only two replicates in this condition. Since the results presented in the current study focus mainly on 6 month old mice, we expect the impact to be minimal. We include this note in the methods section.

      4 . Considering that p53 and SRGs are significantly upregulated in 6months in the APP model, it would be great if (allowing that these samples are still available) the authors can include a staining for apoptotic markers, for example, Active Casp3 or similar. This will allow us to better gauge the gene expression changes presented by the authors especially regarding SRGs.

      Unfortunately, we do not have these slides but in the revised version we will provide qPCR data for some of these markers.

      5 . Under subheading: Hsf1 accelerates B2 RNA processing, 3rd paragraph when the authors comment on known hsf1 binding sites on SRG genes, please correct from: Increased Hsf1-binding was found.... "To the increased number of hsf1 binding sites were found", unless the authors would like to show increased Hsf1 binding by performing CHIP-seq for Hsf1 in the hippocampus at least at the 6-month time point between Wt and APP mice.

      We have changed the text accordingly.

      Reviewer #1 (Significance (Required)):

      B2 RNAs, encoded from SINE B2 elements has been directly implicated in stress response by its inherent ability to bind RNA Pol II and suppress stress response genes (SRG) in homeostatic conditions. However, upon stimuli, B2 RNAs are cleaved and degraded, resulting in the release of RNA pol II and upregulation of SRGs. Previous work from the senior author identified PRC2 component EZH2 to be the B2 RNA processing factor, cleaving B2, and releasing POL2. SRGs are upregulated upon stress, for example in age-associated neuropathologies like Alzheimer's disease (AD). Considering that the hippocampus is a primary target of amyloid pathologies as well as since SRGs are suggested to be key for the function of a healthy hippocampus, the authors set to understand the role of B2 RNAs that are linked to SRG regulation in the mouse hippocampus with amyloid pathology. They use disease-relevant in vivo and in vitro models combined with unbiased RNA seq data analysis for this endeavor, which indicates the potential relevance of B2 RNAs in APP mediated neuronal pathologies in mice as well as identifies Hsf1 as the factor cleaving B2 RNAs in the hippocampus.

      The work is interesting and identification of Hsf1 as the processing factor for B2 RNAs in the hippocampus is significant. I would like to credit the authors for their elegant in vivo experimental design in Figure 2.

      REVIEWER 2

      Reviewer #2 (Evidence, reproducibility and clarity (Required)):

      **Summary:**

      This manuscript follows from previous work by the corresponding author showing that SINE-encoded B2 RNAs function as regulators of the expression of stress response genes (SRGs). Specifically, stimulus triggers the processing of repressive B2 RNAs that are bound at the SRGs, thereby activating SRG transcription. In this work, the authors investigate whether a similar mechanism might be controlling the expression of genes in models of amyloid beta neuropathology (i.e. mouse hippocampi from an amyloid precursor protein knock-in mouse model, and a cell culture model of amyloid beta toxicity). They performed RNA-seq in these models. Their data show a correlation between the progression of amyloid pathology, expression of genes thought to be regulated by B2 RNA, and the processing of B2 RNA. In addition, they show biochemical data supporting a role for Hsf1 in enhancing the processing of B2 RNA. Knockdown of Hsf1 also reduced B2 RNA processing and the expression of SRGs.

      **Major comments:**

      Major point 1. The reviewer asks: “In the RNA-seq data one cannot distinguish between Pol III transcribed B2 RNA and Pol II transcribed B2 RNA (typically embedded within introns and UTRs of mRNAs). The models they present, and the structures they show, clearly imply regulation by Pol III transcribed B2 RNA. However, there is no way to know that the short B2 RNAs they sequence aren't coming from degraded mRNAs. This needs to addressed. Minimally, in writing as a caveat of their model. Ideally, it would be addressed experimentally.”

      That’s a very interesting point, as it implies that the regulatory role of B2 RNAs may extend from PolIII transcribed B2 RNAs into B2 RNAs embedded into mRNAs (likely nascent ones) that may be also under the same endogenous ribozyme activity of this sequence, suppress PolII and are processed in response to stimuli. The RNA RIN values of our samples were pretty high except one 3m old mouse sample which was for this reason excluded from further analysis. Moreover, during the library construction shorter and longer RNAs have been separated. Thus, any generation of B2 RNA fragment that may have originated from mRNA should be biologically but not technically related and must have happened in the cell before our RNA extraction. To address this point, we now provide a new supplementary figure (Suppl. Figure 8), where we have separated the B2 elements against which we map the RNA fragments into two categories, those that fall within exonic/genic regions and those outside of these regions. Although B2 RNAs are produced by multiple copies in the genome, each copy does harbor multiple SNPs, insertions and deletions, which means that each B2 RNA fragment is mapped to a specific set of B2 elements and not to all of them. In other words, despite multiple mapping a level of spatial specificity is maintained. If the B2 RNAs we map were coming exclusively from either only Pol III B2 elements or mRNA embedded B2 elements, we would expect at least some difference in the distribution of fragments between B2 elements of these two categories, as the second one overlaps with mRNAs. As shown in the new supplementary figure 8, the fact that distribution models are very similar between the two categories indeed supports the hypothesis that both types of B2 elements may contribute to B2 RNA processing. Most importantly, the profile of B2 RNAs in genic regions shows that B2 RNA processing is not random but follows the same processing rules as B2 RNAs from Pol III promoters. Given the limitations posed by the repetitive nature of B2 RNAs, it remains difficult though to provide an exact number regarding the portion of B2 RNA fragments produced by each category and this is clearly noted in our revised discussion part. However, even the indication that B2 RNAs embedded in mRNAs may also play an important role in our model provides a new perspective that should be investigated further in future studies.

      Major point 2. The reviewer asks: “The direct regulation of SRGs by B2 RNA was not shown in their model systems for amyloid beta neuropathology. Rather, the authors' used the genes identified in their prior studies as B2 RNA-regulated, which I believe were in the NIH3T3 cell line. Given that transcription is highly cell-type specific, these genes might not be regulated by B2 RNA in mouse hippocampi or their cell culture model, despite the correlations shown. This needs to be addressed. Ideally, a targeted approach to show that transcription of even a couple genes in their system is indeed regulated by B2 RNA would provide stronger support for their conclusions.”

      We agree with the reviewer and we now provide a new figure (Fig.6D-F) with the targeted approach that this reviewer proposed. In particular, we have tested whether fragmentation of full length B2 RNAs is in connection with activation of target genes also in our biological system (HT22 cells) as it did in NIH/3T3 cells in our Cell paper. We now show in new Figure 6 that this is indeed the case.

      Major point 3. The reviewer proposes a number of additional information that needs to be provided: “The following bioinformatics analyses would strengthen their conclusions. This should be straightforward to do because it involves data they already have, and perhaps analyses they have already have performed.”

      a. Regarding the plot in Figure 3A (lower panel). The same plot should be shown for the 3m old and the 12m old APP mice (i.e. not just the 6m data). This would show the specificity of processing B2 RNA and that it indeed correlates with disease progression.

      We now provide this plot as new supplementary figure (Suppl. Figure 3). It shows that increased B2 RNA processing coincides only with the active neurodegeneration phase at 6 months and not the terminal stage.

      b. Regarding the plots of B2 RNA processing rate. This value could increase either due to more short RNAs or less full length RNA. Which is it for the 3m, 6m, and 12m APP mice? Showing the short and long B2 RNAs as boxplots (as opposed to only the processing rate) would address this and also provide additional insight into the regulation involved. The same applies to the data in Figure 6. (As an aside... do the authors mean processing ratio as opposed to rate? I'm not clear where the time component is coming into play to call this a rate.)

      Old Figure 6 is now Figure 7. We now provide all these figures that show that increase in processing ratio at 6 months is mainly due to increase in the processed fragments and not a decrease in full length B2 RNAs. For APP mice these are new Figures 3E and F, and for HT22 cells , these are new Supp. Figures 6B and C.

      c. The random genes in Figures 2E and 6E are plotted as heat maps, but statistical significance is hard to see. What do boxplots of the random genes look like, and is the significant difference between 6m old APP and 6m old WT then lost?

      Old Figure 2E is now new Suppl. Figure 1C, while old Figure 6E is now new Suppl. Figure 7C. We now provide these boxplots in new supplementary figures 1B and 7B.

      Major point 4. The reviewer comments: “ It is interesting that B2 RNA self-processing is enhanced by both Ezh2 and also Hsf1. It would strengthen the data to perform a control with a protein prepared more similarly to the Hsf1 (rather than PNK) to confirm that the enhanced B2 RNA breakdown is indeed attributable to Hsf1 and not a contaminant in the protein prep. Similarly, the authors should provide information on which RNA was added as the negative control for Hsf1-stimulated breakdown (i.e. the ~80 nt RNA).”

      This point is also discussed in Reviewer 1 point 7. The ribozyme endogenous activity of B2 RNA has been shown already in two previous studies that performed incubations with control RNAs and proteins. We are currently preparing and will provide these additional incubations as anew supplementary figure in the revised manuscript.

      **Minor comments:**

      1 . Regarding the GO analyses in Figure 1 (panels B, C, and D). I wasn't clear whether the authors are showing all statistically enriched terms, or only those relevant to neuronal processes and learning. I recommend showing a supplemental table with all terms that have an adjusted p value below a specified cut-off (e.g. 0.05).

      The statistical threshold used was an EASE score of 0.05 and all presented terms were above this threshold. In the initial manuscript we filtered only the top 5 terms in tissue enrichment and the top 10 terms for GO Biol process and Cell Compartment that had passed the threshold. We now provide all the terms that passed the threshold as a new Supplementary Table 2, including gene counts, exact gene numbers and related statistics.

      2 . The authors show several figures that are not new data (2B, 4A, 4B, Suppl. Fig 1 and 2). I think it would be more clear if these data were summarized and referenced in the results, rather than shown.

      Old Suppl. Fig1 and 2 that were results of previous studies or web resources directly available (such as Human Protein Atlas) have been now removed and they are now just referenced in the text. Old Figures 4A and 4B have been removed from the main figures but may be helpful to the readers if they are still available in the Supplement (currently as Suppl. Figure 4A and B), as not all users are familiar with the RNA-seq browsing tools of Allen Brain Atlas resources. Regarding figure 2B that contains data from our previous study on this exact cohort of mice: If the reviewer and the editor agree we recommend that it remains in the main figure (with the appropriate image credit citations), as it provides in an efficient way the clear connection between amyloid load and our results at the molecular level, and, most importantly, it clearly draws a line in amyloid pathology progression between 3m old and 6m old, that agrees with our findings in the RNA-seq data of these mice.

      3 . In Figure 3A the schematic shows that B2 is 155 nt, the plots in Figures 3A,B,C show B2 RNA is 120 nt, and Figure 5 shows the RNA is 188 nt. Can the authors please clarify these differences?

      The full length of B2 consensus sequence is 188nt and this is the one we use for the in vitro experiments. However, the structure of the B2 RNA has been resolved only for the first 155nt by the Kugel lab, and this is the only publicly available structure that we can reference in our figures. For the mapping of 5’ends of short fragments in Fig.3A we have used the same range tested in our Cell paper to maintain consistency of the results. The reason why this 120nt threshold was selected in the Cell paper was to exclude artifacts from short RNAs mapping partially in our metagene as well as downstream of those B2 elements that are shorter from the consensus sequence. We now explain in methods section these differences.

      4 . In the Methods section, the sequence of the g block template didn't contain the T7 promoter sequence that was used as the forward primer for PCR amplification?

      We have now included this sequence in lower case.

      5 . In Figure 6B, why were Hsf1 levels not decreased in the R treated cells after treatment with the LNA?

      Old Figure 6B is now new Figure 7B. Please see response to Reviewer 1, major point 12.

      Reviewer #2 (Significance (Required)):

      Finally, this reviewer generally remarks that “The models presented for the regulation of stress response genes (SRGs) in amyloid beta neuropathologies are compelling. As are the correlations they found between the progression of amyloid pathology, expression of genes thought to be regulated by B2 RNA, and the processing of B2 RNA. This is a unique direction of research for brain disease and represents an interesting conceptual advance. Most prior studies in this area use common model cell lines, and this lab seems well-positioned to unravel the proposed molecular mechanisms in neuronal systems.”

      We appreciate the encouraging comments made by this reviewer.

      REVIEWER 3

      Reviewer #3 (Evidence, reproducibility and clarity (Required)):

      This manuscript describes a regulatory mechanism involving Hsf1 and B2 RNAs in the control of stress response genes (SRGs) during amyloid induced toxicity. In particular Hsf1, upregulated in 6m old APP mice and in HT22 cells treated with beta amyloid peptides, is shown to stimulate the B2 RNA destabilization leading to SRGs activation. While in healthy cells this upregulation can be reverted once the stimulus is removed, the pathological condition fuels the circuitry leading to p53 upregulation and neuronal cell death. The authors previously described the same mechanism acting during cellular heath shock response but in this case the protein identified as trigger of B2 RNA destabilization and SRGs activation was EZH2 (Zovoilis et al, 2016).

      This reviewer generally remarks that “Indeed, the first part of the manuscript describes additional analyses of the previous data that prompts further investigation on the potential role of B2 RNA in AD condition. Nevertheless, it is not clear how the prior findings obtained in not biologically related cellular models might be used to obtain helpful indication of B2 RNA neuronal activity.”

      We thank the reviewer for this comment. Indeed, the current study’s main aim was to expand the findings of our previous work on the role of B2 RNA in cellular response to thermal stress in NIH/3T3 cells to other types of cellular response to stress, in our case to amyloid toxicity and the resulting amyloid pathology in neural cells. Response to thermal stress (Heat Shock) has been used for years as a basic study model for cellular response to stress. Proteins and gene pathways initially identified in heat shock have been subsequently shown to play identical pro-survival roles in other biological systems and there are studies showing the role of Hsf1, heat shock related proteins and cell stress response pathways in neural cells and the mammalian brain (we will provide these references in the revised version). For example, pathways such as the MAPK pathway and early response genes, that constitute the basis of response to heat shock, have been shown in studies by us and others to be activated and play a critical role in hippocampal function. Thus, examining the role of B2 RNA in the context of neural response to stress constituted a natural continuation of our previous study in NIH/3T3 cells. The fact that the list of B2 RNA regulated SRGs was found to be highly enriched in neuronal tissue terms and cellular compartments related to neuronal functions plainly confirms the close relationship among cellular response pathways in the two biological systems. Due to these facts we were compelled to investigate in more detail our previous findings also in a neural cell model. However, as discussed in point 2 of Reviewer 2, the initial manuscript did not confirm the direct control of B2 RNA on expression of target genes also in our cellular model. This information is now part of the new figure 6 and we thank both reviewers for bringing this to our attention.

      The reviewer also remarks that “The research fields of non coding RNAs and neurodegeneration are attractive and challenging and, in my opinion, the molecular circuitry involving B2 RNAs might add important insights for understanding beta amyloid toxicity and neuronal death; however, the data provided are not in the shape making the manuscript suitable for publication: some controls are missing, the way the experiments are presented is not easy to follow and more importantly the authors does not provide any data (tables or lists) of the NGS experiments and the study lacks validation of them. Therefore, in my opinion the manuscript needs a profound revision before to be considered for publication in Review Commons.”

      Based on this reviewer’s and the other reviewers’ suggestions we now provide additional controls, detailed tables and gene lists, and qPCR validation of these results. We have also substantially revised the text in the first section of the results and beginning of the discussion, to make our rational for testing B2-SRGs more clear and easier to follow.

      **major concerns:**

      Major point 1. The reviewer asks: “The first paragraph of the Results is entirely dedicated to re-analyze the data previously published by the same group (Zovoilis et al., 2016). However, this is not adequately explained. In line with this, the table 1 is not required since the data are already provided by Zovoilis et al., 2016, unless the authors handled the data using additional new criteria that have to be explained.”

      We now explain our rational for using this data in more detail in the text. Please see also response to the general comment of this reviewer and response to the next point.

      In the Zovoilis et al (2016) study, the data presented did not include the list of regulated genes in a direct way but as part of the annotation of the B2 CHART peaks. This may pose difficulty to non-experts to extract the gene list from that data and we thought to include them as separate gene list here so that readers can directly use it for their analysis. Nevertheless, if the reviewer or the editor think that the list is redundant, we can surely omit it.

      In addition, the reviewer comments: “Moreover, Zovoilis and colleagues (2016) focused on SRGs regulated upon heat shock and using NIH/3T3 and HeLa cell lines, therefore, it is difficult to me understand how, searching for "cellular function connected with B2 RNA regulated SRGs", the list resulted enriched of neuronal tissue terms or cellular compartments related to neuronal functions. Please clarify this point since the following analyses are based on these findings.”

      Neural pathologies, such as amyloid pathology in brain, are often connected with cellular stress due to proteotoxicity. The ability of neural cells to respond to proteotoxicity challenges is connected with various molecular mechanisms, including stress related proteins that were firstly described in the context of heat shock. Thus, both contexts (heat shock and amyloid toxicity) refer to cellular response to stress, which explains why genes identified to be regulated during stress response in NIH/3T3 cells constitute part of the basic stress response toolbox that neural cells have also been described to possess. We have now modified the text accordingly to make our rational more clear.

      Major point 2. The reviewer comments: “In Figure 1F there is no arrow indicating that some of the SRGs regulate directly miR-34 as stated in the main text. Moreover, it is more appropriate to replace SRGs with learning‐associated genes both in the figure and in text (2nd paragraph of the results) since Zovoilis and colleagues focused on them. Finally, they did not show in their manuscript the rescue of p53 expression mediated by mir-34; indeed, for miR-34-p53 regulatory axis Zovoilis and colleagues referred to Peleg et al, 2010 and Yamakuchi & Lowenstein, 2009. Please fix all these concerns.”

      We have restructured the figure as suggested by the reviewer and made clear the distinction between learning genes and B2 RNA regulated SRGs (B2-SRGs) from the two different studies. In connection with point 1 of Reviewer 1, we believe that new Figure 1E, that includes the exact number of B2-SRGs that are learning associated, will represent more efficiently and accurately the data. We have also corrected in the text the citation regarding miR-34c and p53 in both the introduction and first section of the results (last paragraph).

      -The Fig.1A and Fig.1F are wrongly indicated at the end of the sentence "....levels of these genes are normally downregulated in 6m and 12m old mice compared to 3m old mice (p=0.02 and p=0.04, respectively)"; please correct this point.

      The error has been corrected.

      Major point 3. The reviewer comments regarding Figure 2:

      a) Since three mice for each condition have been used for the RNA seq analyses, please provide a blot with the Principal Component Analysis (PCA).

      Please see also response to minor point 3 of Reviewer 1. We provide the PCA plots for WT and APP mice in the new Supplementary Figure 9 and we also provide a comparison of the six month old mice with the HT cell samples as well as a correlation matrix for 6 month old mice in the same figure.

      b) Fig 2F comes first of Fig 2E in the text, however, I suggest to move this latter to supplementary material.

      Old figure 2E has now been moved to supplementary material as new Supplementary Figure 2C and we also provide in a boxplot the exact gene expression levels as new Supplementary Figure 2B.

      c) In general, this study lacks validation of the RNA-seq results. Western blot and/or qRTR-PCR to verify the variation of p53 and of some selected SRGs have to be provided.

      In the current revised version we already provide qPCRs for p53 and Hsf1 in APP mice and we will include additional genes in the final version.

      d) It is also not clear how the authors defined SRGs in the hippocampus: do they correspond to learning‐associated genes described by in Zovoilis et al, 2011 or to B2 RNA H/S regulated genes by Zovoilis et al, 2016?

      The way we presented B2 RNA SRGs in the results with regard to learning associated genes was indeed unclear. We now present the distinction between the two gene categories and their relationship as a new Fig.1E panel and we also provide detailed gene lists of common genes and the exact numbers (please see also response to Review 1, major point 1).

      -APP 12 month old mice show the sever phenotype of the terminal AD-like pathology, however this does not correlate with significant SRGs and B2 processing increase. Can the author make a comment on this?

      That’s a very important point and we thank the reviewer for raising this point. We now comment on this in the discussion part explaining how our findings are characteristic of the initial active neurodegeneration phase of amyloid pathology rather than more terminal stages.

      Major point 4: The reviewer comments regarding Figure 5:

      a) a gel with no-protein control for the time course of panel B was cited in the text but missing among the panels. Moreover, the time course shown in the graph in 5C does not correspond to the one in 5B.

      Indeed, the no-protein control time line should refer only to panel C and not to B, we have now corrected the text. Nevertheless, we now present in the new Supplementary Fig. 5 the gels, based on which the graph in panel C was calculated, including also the gel with no protein timeline. The time course shown in the initial 5C had been mislabeled. It has now been corrected. We apologize for this and we thank the reviewer for bringing this to our attention.

      b) 5G indicates that four samples for each condition have been analysed by RNA-seq, since they do not seem to be homogeneous please provide a PCA analysis together with the validation by qRT-PCR of a selected group of deregulated genes.

      Old Figure 5G is new Figure 6C. PCA analysis for these samples is now provided in Supplementary Figure 9 and qPCR validation of a number of these genes is provided in new Fig. 7E.

      Moreover, it is not clear whether all the genes shown in the heatmap or a number of them, as stated in the text, were found upregulated in 6m old APP mice. Please clarify this point and modify the figure and the text accordingly. A Venn diagram showing the overlap between genes upregulated in 42vsR treatment and those upregulated in 6m old APP mice might help the comprehension of the experiment.

      Please see response to Reviewer 1, point 9. We now provide as new supplementary tables the exact overlapping lists and mention these numbers in the text.

      Major point 5: The reviewer comments regarding Figure 6 (now labeled as Fig.7):

      a) The evaluation of the levels of Hsf1 mRNA and protein upon LNA transfection is missing for both R and 42 treated HT22 cells. From TPM in panel B, Hsf1 downregulation seems to have been more effective in 42 than in R condition. This would mess up the interpretation of the data.

      We now provide qPCR data for Hsf1 gene expression levels which confirm the ones from the RNAseq. The reason why Hsf1 downregulation seems not to affect the R condition is discussed in our response to Reviewer 1, major point 12, and the respective explanation is provided in the revised text.

      b) Again, in this case any validation of the RNA seq data is provided (any B2 regulated SRGs).

      Now, we provide qPCR data for these genes in Fig.7B and new Fig.7E

      c) Panels E and F should be swapped or panel E moved to supplementary material.

      Panel E is now moved to supplementary material as new Suppl. Figure 7C.

      Major point 6. The reviewer comments: “In a previous paper the authors discovered B2 RNAs as a class of transcripts bound to EZH2 and this interaction leads to B2 RNA destabilization in heath shock (H/S) condition. The authors also conclude that the genes controlled by B2 RNAs may not overlap with the ones controlled by Hsf1 during H/S. The author should make a comment on this explaining why during H/S B2 RNAs work independently from Hsf1 and on different target SRGs while, during beta amyloid stress ,the two act together on the same SRGs. Moreover, as shown for EZH2, Hsf1-RIP experiment should be performed in order to confirm the direct involvement of Hsf1 in the SRGs-B2 destabilization.”

      In the last two paragraphs of our discussion we indicate that B2 RNA regulation is a new process implicated in the response to stress in amyloid pathology but certainly not the only one. We have revised the text in this part accordingly in the revised version to prevent any confusion. We are currently performing a series of RIP-seq experiments with various antibodies. As, to our knowledge, there is no prior published study performing RIP-seq or CLIP-seq for any tissue using Hsf1 antibodies, the success of this experiment is not guaranteed and depends on the existence of appropriate antibodies.

      Major point 7. The reviewer comments: “There is any table listing the results of the RNA seq experiments performed in this paper: control vs APP 3-6-12 m old mice and in R vs 42 treated HT22 cells in presence or absence of LNA against Hsf1. Please provide these data.”

      We now provide these lists as new supplementary tables. Please see response to major points 1 and 9 of reviewer 1.

      Major point 8. The reviewer comments: “In the discussion the authors claim that healthy cells are able to restore the expression of Hsf1, SRGs and B2 RNA upon removal of the stress. Since there are evidence for the rescue of SRGs and B2 RNA expression post H/S, no data are available for Hsf1, SRGs and B2 RNA upon the removal of 1-42 beta amyloid peptide. This might be a nice information to add to the manuscript.”

      This would indeed substantiate further our results in our HT22 cell model. We have now performed this experiment, in which HT-22 cells were removed from the amyloid 42 (and the respective R peptide control) and left to recover for 12 hours before estimating through RT-qPCR the Hsf1 levels ( see graph below, REC corresponds to recovered HT-22 cells). Hsf1 levels in 42-REC have returned to the same levels as in R, p We currently perform the RT-qPCRs of these samples also for B2-SRGs and will include them in the final version as a supplementary figure.

      **Minor criticisms:**

      -In the introduction the reference Yamakuchi M and Lowenstein CJ, (2009) MiR‐34, SIRT1 and p53: the feedback loop. Cell Cycle, should be added in the sentence: "In contrast, hippocampi of mouse models of amyloid pathology and post- mortem brains of human patients of AD.....and neural death (Zovoilis et al., 2011)."

      We have now changed the text at that point accordingly and also updated the legend of Figure 1F that also refers to this same study.

      -Authors refer to Hernandez et al., 2020 to state that B2 self cleavage is stimulated by some proteins however, Hernandez and colleagues studied only the effect of EZH2 protein. Please rephrase the sentence accordingly.

      Text has been modified accordingly.

      -Indicate a reference for the sentence: "......Ezh2, was reported as being responsible for the B2 RNA accelerated destabilization and processing during response to stress."

      The respective citation was added.

      -The format of many references is not consistent and has to be revised.

      We have switched to the Vancouver style. Some references in the legend and methods sections are referred independently from EndNote in case these text sections have to be moved to supplement in the final version in order to not create inconsistencies with endnote.

      Reviewer #3 (Significance (Required)):

      Finally, this reviewer generally remarks that “The research fields of non coding RNAs and neurodegeneration are attractive and challenging and, in my opinion, the molecular circuitry involving B2 RNAs might add important insights for understanding beta amyloid toxicity and neuronal death.

      However, this manuscript does not really add technical advances since the authors employed experimental approaches and bioinformatic analyses previously published by Zovoilis and colleagues in 2011 and 2016.”

      Our aim in the current manuscript was not to introduce a new method or experimental approach but rather to study the mechanisms behind B2 RNA regulation of gene expression in neural cells and particularly in amyloid pathology. Nevertheless, the current study constitutes the first reported short-RNA seq in this tissue and offers for the first time the ability to study B2 RNA processing in this tissue which is not possible with standard small and long RNA-seq.

      The reported findings might of interest of an audience of experts in non coding RNAs and neurodegeneration. The area of my expertise almost regards the biology of non coding RNAs from biogenesis to function manly focusing on neuronal and muscular systems both in physiological and pathological conditions.

    1. Note: This rebuttal was posted by the corresponding author to Review Commons. Content has not been altered except for formatting.

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      Reply to the reviewers

      We thank the reviewers for their useful suggestions to improve the manuscript and their support for publication. We have addressed all the comments that have been raised and carried out the suggested additional analyses, resulting in a significantly improved revised version of the manuscript. We provide hereafter a detailed point-by-point response to all questions and comments of the three reviewers.

      Reviewer #1 (Evidence, reproducibility and clarity (Required)):

      Centriole structure has been an attractive but challenging research topic for years. Pierre Gonczy's group has been working on its structure using cryo-electron tomography (cryo-ET). While the axoneme, which has longitudinal periodicity, was analyzed by several groups by cryo-ET for more than a decade, cryo-ET study on the centriole suffers from poor signal to noise ratio due to its limited length and thus fewer periodicity. They chose the centriole of flagellate Trichonympha, which have exceptionally long centrioles and thus offer opportunity of relatively straightforward sub-tomogram averaging. Their approach has been successful, and they revealed intermediate resolution structure of the cartwheel, key of 9-fold symmetry formation, and it's joint to triplet microtubules (Guichard et al. 2012, 2013, 2018).

      In this work, they employed modern state-of-art cryo-ET technique, such as direct electron detection and 3D image classification to upgrade our knowledge of centriole structure. In their past works, the central hub of the cartwheel, made of SAS-6 protein forming 9-fold complex, was described as an 8nm periodic object. With improved spatial resolution, they provided further detail with clear polarity, which will deepen our thought about the initial stage of ciliogenesis. They also compared two Trichonympha species (spp and agilis) as well as another flagellate, Teranympha mirabilis, and extended their intriguing evolutional and mechanical hypotheses based on structural differences.

      Despite improved spatial resolution, it is still not possible to identify proteins in the cryo-ET map (cellular cryo-ET will not reach such high resolution in the near future). Therefore, this work is rather geometrically descriptive, which will inspire molecular biologists to identify molecules by other methods. Nevertheless, this work demonstrated capability of cellular cryo-ET, especially analysis of structural heterogeneity. Thus, while biological topics handled are rather specialized for cilia from flagellate, this work will attract attention of any biologist interested in molecular structure in vivo. It is worth for publication in a high Journal after addressing the points below. This reviewer believes that the authors can address these points easily with additional analysis.

      We are grateful to the reviewer for the favorable evaluation and the many valuable suggestions, in particular concerning the processing pipeline, which we addressed by additional analyses, as detailed below.

      Major points:

      1. Entire scheme A graphic diagram of the entire cartwheel area, summarizing this work, is necessary for the readers' understanding (similar to Fig.6 of the other manuscript, Klena et al.).

      We thank the reviewer for this interesting suggestion, which we fully adhere to. As a result, we have generated a graphical summary of the work, which is shown in the new Figure panels 6B-F. Moreover, Figure 6A provides an evolutionary perspective regarding the presence of the CID and of what is now referred to as the fCID (filamentous CID, previously: FLS, see response to reviewer 3). This also helps to link our findings with the companion manuscript by Klena et al. This new Figure 6 is referred to extensively in the discussion of the revised manuscript (pages 13-16).

      Then average scheme should be shown in more detail, especially assumption of periodicity, Materials and Methods. The cartwheel hub was averaged with 25nm periodicity (as discussed below). Was the pinhead averaged with 16nm (as detected by FFT in Fig.S2L)? How about the triplet?

      This reviewer is not completely sure if the longitudinal averaging strategy is justifiable. Since periodicity of each domain is not trivial, logically the initial average must be done with the size of least common multiple (or larger). It is likely 96nm, assuming 25nm of the central hub is 3 times of microtubule periodicity and 16nm of the pinhead is twice of MT. 96nm average should be possible with a long cartwheel in this work. Alternative, in case periodicity is independent of MT and thus there is no least common multiple, is random picking and classification mentioned in "4. Periodicity". This should also be possible, since they can pick enough number of particles from long cartwheels.

      We apologize that the initial version of the manuscript was not sufficiently clear regarding the averaging pipeline that was pursued. To rectify this, we now provide a new Figure S1B to graphically explain the approach followed for STA. As depicted in this figure panel, the step size for sub-volume extraction was 25 nm both centrally and peripherally. This step size was selected because it corresponds to ~3x the major periodicity of ~8.5 nm observed in the power spectra of the sub-volumes. The 25 nm step size is larger than that previously used (i.e. 17 nm in Guichard et al. 2013), in order to identify potential features with larger periodicities. The fact that the step size was of 25 nm in all cases is now mentioned explicitly in the Materials and Methods section of the revised manuscript (line 649).

      We agree with the reviewer that 96 nm averaging is possible given the long cartwheel analyzed here, and such a piece of data was in fact included in the original submission, although with a different purpose. Indeed, we carried out STA using ~(100 nm)3 sub-volumes (with binning 3 to reduce computational time), the results of which are reported in Figure S7 (previously Fig. S6). For the purpose of this analysis, we focused on the lateral organization of the cartwheel, but did not use this dataset to explore other periodicities because of the limitations inherent to a binning 3 data set.

      • Classification*

      The authors analyzed structural heterogeneity inside the cartwheel hub, employing reference-free classification by Relion software. The program reveals multiple coexisting structures - two from Trichonympha agilis and three from Teranympha, respectively. Whereas this is an exciting finding and shows future research direction of this field, interpretation of this classification must be done carefully. ** It is puzzling that major (55%) population of T. agilis shows more ambiguous features than the minor population (45%), while spatial resolutions by FSC are not so different - for example, Fig.2H vs Fig.S5C. In case of Teranympha, it is even more drastic - Fig.4D (major class) seems blurred along the centriolar axis, compared to Fig. 4E (minor class). This reviewer is afraid that these "major" classes might contain more than one structure and after subaveraging be blurred in detailed features. The apparent good spatial resolution could be explained, when two structures coexist and subtomograms are aligned within each subclass. Probably lower resolution at the spoke region of the major class (Fig.S2A) than that of the minor class (Fig.S2D) is a sign of heterogeneity within this class. Another risk could be subtomograms with poorer S/N being categorized to one class (due to lack of feature to be properly classified). Fig.S5F (black dots localized in one tomogram) raised this concern.

      The following investigation will help to solve this issue. 1. Extract and re-classify subtomograms belonging to the major population. 2. Direct observation of tomograms. The authors could plot two classes of Teranympha (as they did for T. agilis in Fig.S5) and find features of the cylindrical cartwheel hub in two conformations (as shown Fig.4DE). Since such a feature was directly observed in tomograms from the other manuscript (left panels of Fig.S6AC in Klena et al.), it should be possible in this work as well.

      We agree with the reviewer that the interpretation of the classification must be done with care, and share her/his interest in better understanding the structural variability between cartwheels classes in T. agilis and T. mirabilis. Although poor S/N may in theory result in erroneous joint classifications, we note that all maps in the original submission stemmed from extensive focused 3D classification, which removed defective and spurious sub-volumes, nevertheless defining distinct classes in the cases reported. Obviously, however, we cannot exclude that much larger data sets and future software advances may lead to the identification of additional features that would allow further sub-classes to be identified.

      Regardless, we followed the two suggestions the reviewer offered to us and have (1) extracted and re-classified sub-tomograms belonging to the major populations and (2) undertaken a direct observation of tomograms. These two points are developed in turn below.

      (1) We have performed a further round of classification of the major populations in T. agilis (55 % class) and T. mirabilis (64 % class), to assess whether additional sub-classes might be identified and thus help further improve the quality of the central cartwheel map. However, this additional round did not yield new sub-classes nor notable improvement in the map quality as judged by visual inspections. We show in Rebuttal Figure 1 a comparison in each case of the original STA and the corresponding STA upon such re-classification. Importantly, all conclusions spelled out in the original submission hold upon further re-classification, indicating that the initial classification converged to the best map quality based on the current data set and available computational resources.

      (2) We have followed the suggestion of the reviewer and now show raw tomograms to confirm that the classes correspond to bona fide structures and not to processing artefacts (new Figures S1C-F). The resulting new Figure S1D for instance shows that the striking variations observed between classes in the T. agilis STA are also visible in the raw tomogram. The more subtle variations among T. mirabilis classes are more difficult to observe in the raw tomogram, but inherent variations that reflect the presence of two classes are nevertheless observed.

      Furthermore, following the reviewer’s suggestion, we now mapped the distribution of the two T. mirabilis cartwheel classes onto tomograms, revealing that both classes can occur next to each other within the same centriole (new Figure S8E).

      • Periodicity mismatch*

      In Fig. 2CD, periodicity of CID has discrepancy from that of the stacked SAS-6 ring (8.5nm and 8.0nm). Do the authors think this is a significant difference or within an error? The same question can occur to other subtomogram averages. It would be nice to show errors as shown in their other manuscript (Fig.3C of Klena et al.) and clarify their idea. If it is systematic difference of periodicity between the stacked ring and CID, this shift will be accumulated through the entire cartwheel region - after 100nm, 8.5nm/8.0nm difference can be accumulated to ~6nm, which should change the entire view of the subtomogram - and the main factor to be classified (periodicity mismatch). This artifact (or influence) should be removed (or separately evaluated) by masking CID (out and in) and run classification separately. By clarifying this, the quality of the major subaverages (mentioned in the previous paragraph) could be improved.

      The reviewer wonders whether there might be a periodicity discrepancy within one map, for instance between CID and spokes in the T. spp. cartwheel map (Fig. 2C and Fig. 2D). Here, the periodicity determined from the STA maps is 8.5 ± 0.2 nm (SD, N=4) for the CID and 8.0 ± 1.5 nm (SD, N=2) for the spokes. Based on these standard deviations, there is indeed no significant difference between the two, and thus no periodicity discrepancy. The same applies for measurements in T. agilis and T. mirabilis. The SDs were reported already in the figure legends of the original submission, and we would prefer to leave them there if possible and not mention them in the figures, which are pretty busy as is. We apologize if this was not clear enough in the initial manuscript. Likewise, one may wonder whether there might be periodicity discrepancies between structures from distinct maps, for instance between CID and A-links from T. spp. (Fig. 2C and Fig. 3D). Again, the measurements are within error, since the distance between adjacent CIDs is 8.5 ± 0.2 nm (N=4) and between adjacent A-links 8.4 ± 0.4 nm (N=6); a similar conclusion applies for the corresponding measurement comparisons in T. agilis and T. mirabilis. The figure legends have been altered in the revised manuscript to spell out that there are no significant differences between periodicities (lines 856-858).

      Furthermore, we would like to stress that, by definition, STA value are average distances. For instance, in the case of T. spp., the central cartwheel STA was obtained from 511 sub-volumes, and thus the reported N=2 represents the average distance from 511 sub-volumes. Since this is an average, errors can therefore not accumulate over longer distances. This point has also been clarified in the figure legends (line 856-858).

      • Periodicity*

      They averaged subtomograms extracted with spacing of 252A with initial average as the first template (p.18 Line22). This means they assumed 25nm periodicity from the beginning and excluded different or larger unit size (if they take search range wide, they could detect difference periodicity, but will still be biased by initially assumed 25nm). 25nm average allowed them to see more detail than before (when they assumed 8nm periodicity), but there is still a risk of bias from references. To avoid this risk, this reviewer would propose classification of randomly extracted (but of course along the cylindrical hub or along the triplet microtubules, so one-dimensionally random picking) subtomograms. This experiment will end up with multiple sub-averages, which are 25nm (or multiple times of that) shifted from each other. Then it will prove their assumption.

      We agree with the reviewer that in theory the choice of periodicity could introduce a bias. This is why we have chosen a larger step size than in our initial work, corresponding to ~3x the major periodicity of ~8.5 nm observed in the power spectrum of the sub-volumes, as mentioned above. Regardless, following the reviewer’s suggestion, we have now explored other types of periodicities by re-analyzing the dataset through extraction of non-overlapping sub-volumes along the proximal-distal centriole axis. In doing so, we randomized the starting position of the first box between tomograms, reaching the same goal as with random picking but maximizing the number of sub-volumes. We carried out this analysis for all T. spp., T. agilis and T. mirabilis cartwheel classes, and found no notable differences that would affect the conclusions of the manuscript compared to the initial overlapping sub-volume classification, albeit generally with a noisier STA due to the lower number of sub-volumes. A comparison of the two approaches is provided in Rebuttal Figure 2. Moreover, all the points regarding the choice of periodicity have been further clarified in the expanded Materials and Methods section (pages 19-21).

      Minor points:

      They discussed difference of stacked SAS-6 rings in the cartwheel from various species. How much is the sequence difference of SAS-6 among these species?

      Unfortunately, no genomic or transcriptomic data has been published for the species investigated here, although the sparse molecular data available from small subunit rRNA sequences allows one to establish an overall molecular phylogeny. We previously identified a SAS-6 homologue in T. agilis (Guichard et al. 2013), which shares 20 % identity and 45 % similarity with C. reinhardtii SAS-6. Despite low sequence conservation, the structural conservation of SAS-6 is predicted to be high between the two organisms (Guichard et al. 2013). We apologize if these points were not expressed sufficiently clearly in the initial rendition and have adapted the wording in the revised manuscript (lines 325-332).

      Are the authors sure that CID is nine-fold symmetric? It is not trivial.

      We thank the reviewer for bringing up this interesting point. We have applied 9-fold symmetrization to the entire central cartwheel comprising spokes, hub and CID/ fCID, a choice guided by the apparent 9-fold symmetry of the spokes and peripheral element. We investigated the impact of symmetrization on the CID by relaxing symmetry from C9 to C1 during refinement, but did not observe a difference, and thus continued with C9 symmetry, which improves map resolution by S/N ratio enhancement and additional missing wedge compensation. In addition, we have also analyzed the CID without symmetrization, as reported in Figure S7 (previously: Fig. S6). Note that these maps were generated with larger sub-volumes centered on the spokes to comprise hub, spokes and microtubule triplets, explaining the resulting lower resolution, as the missing wedge is not compensated. Despite these limitations, however, the unsymmetrized CID shown in Figure S7A and S7E resembles the one in the symmetrized maps of Figure 2, indicating that the CID indeed exhibits 9-fold radial symmetry. That this is the case is spelled out explicitly in the revised manuscript (lines 1145-1147).

      Fig.1C: Another cross-section from the distal region will be helpful. A longer scale bar is better for readers' understanding.

      We understand that the reviewer is curious about the distal region, and cross-section views of resin-embedded sections from T. agilis are available and could be provided if necessary. However, given that the focus of the manuscript is strictly on the cartwheel-bearing proximal region, we felt that featuring the distal region in detail would break the narrative. Therefore, we suggest to keep Figure 1 as in the original manuscript. Following the reviewer’s suggestion, we increased the size of the scale bars from 10 nm to 20 nm in Figure 1C as well as in the corresponding Figure S8C.

      Fig.S6F: It would be informative if the subclasses (25% and 20%) are distinguished in this mapping.

      As per the reviewer’s request, we provide in Rebuttal Figure 3 a side-by-side comparison of the T. agilis 25 % and 20 % classes centered on the spokes, which are noisier than the composite 45 % class due to the lower number of sub-volumes in each sub-class. Given that there are no notable differences between the two maps that would affect any of the conclusions of the manuscript, we feel it is best to keep what is now Figure S7F (previously: Fig. S6F) unchanged in the revised manuscript.

      A figure to explain the classification scheme will help readers understand. How many subtomograms did classification started? Were the 45% class classified into two (25% and 20%) groups by two-step classification or at once (the entire subtomograms were classified into three groups directly?

      We thank the reviewer for this useful suggestion. As a result, we have generated a new Supplemental Figure S1G-J that provides a graphical overview of the classification scheme, together with sub-volume numbers for all deposited maps, thus nicely complementing Table S1.

      Reviewer #1 (Significance (Required)):

      Nevertheless, this work demonstrated capability of cellular cryo-ET, especially analysis of structural heterogeneity. Thus, while biological topics handled are rather specialized for cilia from flagellate, this work will attract attention of any biologist interested in molecular structure in vivo. It is worth for publication in a high journal after addressing the points above. This reviewer believes that the authors can address these points easily with additional analysis.

      We reiterate our thanks to this reviewer for her/his favorable evaluation and detailed suggestions, which enabled us to generate a strengthened manuscript.

      Reviewer #2 (Evidence, reproducibility and clarity (Required)):

      Here, Nazarov and colleagues report sub-tomogram average (STA) maps of centrioles with 16 to 40 Å resolution from Trichonympha spp., Trichonympha agilis, and Teranympha mirabilis. Even though the authors have previously described the centriole architecture of T. spp, these STA maps of higher resolution revealed new features of centrioles, like polarized Cartwheel Inner Density (CID) and the pinhead. They also observed Filament-like structure (FLS) from T. mirabilis which seems to correspond to the CID from other species. Interestingly, they suggest that one and two SASS6 rings are stacked in an alternative fashion to make the central hub in T. mirabilis (Figure 5). The following issue should be addressed:

      Major points

      • Figure 4E. Authors mentioned in the manuscript that "We observed that every other double hub units in the 36% T. mirabilis class appears to exhibit a slight tilt angle relative to the vertical axis". When I see the other side, it does not seem to be tilted. Could the authors explain this?*

      We apologize that this aspect was not explained in sufficient detail. The left and right sides of the hub indeed appeared different in transverse views across the cartwheel center (previous Fig. 4E). This was because the area we selected in the original submission was centered on one emanating spoke. Due to the 9-fold symmetry one spoke density was selected on the right side, while the region between two spokes was displayed on the left side (as was illustrated by the slice across the center in previous Figure 4A; dashed rectangles in 4.0 nm panel). We have now selected a larger area to include spokes from both sides of the hub and thus better visualize this offset as shown in the modified Figure 4D-E.

      Reviewer #2 (Significance (Required)):

      I believe these results are of interest for all centrosome researchers and would like to recommend this manuscript be published in the EMBO journal which is affiliated with the Review Commons.

      We thank the reviewer for the recommendation to submit the revised manuscript to EMBO Journal, which we have followed.

      Reviewer #3 (Evidence, reproducibility and clarity (Required)):

      In this manuscript Nazrov et al., use cryo-electron tomography (CET) to analyse the structure of the centriole cartwheel. The Gonczy lab have previously generated a ground-breaking structure of the cartwheel from Trichonympha spp (T. spp.) (Guichard et al., Science, 2012; Guichard et al., Curr. Biol., 2013). This work is a direct continuation of those studies but using modern technology to get higher resolution images of the T. spp. cartwheel and comparing this to the cartwheel from Trichonympha agilis and from another distantly related flagellate Teranympha mirabilis.

      The data is generally well presented and of high quality. I am not an expert in CET, so it would be advisable to get the opinion from a reviewer who is, but the Gonczy lab are experienced in these techniques so I would not anticipate any problems. I have to admit that the title of the paper did not excite me, and I expected this to be a very worthy, but incremental study. It was a pleasure to find out that the extra detail provided by the increased resolution has revealed several new and unexpected features that have important implications for our understanding of cartwheel assembly and function. Most important are the potential asymmetry of the cartwheel hub, apparent variations in the packing mechanism of the stacked rings (even within the same cartwheel), and the potential offsetting of ring stacking. These findings will be of great interest to the field, and so I am strongly supportive of publication in The EMBO Journal. I have only a few points that I think the authors should consider.

      We thank the reviewer for this positive feedback and the recommendation to submit to EMBO Journal, which we hereby follow.

      Prompted by the comment of the reviewer, we revised the title to make it more informative and appealing to readers: “Novel features of centriole polarity and cartwheel stacking revealed by cryo-tomography”.

      • Nazarov et al., conclude that the cartwheel structure is intrinsically asymmetric. This is most convincingly based on the displacement of the CID within the hub, but they state that the Discussion that the potential offset between the Sas-6 double rings generates an inherently polar structure. I didn't understand why this is the case. Looking at Fig.S9A,B I can see that the offset in B could tilt to the left (as shown here) or to the right (if the structure was flipped by 180o). But I couldn't see how this makes this structure polar in the sense that a molecule coming into dock with the structure could only bind to one side of the offset structure shown in B, but to both sides of the aligned structure shown in A. I think this needs to be explained better, as it is crucial to understand where any potential polarity in the cartwheel structure comes from.*

      We apologize for not having been sufficiently clear about how two SAS-6 rings with an offset could impart organelle polarity. The reviewer is correct that an offset between superimposed rings alone is not sufficient to generate polarity at a larger scale. The important point we would like to stress, however, is that we discovered concerted polarity in multiple locations, from the central hub to the peripheral elements as illustrated in Fig. S7C-D, S7G-H, S7K-L and S7O-P (previously: Fig. S6). Prompted by the reviewer’s comment, we now better emphasize the asymmetric tilt angles of merging spokes, as highlighted also in the improved Figure S7. This asymmetric spoke tilt angle allows one to discriminate the proximal and distal side of a double SAS-6 ring, which is now explained better in the text (lines 259-263 & 502-510).

      • Related to this last point, in a co-submitted paper Klena et al. do not report such an asymmetry in the hub structures they have solved from several different species (neither in the tilting of the hub, or the displacement of the CID). I think it would be worth both sets of authors commenting on this point.*

      We agree that comparing and contrasting the results of the two companion manuscripts is important and we have updated the text as a consequence in several places (lines 444, 467, 507, 536, 985, 1000). We know from our previous work (Guichard et al. 2013) that the asymmetry of the hub and spoke is not visible at lower resolution. In the accompanying manuscript by Klena et al., no offset in the hub or asymmetric CID localization is reported, probably due to lower resolution and differences between species.

      • The authors data strongly suggests that the T. ag. and Te. mir. hubs are composed of a mixture of single and double Sas-6 rings. In contrast, the T. spp. cartwheel only has a single class of rings, but it wasn't absolutely clear if the authors think this comprises a single or double ring. In the text it is presented as though the elongation of the hub densities in the vertical direction is a new feature of the T. ag cartwheel (Fig.2H,I), but to me it looks as though this is also apparent in the T. spp. cartwheel (Fig.2C,D). The authors should address this directly and, if they believe that T. spp. has a double ring, they should comment on whether this more regular structure seems to have offset rings. If not, then the offset rings are unlikely to be the source of asymmetry that leads to the asymmetric displacement of the CID. Finally, if the authors think these are double rings, they should also be clear that they would now slightly re-interpret their original T. spp. cartwheel model (Figure 2, Guichard et al., Curr. Biol.). There is no embarrassment in this-a higher resolution structure has simply revealed more detail.*

      We apologize if the conclusions drawn about T. spp. cartwheel hubs were not sufficiently clearly expressed. Like the reviewer, we think that elongated hub elements are also discernible in T. spp., something that is also illustrated by the intensity plot profile in Figure 2C (double peaks on light blue line). These points are spelled out more explicitly in the revised manuscript (lines 177-179). In addition, to emphasize the conservation of the double hub units in both Trichonympha species, we have likewise adapted the text for T. agilis (lines 198-201).

      As for the offset observed within T. spp. spoke densities in Figure S10H, we interpret this as evidence for an offset of the double ring at the level of the hub, although we have not observed such offset in T. spp. for reasons that are unclear. The fact that this revises our previous interpretation based on a lower resolution map of T. spp. was already mentioned in the initial submission but is now better emphasized (lines 171-172 & 179-181).

      • The authors conclude that T. mirabilis cartwheels lack a CID and instead have a filament-like structure (FLS). I wonder whether it is more likely that the FLS is really a highly derived CID that appears to be structurally distinct when analysed in this way, but that will ultimately have a similar molecular composition. This situation might be analogous to the central tube in C. elegans, which by EM appears to be distinct from the central cartwheel seen in most other species, but is of course still composed of Sas-6. This historical tube/cartwheel nomenclature is now cumbersome to deal with, so perhaps it would be better to be cautious and not give the T. mirabilis structure a completely new name-how about "unusual CID" (uCID).*

      We share the view that the CID and the “FLS” –the term used in the initial submission- may have a related molecular composition and function, as we had also speculated in the discussion of the original submission. Following the reviewer’s suggestion, and in an effort to have a more uniform nomenclature, we propose to dub the T. mirabilis structure “filamentous CID” (fCID). This highlights better the similar location of these two entities and their potential shared function, while stressing the filamentous nature of the fCID. We further emphasize this point by providing the new Figure 6A to compare the presence of the two entities in select species. The discussion has also been adapted accordingly (pages 13-14).

      Rebuttal Figure Legends

      Rebuttal Figure 1: Re-classification of major classes

      (A-D) Transverse (top) and longitudinal (bottom) views of T. agilis (A, B) and T. mirabilis (C, D) central cartwheel 3D maps. The final major classes reported in the manuscript (A: 55 % class, C: 64 % class) were subjected to re-classification, which again yielded one major class in each case, with no notable improvement (B, D).

      Rebuttal Figure 2: Reclassification with non-overlapping sub-volumes

      (A-F) Transverse (top) and longitudinal (bottom) views of T. spp. (A, B) T. agilis (C, D) and T. mirabilis (E, F) central cartwheel 3D maps. The final maps reported in the manuscript (A, C, E) were generated with a 25 nm step size, yielding overlapping sub-volumes, whereas the maps in (B, D, F) were generated from non-overlapping sub-volumes, with no notable differences between the two that would affect the conclusions of the manuscript.

      Rebuttal Figure 3: Polar centriolar cartwheel upon sub-classification

      (A-C) 3D transverse views of non-symmetrized STA centered on the spokes to jointly show the central cartwheel and peripheral elements in the T. agilis 45 % class (A), as well as separately in the 25 % class (B) and 20% class (C). No notable differences are apparent following such re-classification, apart from the output being noisier due to the lower number of sub-volumes in each sub-class.

    1. Thefamily becomes exhausted and, strange though it may seem,begins to prepare for the worst. It was like that in my family.There were four siblings, two men and two women, my fatherand my mother. Only he, the baby brother, the most protectedand loved one chose to use drugs. My parents suffered a lotwhen he left for good, because actually he had been graduallyleaving us for a long time. For us (siblings) it was hard, too,but we got married, remember him with longing every nowand then, but we got back on our feet. It was much worse formy parents; they felt guilty, when he died the pain increased.The situation is better now, I think they are more relieved,but they won't confess to it. Living with a drug dependentis very hard, I did to him everything I could and also what Icouldn't. Then I eventually I realized that one share was his todo, but h

      This quote is interesting because it shows the thinking behind why it can bring relief to the family. "Because actually he had been gradually leaving us for a long time." The family will notice when the individual is not themselves anymore, in a way it is as if the individual had "died" even before he actually did. This is because the family expects the individual in someway to overdose, either accidentally or on purpose. The topic of overdosing is one the families mind. When the individual does die it brings relief because they no longer have to worry about that person and the stress that they experienced goes away.

    Annotators

    1. But in the very first pages of the ElementaryStructures Lévi-Strauss, who has begun by giving credence to these con-cepts, encounters what he calls a scandal, that is to say, something whichno longer tolerates the nature/culture opposition he has accepted,something which simultaneously seems to require the predicates of natureand of culture. This scandal is the incest prohibition. The incest prohibitionis universal; in this sense one could call it natural. But it is also aprohibition, a system of norms and interdicts; in this sense one couldcall it cultural

      I think that calling incest universally prohibited is incredibly humanist. For example, male-born clownfish, in times of genetic scarcity, may change their sex and sometimes mate with their "sons". In any human context, this is taboo and actually just incompatible with our hormone systems, but I think we need to remember that we are still animals, and calling the incest prohibition natural is something that is incorrect. What are some animal social constructs that challenge human societal constructs that we pose as "universal?"

    1. Note: This rebuttal was posted by the corresponding author to Review Commons. Content has not been altered except for formatting.

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      Reply to the reviewers

      Note from the authors (AU): This manuscript has been reviewed by subject experts for Review Commons. The authors would like to thank the reviewers for their comments to the manuscript, and the editor for patience with our response. Our reponse was delayed due to the COVID-19 lock-down situation in our institution. Now we are pleased to provide the following point-by-point response, as detailed below.

      Reviewer #1 (Evidence, reproducibility and clarity (Required)):

      The manuscript by Suomalainen et al. describes a fluorescence-based approach combined with high-resolution confocal microscopy to study the heterogeneity of adenovirus infection in a population of human cells. The main focus of the authors is the detection of viral transcripts in infected cells, how this correlates with viral genomes, the cell state, and how it varies between different cells in a single population. The paper is generally well written and easy to read, with a few typos, although I found parts of it to be somewhat length and repetitive. Particularly the results section could be pruned somewhat for readability and clarity. The major limitation of the study as it stands is it's overall impact and novelty, which limits journal selection somewhat. A very similar study was recently published, which the authors cite (Krzywkowski et al, 2017). Nevertheless, I think the study design is rigorous and well executed, but I do have some specific comments which may enhance it's overall impact and novelty.

      **Major:**

      Results "Visualization of AdV-C5..." section:

      Why not also look at normal cells that can be synchronized? Cancer cells, such as A549 will by definition be highly heterogenous and at all phases of the cell cycle. Primary non-transformed cells can easily be synchronized by contact inhibition and are much more physiologically relevant.

      AU: In the current manuscript, we concentrated on the early phases of the AdV-C5 infection, on the question how virus gene expression is initiated and whether the cell cycle phase of the host cell impacts the initiation of virus gene expression. Answering these questions requires use of cells that express good amount of virus receptors so that viruses efficiently bind to the cells and infections can be synchronized so that extended time does not elapse between virus addition and accumulation of E1A transcripts; extended time between these two steps would make interpretation of the results more complex since cells could have progressed from one cell cycle stage to another during the experiment. Furthermore, having cells at all phases of the cell cycle is actually a benefit since then the experiment can be carried out under an “unperturbed” condition; all cell cycle synchronization methods have pleiotropic effects on the cells.

      It is true that primary non-transformed cells are physiologically more relevant than cancer cells, but primary cells have issues with donor-to-donor variability and many primary cells express rather low amounts of AdV-C5 receptors, so synchronized infections in these cells are not possible. Furthermore, the extended cell morphology of many normal fibroblast cell lines and the tendency of cell extensions from neighboring cells to overlap makes fluorescent images of these cells incompatible for automated cell segmentation.

      Here, we provide data also from HDF-TERT cells (nontransformed human diploid fibroblasts immortalized by human telomerase expression) to show that two of our key findings from A549 cells are not artefacts of cancer cells. This is, that akin to A549 cells, the infected HDF-TERT cells accumulate high number of E1A transcripts (Fig.1C), and also in these cells nuclear vDNA numbers do not predict the cytoplasmic E1A transcript counts during early phases of infection (S2C Fig). However, since HDF-TERT cells are rather inefficiently infected by AdV-C5, correlation of early E1A transcript accumulation to the cell cycle phase of the host cell could not been done in these cells. We have been unable to identify primary or normal immortalized cells that would be easily available and efficiently infected by AdV-C5 (synchronized infection with short time elapsed between virus addition and accumulation of E1A transcripts).

      "The virus particles bound..." - Can the spatial resolution of a confocal microscope truly differentiate individual particles that are sub-wavelength in size? What about the sensitivity for single particles? Some sort of experiment to show that single particles can be detected should be performed and shown to assure the readers that this is in fact possible. Furthermore, even when based on the particle to pfu ratio, the MOI would still be nearly 2000pfu/cell, so the actual number of observed particles is an order of magnitude lower than what was applied to the cells.

      AU: The fluorescence signal from individual fluorophore-tagged AdV or anti-hexon antibody-decorated particle is bright enough to be picked up by PMT or HyD detectors of the current confocal laser scanning microscopes. In fact, tracking fluorophore-tagged particles of the size of AdV has been a standard microscopy procedure since late 1990’s.

      Because the Reviewers were questioning the apparently high multiplicity of infection used in the experiments, we clarify the difference between “standard” MOI estimations and our infection set-up. First of all, as described in Material and Methods, we estimated the number of physical virus particles in our virus preparations using A260 measurements (J.A. Sweeney et al., Virol. 2002, doi: 10.1006/viro.2002.1406). This method, like all other methods used to estimate virus particle numbers, is likely not 100% reliable.

      Second, we incubated the virus inoculum with cells only for 60 min, after which the unbound viruses were washed away. During this short incubation time only a small fraction of input virus particles bind to cells, and indeed as shown in Fig.1A, a theoretical MOI of 54400 physical virus particles/cell or 13600 physical virus particles/cell yielded Median of 75 and 26 bound virus particles per cell, respectively. Interpretation of the results from the cell cycle assays required that there was a relatively short time between infection and analysis so that cells in a large scale did not change their cell cycle status during the experiment. This required use of a rather high MOI. Furthermore, for collection of a large data set, it is convenient that every cell is infected.

      Third, what exactly does one pfu mean in terms of physical adenovirus particles? There is no clear answer to this, since several parameters affect the pfu. In which cells was the titration carried out? How long was the input virus inoculum incubated with the cells? How many of the virus particles entering the cell actually established an infection? And, as described in A. Yakimovich et al. (J. Virol. 2012, DOI: 10.1128/JVI.01102-12), only a fraction of infected cells produce a plaque. The majority of papers stating that x pfu/cell was used for infection, usually incubate the cells with the virus inoculum for several hours at 37°C, and never make any attempts to estimate exactly how many virus particles entered into the cells.

      Fig. 4 - I am not certain that the observed difference is significant, at least looking at it, beyond the width difference of the peaks, highest expression for both is largely in G1. It would be nice to see this using a western blot of cell cycle sorted cells, which can easily be accomplished using FACS.

      AU: In the highest GFP expression bin, CMV-eGFP expressing cells have 43% cells in G1 and 50% in S/G2/M. In comparison, E1A-GFP expressing cells have 58% cells in G1 and 35% in S/G2/M. The difference in G1 cells in the highest eGFP bin is statistically significant (p Page 15, 2nd paragraph. It would be valuable and informative to determine whether there is heterogeneity in histone association with these different vDNAs and whether these histones exhibit divergent modifications (enabling or restricting transcription). Same as above. I am rather surprised that the DBP signal did not correlate well with vDNA signal, particularly for the larger replication centers. How can this be reconciled? Was there an increase in overall vDNA signal later in infection? It is important to know this as it determines whether the observed vDNA signal is real or could be caused by viral RNA or other background causes (non-infected controls notwithstanding). Can the signal be detected with inactivated viruses (via UV for example?)

      AU: Whether histone modifications impact the transcriptional output of adenovirus genomes early in infection is indeed an intriguing question, but unfortunately this is very challenging, if not impossible, to study at single-cell / single vDNA level with the existing technology. Techniques for single-cell measurements of chromatin states are still in infancy, although some notable advancements in this field were reported in 2019 (e.g. K. Grosselin et al. Nature Genetics, DOI: https://doi.org/10.1038/s41588-019-0424-9 and S. Ai et al. Nature Cell Biology, DOI: https://doi.org/10.1038/s41556-019-0383-5).

      Furthermore, current literature offers a confused picture as to when exactly protein VII on incoming virus genomes is replaced by histones (reviewed in the reference 39, Giberson et al.). Of note, the vast majority of incoming nuclear vDNA molecules scored protein VII-positive with anti-VII staining under the experimental conditions used for the Fig. 2C data. However, we did not include these results into the manuscript because VII-positive signal on vDNAs does not exclude these vDNAs having histones on certain parts of the genome.

      The Reviewer wonders why the DBP signal in Fig.6C does not correlate with vDNA signal. There is no discrepancy here because DBP signal in the figure is a proxy for replicating vDNA whereas the click vDNA signal reports incoming vDNA. The one DBP spot without an associated click vDNA signal could be due to a replication center originated from a replicated viral genome, not from incoming viral genome. The figure shows that incoming vDNAs within the same nucleus initiate replication asynchronously.

      Page 18, 1st paragraph. It would be interesting to determine whether there was association between pol II and those genomes that showed no E1A, similarly to the histone suggestion. What about things like viral chromatin organization? Soriano et al. 2019 showed how E1A and E4orf3 work in tandem to alter viral chromatin organization by varying histone loading on the viral genome.

      AU: This again would be technically very challenging to show. We actually tried to visualize active transcription using an antibody against RNA polymerase II CTD repeat YSPTSPS (phosphor S5), azide-alexa fluor488 and anti-alexa fluor488 antibody to mark EdC-labeled incoming vDNAs and proximity ligation assay for signal amplification. However, this method was not sensitive enough to detect RNA polymerase II association with individual viral genomes. We only detected the proximity ligation signal in replication centers when replicated viral genomes were tagged with EdC.

      Fig. 2. Can you really say that a single dot correlates with a single transcript? Has that been validated in any way?

      AU: Signal amplification with branched DNA technology leads to binding of a large number of fluorescent probes to a mRNA and thus enables detection of single nucleic acid molecules. This has been validated e.g. in A.N. Player et al. 2001. J. Histochem. Cytochem (https://doi.org/10.1177/002215540104900507) and N. Battich et al. 2013. Nature Methods (https://doi.org/10.1038/nmeth.2657).

      **Minor:**

      Page 5, last paragraph. "Transcirpts from the viral late transcription unit,..." This is not correct as recently shown by Crisostomo et al, 2019.

      AU: The data in Crisostomo et al. paper suggest that some late gene expression can occur before vDNA replication, but an abundant accumulation of late transcripts coincides with onset of vDNA replication. However, the Crisostomo et al. study did not test what the levels of late gene transcripts are if the vDNA replication was inhibited. But to acknowledge the possibility that there might be some level of late gene transcription prior to replication of the viral genomes, the sentence is modified as follows: “Transcripts from the viral late transcription unit, amongst them mRNAs for the viral structural proteins, vastly increase in abundance concomitant with the onset of vDNA replication”. Furthermore, we have added the Crisostomo et al. reference here as well.

      Page 10, "... because AdvV-infected cells are less well adherent..." This is not strictly true as loss of attachment only occurs later on in infection. It would be helpful to have statistical significance indicated directly in the figures.

      AU: Although clearly visible cell rounding indeed occurs only late in infection, also during early stages of infection the HAdV-C5-infected cells are less adherent than non-infected cells. In many assays this is not obvious, but the RNA FISH staining procedure includes several incubation and washing steps in rather harsh buffers, and we observed random, sometimes considerable, cell loss with infected cultures but not with non-infected cultures.

      In the revised manuscript we have included the statistical significance P values both into the main text and the figure legends, but not to the figures directly, because the P values were generated with different statistical tests and P values should not be shown/mentioned without stating which statistical test was used. However, we noticed that we had in some cases omitted to mention what was the number of pairs analyzed in some of the Spearman’s correlation tests. This has now been corrected in the revised manuscript.

      The very high MOIs used are concerning, could these have negative effects on the cell viability or overall state?

      AU: We refer to our explanation above about the theoretical MOI and the actual MOI. Furthermore, in the experiment described in Fig.2C (correlation of E1A transcripts per cell vs. viral genomes per cell), 42% of analyzed cells had ≤ 5 viral genomes/cell and 27.5% of analyzed cells had between 6-10 viral genomes per cell; these are not high numbers. We also provide controls that the EdC-labeled genomes are detected with good efficiency. Hence the EdC-labeled genomes per cell are a good estimate of the numbers of virus particles that indeed entered into the cells.

      There are a few typos and such that should be corrected. AU: We have tried to find and correct the typos.

      Reviewer #1 (Significance (Required)):

      As I stated above, the work is interesting and significant, to a degree. The major limitation is that the novelty is low as a paper published in 2017 (cited by the authors) used a very similar approach to investigate a similar problem. In addition, there are multiple other recent papers looking at cell populations in the context of adenovirus infection, and whether a single cell or population based approach is better is unclear. This is something the authors might want to strengthen prior to submission.

      AU: In the current study, we focused on the early phase of HAdV-C5 infection, on how viral gene expression is initiated and how individual nuclear viral genomes proceed to a replicative phase. The Krzywkowski et al. 2017 J. Virol. Paper that the reviewer refers to used padlock probe-based rolling circle amplification technique to simultaneously detect HAdV-C5 genomes and viral mRNAs in individual infected cells.

      The shortcoming of this method is inferior sensitivity compared to the branched DNA technology-based method used by us in the current study. Krzywkowski et al. were able to pick up signals from virus mRNAs and virus genome only relatively late in the infection, i.e. at the time when incoming genomes were expected to have multiplied by replication. Thus the study by Krzywkowski et al. was unable to provide information for the questions addressed in our study, i.e. do the levels of E1A transcripts early in infection correlate with viral vDNA counts in the nucleus and is there variability in the transcription output from individual vDNAs within the same nucleus, or variability in how individual vDNAs within the same nucleus proceed into the replication phase. We hence do provide novel information, and do not consider this as a limitation of our paper.

      We emphasize that population assays are done to attempt to understand molecular basis of a phenomenon by correlations. Instead, deep molecular insights require to-the-point-assays, in the case of transcription, single-molecule live cell assays at the level of single genes. Technically, we (and also the field) are not quite there yet.

      Regardless, our study is a first step towards understanding transcription output of nuclear HAdV-genome at single-cell, single-genome levels. It has revealed insight that was not apparent from population assays. It is clear that the next step will be time-resolved live cell assays with simultaneous detection of transcription output, genome detection and transcription factor clustering on the genomic loci. With current technology the simultaneous detection of all these events is challenging, and requires the development of further technology.

      Reviewer #2 (Evidence, reproducibility and clarity (Required)):

      The authors show heterogeneity of AdV-C5 mRNA transcript quantity and dynamics in different cell types, which is regulated by the cell cycle phase and does not correlate to incoming viral DNA, using single molecule RNA FISH technologies and detection of incoming viral DNA by EdC labeling.

      **Major Comments:**

      The authors change the MOI used in their experiments (7 different MOIs are used throughout the paper) in a manner that appears randomly and without explanation. (54400 for Figure 1A, 1B, 3B, S3B; 37500 for Figure 1C; 23440 for Figure 2A, 2C, S5A; 13600 for Figure 1A, 1D; 36250 for Figure 3C, S3D; 11200 for Figure 4B; 23400 for Figure 6B). The authors should provide explanation, why these changes in MOIs are necessary.

      AU: The MOIs given are theoretical MOIs, and essentially all figures indicate what was the actual MOI, that is, the real number of virus particles entering into the cells. This is beyond what is commonly provided in virology. It is essential, however, since MOI differs between different cell types. Therefore, we prefer to use the actual MOI as shown in Fig.1A, or we indicate the number of vDNAs that were delivered to the cells of interest.

      Variable MOIs had to be used to ensure that different cell lines received comparable numbers of virions, in particular virus particle binding to and entering into the cells. Infection kinetics are different in different types of cells, but can be tuned by MOIs used. Furthermore, different virus preparations were used in the experiments and we performed analyses at different stages of the infection cycle. Due to all these different facettes provided by our experiments, it was impossible to choose one standard (theoretical) MOI for all the experiments.

      The authors use mean fluorescence intensity of E1A probes per cell as estimate for viral transcript abundance for some of their experiments (Figure 1D, E, 3B), and count E1A punctae as measure for E1A transcripts in other experiments (Figure 2C, 3C, 5), without showing data, that these measures correlate. Problematic is hereby, that not all E1A punctae have the same signal intensity, as can be seen in Figure S1, which makes the estimation of the correlation of E1A punctae (= number of transcripts) and fluorescence intensity difficult. The authors should provide both (E1A punctae counts and estimation via fluorescence intensity) for at least one experiment, to prove, that the estimation of E1A transcript levels via fluorescence intensity is feasible.

      AU: The quantification method had to be adjusted to the number of virus transcripts in the cell at the time of analysis. The best quantification method is segmentation and counting the individual fluorescent puncta per cell, but, as stated in the manuscript, this method does not accurately quantify the mRNA puncta from maximum projections of confocal or widefield image stacks when the number of puncta per cell exceeds ~ 200.

      On the other hand, as shown in the quantification below, mean fluorescence intensity measurements per cell do not of course distinguish between cells having one vs. two mRNA puncta. Yet, as shown in the figure below, a relatively good correlation between puncta counting and fluorescence intensity measurements is achieved when cells have ≥ 10 transcripts per cell. Subsets of randomly picked images of the Fig.2C/Fig.5 dataset were included into the analysis (rs is Spearman’s correlation rank coefficient, approximate P p.15: "The nuclear E1A signals in AraC-treated cells were resistant to RNase A, but they were dampened by treatment with S1 nuclease (S6B Fig)." The authors make this statement based on (i) two completely different timepoints (12 h.p.i. for RNaseA treatment, 24.5 h.p.i. for S1 nuclease treatment) and (ii) in different clones of the A549 cells as stated in the methods section on p.21 (Two different clones of human lung epithelial carcinoma A549 cells were used in the study: our laboratory's old A549 clone (experiments shown in Fig. 1, Fig. 3B and S1 Fig., S3B and S3C Fig., S6A and S6B Fig., RNase A treatment) and A549 from American Type Culture Collection (ATCC, experiments shown in Fig. 2 and Fig. 5, Fig. 6, S2B Fig., S4 Fig., S5 Fig., and S6B Fig. S1 nuclease-treatment)). This makes it difficult to interpret, if the data is due to differences in the timepoints or cell types, or if it is due to binding of the E1A probe to single stranded vDNA.

      AU: This is a fair criticism, thank you. We have replaced the RNase A figure S6B in the revised manuscript. A new RNase A experiment was repeated in ATCC A549 cells using the same infections conditions as with the S1 nuclease-treated cells.

      **Minor Comments:**

      p.4: "AdV are non-enveloped, double-stranded DNA viruses that cause mild respiratory infections in immuno-competent hosts, and establish persistent infections, which can develop into life-threatening infections if the host becomes immuno-compromised [reviewed in 6]." Not all AdV cause respiratory diseases, the disease outcome of human AdV depends on the site of primary infection, which differs between the different AdV types.

      AU: We have modified the text as follows: AdV are non-enveloped, double-stranded DNA viruses that cause mild respiratory, gastrointestinal or ocular infections…

      p.7: The authors state, that "At the 17 h time point, about half of the cells had high numbers of protein VI transcripts, and most of them very high numbers of E1A transcripts.", however, the picture shown in Figure 1F shows a different phenotype, with low transcript levels of VI in E1A high cells and high transcript levels of VI in E1A low cells.

      AU: This was perhaps a bit difficult to see in the overlay images since one has to distinguish between green and yellowish green. We have provided the individual channels along the overlay picture in Fig. S1D, and now it is clear that at 17h pi cells with high numbers of VI transcripts have also high numbers of E1A transcripts.

      p.8: "This nuclear E1A signal is due to binding of the E1A probe to single-stranded vDNA in the replication centers (see below)." The authors should state here, that due to the binding of the probes to the single stranded vDNA in the replication centers, the nucleus was excluded from the analysis for Figure 1F in late timepoints.

      AU: We have modified the text according to the Reviewer’s suggestion. The text is now as follows: ‘Due to further studies (see below), we assume that this nuclear E1A signal represents binding of the E1A probe to single-stranded vDNA in the replication centers. Accordingly, the nuclear area was excluded when quantifying the viral transcripts per cell in late timepoints (Fig. 1F).’

      Due to this time point the author cannot state that the E1A staining seen (Fig. 1F; indicated with white arrows) are replication centers; this is just an assumption, since there is no evidence in Fig 1 the author cannot be sure; the author should change the text: "taking the following experiments into account...", "due to further studies (see below)..... we assume that..."

      AU: We have modified the text according to the Reviewer’s suggestion; see also the previous comment above.

      p.8: The authors should mention the figure they refer to, since there is no E1B-55K staining in Fig. 1F

      AU: The text has been modified as follows: Whereas other time points showed relatively few E1A, E1B-55K or VI puncta over the nuclear area (Fig. 1B, 1F, S1A Fig.), clustered nuclear E1A signals were apparent at 23 h.

      p.9: Which test was used to calculate the additional p-values?

      AU: As stated in the Material and Methods section or the figure legends, the p-values were calculated either by a permutation test using custom-programmed R-script (the code has been deposited on Mendeley Data along with other data associated with this manuscript), or by Kolmogorov-Smirnov test using GraphPad Prism. GraphPad Prism was also used to calculate Spearman’s correlation coefficients and the associated approximate p values. In the revised manuscript, we have added the following sentense into the Material and Methods section / Statistical analyses: Spearman’s correlation tests were done using GraphPad Prism.

      p.10: For the experiment for the correlation of viral genomes per cell and E1A transcripts in HDF-TERT cells (Figure S2C), the MOI is missing in the description of the results, as well as in the corresponding figure legends.

      AU: We have indicated the theoretical MOI (~ 4800 virus particles per cell) in the figure legend and in the Material and Methods section. The actual MOI, i.e. the actual number of virus particles entering into the cells, could not be determined due to the long (15 h) incubation time of virus inoculum with the cells, which in turn was required because these cells bind AdV-C5 rather inefficiently. However, between 1 and 32 EdC-labeled virus genomes were detected per cell nucleus at 22 h pi.

      11: calculation of correlation? rs? Why does the author combine S and G2/M phase? Fig. S3A show different values for the phases

      AU: rs is the abbreviation for Spearman’s correlation coefficient, and, as indicated in the Material and Methods, we used GraphPad Prism to calculate the Spearman’s correlation coefficients.

      Different methods to estimate cell cycle stages. DNA content method cannot separate S and G2/M with great confidence, whereas Kusabira Orange-hCdt1 and Azami-Green-hGeminin expressions in HeLa-Fucci cells allow more fine-tuned assessment of the cell cycle phases.

      p.11: "Thus, the total intensity of nuclear DAPI signal can be used to accurately assign G1 vs S/G2/M stage to cells." The authors should also here refer to other papers, which showed that this correlation is feasible, as they did in the methods section (67. Roukos V, Pegoraro G, Voss TC, Misteli T. Cell cycle staging of individual cells by fluorescence microscopy. Nature protocols. 2015;10(2):334-48. Epub 2015/01/31. doi: 10.1038/nprot.2015.016. PubMed PMID: 25633629; PubMed Central PMCID:PMCPMC6318798.), and maybe also refer to a newer paper which deals with this technique: Ferro, A., Mestre, T., Carneiro, P. et al. Blue intensity matters for cell cycle profiling in fluorescence DAPI-stained images. Lab Invest 97, 615-625 (2017). https://doi.org/10.1038/labinvest.2017.13

      AU: The integrated nuclear DAPI signal intensity is indeed a widely used method to assign cell-cycle stage to individual cells. We have added the second reference suggested by the Reviewer to the reference list for this method.

      p.11: "Furthermore, when focusing on the highest E1A expressing cells, i.e. the cells with mean cytoplasmic E1A intensities larger than 1.5 × interquartile range from the 75th percentile, 71.9% of these cells were found to be in the G1 phase of cell cycle, whereas only 55.8% of cells in the total sampled cell population were G1 cells." The authors do not provide any reference to a figure within the manuscript or the supplements, which contains these data. Are these data not shown in the manuscript?

      AU: These values are calculated from the data shown in Fig.3B. The source data supporting findings of this study (maximum projection images, excel files of the CellProfiler and Knime workflows) have now been deposited to Mendeley Data as stated in the Material and Methods / Data availability section of the revised manuscript and listed in Supplementary tables.

      p.12: punctuation mistake; . instead of , To enrich G1 cells. AdV-C-5 (moi ~ 36250) was added. Why does the author switch between signal intensities and counting E1A puncta per cell (limited to 200) in the different experiments to illustrate accumulation of E1A transcripts?

      AU: The same answer as above: the quantification method had to be adjusted to the number of virus transcripts in the cell at the time of analysis. The best quantification method is segmentation and counting the individual fluorescent puncta per cell, but, as stated in the manuscript, this method does not accurately quantify the mRNA puncta from maximum projections of confocal or widefield image stacks when the number of puncta per cell exceeds ~ 200. On the other hand, as shown in the quantification in the new S1C Fig., mean fluorescence intensity measurements per cell do not of course distinquish between cells having one vs. two mRNA puncta, but a relatively good correlation between puncta counting and fluorescence intensity measurements is achieved when cells have ≥ 10 transcripts per cell.

      p.14: "For E1A (or E1B-55K), we did not detect transcriptional bursts with bDNA-FISH probes on nuclear vDNAs, either prior to or after accumulation of viral transcripts in the cell cytoplasm." The authors do not provide any reference to a figure within the manuscript or the supplements, which contains these data. Are these data not shown in the manuscript?

      AU: This statement is based on hundreds of images we have analyzed during the course of the study. It is impossible to show all of these images, so in principle, this is “data not shown”. We have modified the text as follows: With hundreds of images analyzed, we never unambiguously detected transcriptional bursts with E1A (or E1B-55K) bDNA-FISH probes on nuclear vDNAs, either prior to or after accumulation of viral transcripts in the cell cytoplasm.

      p.14: space between number and %

      AU: Thank you for pointing this out. It has been corrected.

      p.15: "This is was also seen in AdV-C5-EdC-infected cells" should be changed to "This was also seen in AdV-C5-EdC-infected cells"

      AU: Thank you for pointing this out. It has been corrected.

      Fig. 1B:

      −figure legend does not indicate how cells were staine −also no description in the continuous text −which E1A transcripts are stained? all? 12S? 13S?

      AU: The first sentence in Results section states that “We used fluorescent in situ hybridization (FISH) with probes targeting E1A, E1B-55K and protein VI transcripts followed by branched DNA (bDNA) signal amplification to visualize the appearance and abundance of viral transcripts in AdV-C5-infected A549 lung carcinoma cells.” Furthermore, the legend to Figure 1 starts with the title “Visualization of AdV-C5 E1A, E1B-55K and protein VI transcripts in infected cells by bDNA-FISH technique”, and the legend to Fig.1B mentions that “cells were stained with probes against E1A and E1B-55K mRNAs or E1A and protein VI mRNAs”. We are of the opinion that this is enough information to understand the figures.

      The main text to Fig.1 also states that “The E1A probes covered the entire E1A primary transcript region and thus all E1A splice variants. The temporal control of E1A primary transcript splicing and E1A mRNA stability give rise predominantly to 13S and 12S E1A mRNAs at 5 h pi (references)”.

      Fig. 1D: −difference in accumulation of viral transcripts is not that visible as in IF staining (Fig. 1B; Fig. 1S);

      Fig. 1 or S1 Fig. do not show IF staining but signals from FISH.

      −graph does not show any difference between E1A and E1B-55K

      AU: The y-axes values in Fig.1D graph are arbitrary units and thus E1A and E1B-55K graphs are not directly comparable to each other. We have included into the revised manuscript S1B Fig., which shows quantification of E1A and E1B-55K fluorescent puncta per cell at the 5 h pi; the difference between E1A and E1B-55K was statistically significant.

      Fig. 1F: −figure legend does not fit with labelling of IF images and continuous text −description says 22 h, while IF labeling and text (p. 7, last lane) mentions 23 h pi

      AU: The figure annotations state the time of analyses as total time after virus addition to cells, whereas text stated the time of analyses as x h post virus removal since we wanted to stress that the input virus was incubated only for 1 h with the cells. However, Reviewers found this confusing, so we have changed the text in the revised manuscript so that time of analysis is stated as total time after virus addition to cells (as in the figure annotations). Only in the Material and Methods section we maintain the original 1 h + x h statement for the time of analysis.

      Fig. 2A: −figure legend: lane 5 Punctuation wrong: azide-Alexa Fluor488. Alexa Fluor647

      AU: Thank you for pointing this out. It has been corrected.

      Fig. 4A: −difficulties to understand −author stated that promoter-driven EGFP expression is clearly dominated by G1 cells for E1A and by S/G2/M cells for CMV, however this is not clearly visible in the graph −no severe differences visible between CMV-eGFP and E1A-eGFP −author should include numbers for quantification and statistical calculations to illustrate the differences

      AU: In the highest GFP expression bin, CMV-eGFP expressing cells have 43% cells in G1 and 50% in S/G2/M (n=2149). In comparison, E1A-GFP expressing cells have 58% cells in G1 and 35% in S/G2/M (n=2258). The difference in G1 cells in the highest eGFP bin is statistically significant (p

      Fig. 4B: −amount of E1A protein levels calculated via IF (signal intensities) −immunofluorescence is not a suitable tool for protein quantification

      AU: It is true that not all antibodies are suitable for IF (or for Western blot), and we cannot be certain that the monoclonal anti-E1A antibody used by us detects all E1A forms with different post-translational modifications with equal efficiency. However, IF is a widely accepted method to estimate protein levels in the cell, especially if the proteins like E1A accumulate in the nucleus (makes segmentation of the signal easy) and give a rather uniform nuclear staining pattern.

      Fig. 5: −in A. it is stated, that E1A bDNA -FISH is not suitable, since it is too short to be detectable. However, in B E1A bDNA-FISH is used. is there a difference? −according to the method part just one E1A mRNA was used for the assays, why is it then not possible to use that one in Fig. 5A? −explanation of the procedure and the experiment is very confusing

      AU: The Reviewer probably refers to Fig.6 here, not to Fig.5. The E1A introns are short (about 100 bases) and cannot be picked up with bDNA FISH probes. In Fig. 6B we were using the E1A bDNA-FISH probes, which were made against the AdV-C5 genome map positions 551-1630 to detect vDNA single strands of the E1A region and these single strands were long enough to be picked out by our E1A probes.

      Fig. S6B: −authors want to show that it is RNase-insensitive, but S1 nuclease-sensitive

      −two different A549 cell clones and two different time points are used for the treatments → not compareable to each other

      AU: This is a fair criticism. We have replaced the RNase A figure in S6B Fig. in the revised manuscript. The new RNase A experiment was carried out in ATCC A549 cells using the same infections conditions as with the S1 nuclease-treated cells.

      Material and Methods: −headings do not indicate which methods are explained −no clear structure AU: We have made minor changes to the headings of Material and Methods section. We have first explained in detail the bDNA-FISH method, but otherwise the order is according to the order of the figures.

      Reviewer #2 (Significance (Required)):

      highly significant manuscript very important for the virology field

      my research topics are human adenoviruses and their replication cycle

      Reviewer #3 (Evidence, reproducibility and clarity (Required)):

      **Summary:** Soumalainen et al have studied adenovirus viral gene expression and replication at a single-cell level. They explore the extent of correlation between incoming genome copy number and early gene expression and progression into the late phase, revealing substantial variation between cells in the numbers of E1A transcripts (the first gene expressed upon infection) that is not explained by differences in the numbers of viral genome templates in the cells. They also explore the relevance of cell cycle stage to this variability and show a positive correlation between G1 cell cycle stage and higher levels of gene activity, which explains at least part of the variation. To form these conclusions they have applied new methods to visualise and quantify single molecules of nucleic acid in single cells. The experiments are all carefully and fully described with full detail of materials. Overall the manuscript is well written and easy to follow.

      **Major comments:**

      All of the experiments appear to be done with rigour and their results reported with due regard to statistical significance etc. My major concern though is that they have been done, perhaps out of necessity to get detectable signals, at very high multiplicities of infection. A well-accepted standard to achieve infection of all cells in a culture is an MOI of 10 infectious units per cell. Even this is acknowledged not to represent the biology of natural infection and it is striking that, where technically feasible, lower MOI studies are more revealing of how a virus actually works. Here, the authors have used counts of particles rather than infectious units to determine MOI and for Ad5, the particle/pfu ratio is typically 20-100. Their MOIs though are 13,000 - 50,000 per cell, implying an infectious MOI of at least 130 for their A549 experiments, which are known to be readily infected by Ad5 from other work.

      AU: Unlike common experiments done by others, we used a synchronized infection and removed the input virus after 1h incubation at 37°C. This type of infection initiation requires high input virus amounts, as opposed to studies in which the virus inoculum is incubated with cells for several hours/days, as is typically done in studies determining the infectious or plaque forming units in virus inoculum. Hence, the MOI used by others involved incubation of inoculum with cells over extended periods of time, and they cannot be compared to our pulsed infection conditions.

      Although the calculated theoretical MOIs (physical particles/cell) were high in our experiments, only 0.1% – 0.2% of input virus particles bound to cells during the 1h incubation period (Fig. 1 A; this estimation is based on the ratios between Median values for the number of cell-associated viruses vs input virus numbers).

      Furthermore, in the experiment described in Fig.2C (correlation of E1A transcripts per cell vs. viral genomes per cell), 42% of analyzed cells had ≤ 5 viral genomes/cell and 27.5% of analyzed cells had between 6-10 viral genomes per cell. Please note, that these are not high numbers.

      The input virus amounts used were selected this way, because we aimed at getting a broader view of how virus transcription at early phases of infection responds to a varying number of virus genomes delivered to the nucleus. Therefore, we did not limit the analyses to a situation with 1 or less than 1 virus particles/genomes per cell.

      In addition, the analyses of how cell cycle phase impacts the initiation of virus gene expression requires a relatively short time between virus inoculation and time point of analysis (i.e. a rather high MOI). Otherwise, as also pointed out by the Reviewer, the cells could have experienced more than one cell cycle phase during the duration of the experiment. Furthermore, although the initial natural infection probably starts with a very low MOI, the second round of infection is a high MOI infection due to a large number of progeny virus particles released from an infected cell.

      Surprisingly, the authors do not see intracellular vDNA copy numbers that are fully reflective of this high MOI, with median intracellular vDNA of 75 /cell at the highest MOI. The authors should consider how the population distribution of vDNA /cell does or does not fit the predicted Poisson distribution. Nonetheless, at these high copy numbers / cell, there must surely be a risk that the variation in gene expression activity arises stochastically, out of competition between genomes for essential transcription factors. Given that multiple cellular factors are each required for E1A transcription, high genome copy numbers could actually inhibit E1A expression relative to cells with more modest copy numbers because limited supplies of individual factors are recruited to different viral genome copies.

      AU: The “discrepancy” between theoretical MOI and the actual observed number of cell-associated virus particles or cell-associated virus genomes is explained above. Furthermore, we would like to point out that we have directly estimated the number of virus particles bound to cells with the input virus amounts used, something that is usually not done in other studies.

      It is indeed theoretically possible that high nuclear genome numbers could lead to inhibition of transcription due to competition for limiting essential host factors. However, if we included only cells with ≤4 vDNA molecules per nucleus into the analysis (total number of cells analyzed was 258), then Spearman’s correlation coefficient for vDNA per nucleus vs E1A mRNAs per cell was 0.186 (p=0.0027). Thus, this would not support the notion that cells with moderate nuclear vDNA copy numbers would have a better correlation between the nuclear vDNA copies vs E1A mRNA counts per cell.

      The vDNA/cell in Fig.2C does not fit predicted Poisson distribution, var/mean=9.129.

      It is important for the analysis of correlation of gene expression with cell cycle that the virus has not, at the time point analysed, already perturbed the cell cycle (a well-known effect of infection) which the authors document in Suppl Fig3B. To my eye, the G1 peak in infected cells is somewhat narrower than in the control while the S/G2 bump is a little greater. The % of cells in each of the two gates needs to be shown to support the conclusion.

      AU: In non-infected sample G1= 54.63% and S/G2/M = 45.37%, in infected cells G1= 51.4% and S/G2/M= 48.6%. We have added this information into the S3B Fig.

      Turning to the experiments documenting a correlation between E1A expression and cell cycle stage, the authors interpret their findings in terms of the stage the cells are at when the analysis was done (G1 stage cells have more E1A transcripts). The key experiment (Fig 3B) is analysed at only 4 h pi, so substantial progression from G2/M back to G1 after virus addition can probably be discounted, but the point should be discussed. The authors also use release from G1 in another cell line to support their argument that G1 supports higher levels of E1A expression (Fig 3C). Here, they elect to exclude all cells with fewer than 50 E1A transcripts from their analysis. The reason for this is completely obscure and isn't obviously justified; conceivably it could bias the outcome of the experiment. At minimum, this decision needs to be carefully explained; ideally, the full data set should be used.

      AU: Fig.3B: As suggested by the Reviewer, we have added to the main text the following explanation: “We used a high MOI infection (median 75 cell-associated virus particles, Fig. 1A) in order to achieve a rapid onset of E1A expression so that the time between virus addition and analysis was short. Thus, it is not expected that a substantial number of cells would have changed their cell cycle status during the experiment.”

      Fig.3C: We show the results also from the full data set of infected cells, i.e., cells with ≥ 1 E1A puncta in S3D Fig. We excluded the cells without zero E1A puncta because with these cells it is impossible to know whether they received no virus or whether E1A transcription had not yet started. Permutation test indicated that the difference between the starved+starved and starved+FCS is statistically significant even in this case. Because both samples are dominated by cells with low E1A counts, we log-transformed the E1A values for the box plot figure.

      The authors note the highest level of E1A activity (as opposed to RNA) was in G1/S cells and suggest that high E1A cells advance preferentially into S. Whilst in line with the literature that E1A promotes progression into S, an alternative explanation is simply that there is a time lag between RNA accumulation and protein accumulation, during which progression through the cycle would be expected.

      AU: This is a valid point, and we have modified the text as follows: “… which could reflect the advancement of high E1A expressing cells into S-phase. However, considering the time between virus addition and analysis (10.5 h), we cannot exclude the possibility that the observed G1/S preference is at least partly due to time-dependent progression of G1 cells to G1/S.”

      **Minor comments:** Fig 1 and elsewhere. Given that the 1 h incubations with virus were done at 37 C, the convention would be to include this period in the time post-infection at which harvest / fix time points are quoted. There is inconsistency between text and legend with 12 h pi being sometimes represented as 11 h after virus removal; this is an unnecessary confusion.

      AU: We have modified the text so that hours pi always include the 1h incubation with the input virus. Only in the Material and Methods section we kept the original 1h virus binding – fixing at xh post virus removal.

      Results description prior to the ref to Fig 1B: unclear what this is supposed to mean.

      AU: We have now slightly modified the first paragraph of the Results section. We mention the benefits of the bDNA signal amplification method and explain the experimental set up, i.e. that the input virus was incubated with the cells only for 1h. We also justify why we used a short incubation for the virus inoculum.

      Fig 4A: provide % of cells in each gate in each histogram.

      AU: In the highest GFP expression bin, CMV-eGFP expressing cells have 43% of cells in G1 and 50% in S/G2/M. In comparison, E1A-GFP expressing cells have 58% of cells in G1 and 35% in S/G2/M. This has been added to the figure, and it is also mentioned in the main text. Furthermore, we added to the text the results from Two Proportion Z-test to show that the proportion difference of G1 cells in the highest bin was statistically significant (p

      Fig 5: bottom right panel x axis label is wrong

      AU: Thank you for pointing out this. This has been corrected.

      In the presentation of Fig 6, it would be much clearer for the reader if the detected replication foci (ss DNA detected as E1A puncta) were referred to as something other than E1A puncta. There is too much scope for confusion with the earlier experiments in which E1A RNA was detected.

      AU: We agree. In the revised manuscript, we refer to these puncta in the text as E1A ssDNA-foci.

      Reviewer #3 (Significance (Required)):

      The study represents the application of state of the art single-molecule visualization techniques to an as yet not understood aspect of virus infection. That said, there is prior experimentation in this area, which the authors fully acknowledge and build upon. The new work is largely descriptive, in that it reveals very clearly the discrepancy between genome copy number and amounts of mRNA without seeking to explain these, beyond the cell cycle analysis. Whilst there is a better correlation between vDNA number and transcript once the data are stratified by cell cycle stage, it is still not strong (Fig 5), indicating that other substantial contributing factors remain to be described.

      The work will be of interest certainly to adenovirologists, but also to others who study virus infections - particularly nuclear-replicating DNA viruses such as herpesviruses - where similar considerations are likely to apply.

      Expertise: adenovirus; gene expression; virus-host interactions; molecular biology

    1. Coronavirus deaths are falling in the US even as cases skyrocket. In the UK, a lower proportion of people hospitalised with covid‑19 are dying. This has led to suggestions that the risk of dying if you are infected with the virus is falling, but the truth may be more complicated. “At this point, I don’t think we have conclusive evidence that the death rate is going down,” says Tessa Bold at Stockholm University in Sweden. Having plateaued at around 20,000 in May, the number of daily confirmed cases in the US began rising in June and has now exceeded 60,000. However, the number of deaths in the US reported as being due to covid-19 has fallen from more than 3000 a day in mid-April to well under 1000.
  5. www.eastbayexpress.com www.eastbayexpress.com
    1. "Part of me feels compelled to advocate for stories about Black historical figures that people may have never heard of."

      In my English class we talk and I learned a lot about them. How they suffered as slaves. I think that English classes talk a lot about them because sometimes we do not know the background of their lives and the injustices they suffered

    1. Donald Graves has argued that by treating students’ writing primarily with correction and judgment, instead of coaching them graciously and sup-portively, we have created a society of people who treat a writing occasion as if they have been invited to an uncomfortable formal dinner. They arrive late, hope not to be noticed, and leave early.

      I think a lot of teachers rush to correct and judge because of time constraints. The syllabus is packed with activities and assignments, and they may feel like they're falling behind, so they overwhelm and discourage students with too much feedback on early drafts hoping they can move on quickly, but good writing cannot be rushed. For this reason, it's important not to overload the syllabus and to allow time for teachers and students to go step by step through the writing process.

    1. Although it is not simple to determine whether the students in this case were exposed to autotelic experience (Csikszentmihalyi, 1990), according to which learning becomes moti-vating by itself, it is possible to depict that the students did not perceive this educational experience as demanding. Thus, transmedia features and gamification contributed to the engagement achieved by the two classes involved in the process. Furthermore, within the concept of participatory learning culture, education is called to foster skills and motivate students to learn instead of teaching certain facts. As learners become responsible for the learning process, motivation becomes one of the most pivotal purposes of educational projects.

      The author's disclosure statement answered my question about any money trail to them, at least that they reported. I too am a product of the learning should be fun thought world. This I suggest is only marginally important if it does not lead to one understanding that deep learning requires a commitment to excellence and embarking on an impassioned lifelong inquiry. Phenomenon approaches if rooted in phenomenology may be similar to this, I do not think they are, but I would not rule out-of-hand the Finnish instantiation being topically grounded. What I suspect is different is not commercializing the educational tools. Have we succumbed in the United States to making everything about profit and not the bliss that comes from the love of learning? I guess one stays tuned-in for the next engaging opportunity.

    2. Although gamification has been applied as a technique throughout the course of history, the term is relatively new.

      Gamification may be new to Nick Pelling but it builds on strategies that came into the public sphere much earlier. Robert McNamara used the term we gamed the strategy in the early 1960s and the concepts go back much further than him. They were breaking down actions into component parts long before the dawn of the Einiac. Gamification, gamed the system, playing the game of life are part of the same intellectual phenomenology of dissecting, analyzing a situation to its fullest and then playing what if strategy games for contingency plan development. Is the newest part its mass-dissemination? Again, I think not. I am exceedingly curious if the authors have a monetary interest in this specific curriculum? This is a crass question and for that I am sorry but this reads like an slightly intellectual sales pitch.

    1. Note: This rebuttal was posted by the corresponding author to Review Commons. Content has not been altered except for formatting.

      Learn more at Review Commons


      Reply to the reviewers

      General comments

      We thank all three reviewers for providing their thoughtful and insightful review comments of our manuscript. We appreciate that the reviewers recognized the significance and impact of our work - “Very little imaging has been done on CAR synapses and to our knowledge this is the first live cell imaging study describing CAR microclustsers” (Reviewer 2); “This is an evolving field and little is known to date. Hence, this study could represent an insightful and important advance to the field” (Reviewer 3). A broad audience from both basic and clinical research sides will be interested in this work: “_This study will have a broad audience. Both scientists that study basic T cell signaling as well as clinicians that use CAR Ts will be interested in this study” (_Reviewer 2); “Audience is to both basic immunologist and cancer biologists” (Reviewer 3).

      Meanwhile, we understand that the reviewers have raised a few major and minor issues, which we attempted to address. Most importantly, as suggested by both reviewer 1 and 3, we performed new experiments showing that LAT is not required for microcluster formation of the 1st generation of CAR (new Fig 4 and EV5). This finding suggests that the CAR-independent signaling is due to the intrinsic CAR architecture, and is not dependent on the co-signaling domains of CD28 and 4-1BB.

      With the successful solutions to other issues, we believe the manuscript has been significantly improved and is ready for publication. Below we will provide point-to-point responses to each reviewer’s comments.

      Reviewer #1 (Evidence, reproducibility and clarity (Required)):

      The authors compare the TCR alone to a CAR that contains signaling modules from three receptors- TCR, CD28 and 41BB. The data quality if good and the experiments done are. The difference is quite clear, and I would even like to see a little more of the evidence related to failure of the TCR system.

      We appreciate the general positive comment of this reviewer.

      More specifically:

      Su and colleagues show that a third generation CAR with TCR zeta, CD28 and 41BB signal transduction pathways can activate a T cell for microcluster formation and Gads/SLP-76 recruitment, but not IL-2 production, without LAT. This is surprising because LAT is generally considered, as is up held here, as an essential adapter protein for T cell activation. However, this is not a "fair" experiment as the CAR has sequences from TCR, and two co-stimulatory receptor- CD28 and 41BB. It would be important and very straight-forward to test first and second generation CARs to determine if LAT independence is a function of the CAR architecture itself, or the additional costimulatory sequences. If it turns out that a first generation CAR with only TCR sequences can trigger LAT independent clustering and SLP-76 recruitment then the comparison would be fair and no additional experiment would be needed to make the point that the CAR architecture is intrinsically LAT independent. If the CD28 and/or 41BB sequences are needed for LAT independence then the fair comparison would be to co-crosslink TCR, CD28 and 41BB (an inducible costimulator such that anti-CD27 might be substituted to have a constitutively expressed receptor with this similar motifs) should be cross-linked with the TCR to make this a fair comparison between the two architectures.

      We agree with the reviewer that it is critical to make a “fair” comparison between TCR and CAR by testing the 1st generation CAR, which only contains the TCR/CD3z domain. Our new data showed that LAT is not required for microcluster and synapse formation of the 1st generation of CAR, in both Jurkat and primary T cells (new Fig 4 and EV5). This result is similar to our previously reported result from the 3rd generation CAR, although the 1st generation CAR induced less IL-2 production and CD69 expression in LAT null cells than the 3rd generation CAR did (new Fig 6). This suggests that the LAT-independent signaling is intrinsic to the CAR architecture, as the reviewer suggested. The co-signaling domains from CD28 and 4-1BB contribute to, but are not required for bypassing LAT to transduce the CAR signaling.

      The authors may want to cite work from Vignali and colleagues that even the TCR has two signaling modules- the classical ZAP-70/LAT module that is responsible to IL-2 and a Vav/Notch dependent module that controls proliferation. Its not clear to me that the issue raised about distinct signaling by CARs is completely parallel to this, but its interesting that Vignali also associated the classical TCR signaling pathway as responsible for IL-2 with an alterive pathways that uses the same ITAMs to control distinct functions. See Guy CS, Vignali KM, Temirov J, Bettini ML, Overacre AE, Smeltzer M, Zhang H, Huppa JB, Tsai YH, Lobry C, Xie J, Dempsey PJ, Crawford HC, Aifantis I, Davis MM, Vignali DA. Distinct TCR signaling pathways drive proliferation and cytokine production in T cells. Nat Immunol. 2013;14(3):262-70.

      We appreciate the reviewer’s mentioning this paper from Vignali’s group. It provides insights into understanding LAT-independent signaling in CAR T cells. We cited this paper and added a discussion about the mechanism of LAT-independent signaling.

      I would be very interested to see a movie of the LAT deficient T cells interacting with the anti-CD3 coated bilayers in Figure 2A. Since OKT3 has a high affinity for CD3 and is coated on the surface at a density that should engage anti-CD3 I'm surprised there is no clustering even simply based on mass action. The result looks almost like a dominant negative effect of LAT deficiency on a high affinity extracellular interaction. It would be interesting to see how this interface evolves or if there is anti-adhesive behavior that emerges.

      We now presented a movie showing the detailed process of LAT deficient GFP-CAR T cells landing on the bilayers coated with OKT3 (new Movie EV5), in which the bright field images delineate the locations of the cells, the OKT3 signal marks TCR, and the GFP signal marks CAR proteins on the plasma membranes. No TCR clusters (as indicated by OKT3) were formed during the landing process. We think the binding of bilayer-presented OKT3 to TCR is not sufficient to trigger TCR microclusters. However, TCR microclusters could form in LAT-deficient cells if OKT3 is presented by glass surface. This point is raised by reviewer 2. We added a discussion on the difference between bilayer and glass-presented OKT3 in inducing microcluster formation.

      Reviewer #1 (Significance (Required)):

      While it interesting that the CAR is LAT independent, its obvious that the signalling networks are different as the CAR has two sets of motifs that are absent in the TCR, so the experiments as presented are not that insightful about the specific nature of the differences that lead to the different outcomes. At present its not a particularly well controlled experiment as the third gen CAR is changing too many things in relation to the TCR for the experiment to be interpreted. It would be easy to address this is a revised manuscript. To publish as is the discussion would need to acknowledge these limitations. The work is preliminary as science, but it might be useful to T cell engineering field to have this information as a preliminary report, which might be an argument for adding discussion of limitations, but going forward without more detailed analysis of mechanism.

      This is an excellent point and we have addressed it. See our response above on the new data of the 1st generation CAR.

      Reviewer #2 (Evidence, reproducibility and clarity):

      Summary:

      Provide a short summary of the findings and key conclusions (including methodology and model system(s) where appropriate).

      In this study, the authors have interrogated CAR signaling by imaging CD19-CAR microclusters as well as T cell signaling molecules recruited to CAR microclusters. They report differences spatial assembly between CAR and TCR microclusters that form on a lipid bilayer containing ligand. They also report that LAT is not required for CAR microcluster formation, recruitment of downstream signaling molecules or IL-2 production in Jurkat cells, while in primary T cells IL-2 production by CARs show more of a LAT dependence. From these observations, they conclude that CAR T cells have a rewired signaling pathway as compared to T cells that signal through the TCR.

      Major comments:

      • Are the key conclusions convincing?

      The conclusions made by the authors about CAR microclusters are convincing. However, the conclusion that there is a "rewired signaling network" different from TCR microclusters needs to be more convincingly demonstrated in side-by-side comparisons of TCR and CAR microclusters and synapses.

      1. One of the key conclusions in this study is that CAR microclusters form in the absence of LAT, but TCR microclusters require LAT (in JCam2.5 cells in Fig. 2 and primary T cells in Fig. 4B). The requirement of LAT for formation of TCR microclusters is surprising, given multiple reports (one of which the authors have cited) that TCRz and ZAP70 clusters form normally in the absence of LAT (pZAP microclusters form normally in JCam2.5 cells Barda-Saad Nature Immunology 2005 Figure 1; TCRz clusters form normally in LAT CRISPR KO Jurkat cells Yi et al., Nature Communications, 2019 Figure 5). The authors should carefully evaluate TCRz and ZAP70 clusters (that form upstream of LAT) in their assays.

      We thank the reviewer for raising this excellent point. LAT-independent TCR clusters were reported in the two papers mentioned by the reviewer, which we think is convincing. However, there is a key difference in the experimental settings between these two papers and ours. We use supported lipid bilayer to present MOBILE TCR-activating antibody to activate T cells, whereas these two papers used IMMOBILE TCR-activating antibody attached to the cover glass. We reasoned that the mobile surface of supported lipid bilayer more closely mimics the antigen-presenting cell surface where antigens are mobile on the membrane. We added a new discussion about the difference between supported lipid bilayer and cover glass-based activation.

      We agree with the reviewer on the careful evaluation of TCR and ZAP70 clusters. We had showed the data of TCR clusters as marked by TCR-interacting OKT3 (Fig 3A). We performed new experiments on ZAP70 clusters (new Fig EV3). Our data suggest that, similar to TCR clusters, ZAP70 clusters are not formed in LAT-deficient T cells, if activated by OKT3, but are formed if activated by CD19.

      1. The authors make major conclusions about LAT dependence and independence of TCR and CAR microclusters respectively, by using JCam2.5 Jurkat cells and CRISPR/Cas9 edited primary cells. Of relevance to this conclusion, differences in the phosphorylation status of ZAP70 and SLP76 have been described between JCam2.5 cells lacking LAT (in which LAT was found to be deleted by gamma radiation) and J.LAT cells (in which LAT was specifically deleted by CRISPR/Cas9 in Lo et al Nature Immunology 2018). Of importance, pZAP and pSLP76 appeared fairly intact in J.LAT cells, but absent in JCam2.5 cells (Lo et al., Nat Immunol. 2018, Supp Fig 2). Therefore, the authors should evaluate TCRz, ZAP70, Gads and SLP76 in TCR and CAR microclusters in J.LAT cells. This may partly explain the discrepancy in LAT requirement for IL-2 production in JCam2.5 cells and primary cells with LAT CRISPRed out.

      Jcam2.5 is a classical well-characterized LAT-deficient cell line that has been continuously used in the T cell signaling field (Barda-Saad Nature Immunology 2005, Rouquette-Jazdanian A, Mol. Cell, 2012; Balagopalan L, J Imm. 2013; Carpier J, J Exp Med, 2018; Zucchetti A, Nat. Comm. 2019). We agreed with the concern that the reviewer raised on the absence of pZAP70 and pSLP76 in JCam2.5 cells. As the reviewer suggested, we obtained J.LAT, which is LAT null but has intact pZAP70 and pSLP76. We introduced CAR into J.LAT and the wild-type control and performed the clustering assay as we did for Jcam2.5. Our results showed that, similar to Jcam2.5, CAR forms robust microclusters in J.LAT cells (new Fig EV2). More importantly, we presented data confirming the LAT-independent CAR clustering, SLP76 phosphorylation, and IL-2 production in human primary T cells (Fig 7). Therefore, the data from three independent cell sources support our conclusion on LAT-independent CAR signal transduction.

      1. Since the authors are reporting differences between CAR synapses and TCR synapses, the authors should show side by side comparison of CAR and TCR synapses in Figure 1F.

      We focused on characterizing CAR synapse in this manuscript and did not make any conclusion on the difference between TCR and CAR synapse. We are cautious about comparing CAR synapse to TCR synapse for technical reasons: it is critical to use antigen-specific TCRs (e.g. mouse OTI as a common model) to study the TCR synapse pattern so that the study will be physiologically relevant. However, we use human T cell line and human primary T cells for the CAR study. The technical barrier to introduce an antigen-specific TCR complex into these cells, and to activate these cells by purified peptide-MHC complex, is very high. And the result is interesting, but beyond the scope of the current work.

      1. The authors should evaluate Gads microcluster formation in response to TCR stimulation via OKT3 (in Figure 4A). Given that it has been reported that TCRz, Grb2 and c-Cbl are recruited to microclusters in Jurkat cells lacking LAT by CRISPR deletion (Yi et al., Nature Communications, 2019), it is important to establish the differences between TCR microclusters and CAR microclusters in side by side comparisons in their assay system.

      As the reviewer suggested, we evaluated Gads microcluster formation with TCR stimulation and found that Gads did not form microclusters in LAT-deficient cells (new Fig 5A). Because we only made conclusions on the Gads-SLP76 pathway, we think investigating Grb2 and c-Cbl microcluster, though interesting, is beyond the scope of this manuscript.

      1. Similar to the comment about Gads above, the authors should evaluate pSLP76 microcluster formation in response to TCR stimulation via OKT3 in primary T cells lacking LAT in Figure 4C, i.e. side by side comparisons of pSLP76 in TCR and CAR synapses (with and without LAT) should be shown.

      We totally agree and performed new experiment on pSLP76 in human primary T cells. Our data suggested that, similar to Jurkat, pSLP76 microclusters remain intact in LAT null primary cells (new Fig 7D and 7E).

      • Should the authors qualify some of their claims as preliminary or speculative, or remove them altogether?
      1. The data shown in Figure 3C shows a reduction in conjugate formation from 80% (WT) to 30% (LAT -). This is a severe reduction and does not support the authors' claim in the corresponding Figure legend that "LAT is dispensable for cell conjugate formation between Jurkat T cells expressing CAR and Raji B cells" and the Abstract that "LAT.....is not required for....immunological synapse formation". Statistical analysis for variance should be shown here.

      We agree with the reviewer’s judgement. This cell conjugation analysis was performed using Jcam2.5 cells. As pointed by the reviewer, Jcam2.5 has additional defects in ZAP70 and SLP76 in addition to the lack of LAT. Therefore, we performed the same analysis again using J.LAT cells, which was recommended by the reviewer. Our new data showed that J.LAT cells form conjugates with Raji B cells in a similar rate as the wild-type cells do, as evaluated by statistical analysis (new Fig 6A). Therefore, we think these new data support the claim that LAT is dispensable for cell conjugate formation.

      1. In a similar vein, based on data from Movie S5 (where in a single cell, CAR microclusters translocate from cell periphery to center), and Figure 3C where (as described above in point 1) conjugate formation appears to be severely reduced, the authors conclude in the Results and Abstract that "LAT....is not required for actin remodeling following CAR activation". This conclusion is not supported by the data and the authors should remove this claim. Alternatively, actin polymerization in CAR expressing cells (that are LAT sufficient and deficient) can be easily evaluated using phalloidin or F-Tractin.

      As suggested by the reviewer, we evaluated actin polymerization in TCR or CAR stimulated cells using a filamentous actin reporter F-tractin. Our data showed that LAT is required for TCR-induced but not CAR-induced actin polymerization (new Fig 5C). Therefore, our results support the claim that LAT is not required for actin remodeling following CAR activation.

      • Would additional experiments be essential to support the claims of the paper?<br> Request additional experiments only where necessary for the paper as it is, and do not ask authors to open new lines of experimentation.

      Yes. Please see major comments above.

      • Are the suggested experiments realistic in terms of time and resources? It would help if you could add an estimated cost and time investment for substantial experiments.

      Yes. It should take 3 months to complete these experiments, since reagents and experimental systems to do these experiments already exist.

      • Are the data and the methods presented in such a way that they can be reproduced?<br> Yes. Methods are clearly explained.

      We appreciate the reviewer’s recognition of the clarity of the methods part.

      • Are the experiments adequately replicated and statistical analysis adequate?

      There is no statistical analysis to evaluate differences between samples in Figures 3 and 4. These must be included.

      We now added statistical analysis in Fig 5B and 6A (old figure 3 and 4).

      Minor comments:

      • Specific experimental issues that are easily addressable.

      Please see Major Comments above. We believe that the recommended experiments are not difficult to execute since reagents exist and experimental systems are already set up.

      • Are prior studies referenced appropriately?

      Authors reference 13 and 14 for the following sentence in Results section 2: "Deletion or mutation of LAT impairs formation of T cell microclusters". However, in Reference 14 Barda-Saad et al., actually show that pZAP clusters are intact in JCam2.5 cells lacking LAT. Perhaps authors should clarify that LAT (and downstream signaling molecule) microclusters are impaired when LAT is deleted or mutated.

      As the reviewer suggested, we now clarified that clustering of LAT downstream binding partners is impaired when citing reference (Barda-Saad et al).

      • Are the text and figures clear and accurate?

      Yes. But would be helpful if authors specify what "control" is in Fig. 3B and C. In Figure 3B it is lipid bilayers without CD19, while in 3C it is K562 cells that do not express CD19.

      We now specified “control” in the figure.

      • Do you have suggestions that would help the authors improve the presentation of their data and conclusions?

      Would be helpful if authors specify in every Figure or at least Figure legend the experimental bilayer system/ligand used, since they use both OKT3 and CD19 as ligands in the paper.

      We now specified the ligand in the figure or legend.

      Reviewer #2 (Significance):

      • Describe the nature and significance of the advance (e.g. conceptual, technical, clinical) for the field.

      If CAR microclusters and synapses are appropriately compared in a side by side comparison with TCR microclusters and synapses (as described in comments above), this study will be a conceptual advance in the field of CAR signaling. CAR microclusters have not been studied previously.

      • Place the work in the context of the existing literature (provide references, where appropriate).

      Very little imaging has been done on CAR synapses and to our knowledge this is the first live cell imaging study describing CAR microclusters.

      We appreciate this reviewer’s comment on our work as a conceptual advance in understanding CAR signaling.

      • State what audience might be interested in and influenced by the reported findings.<br> This study will have a broad audience. Both scientists that study basic T cell signaling as well as clinicians that use CAR Ts will be interested in this study.

      We appreciate this reviewer’s recognition of the broad audience of this manuscript.

      • Define your field of expertise with a few keywords to help the authors contextualize your point of view. Indicate if there are any parts of the paper that you do not have sufficient expertise to evaluate.

      T cell signaling and imaging of proximal T cell signaling responses.

      Reviewer #3 (Evidence, reproducibility and clarity):

      This manuscript by Dong and colleagues characterizes the molecular requirements and consequences of engaging a third-generation chimeric antigen receptor (CAR) directed to CD19. Utilizing a biological system of JCaM2.5, a Jurkat T cell mutant with dramatically low levels of LAT, expressing a CAR directed to CD19 fused to the cytoplasmic tails of CD28, 4-1BB and CD3z that is activated by CD19/ICAM1 reconstituted lipid bilayers, the authors demonstrate LAT is not required for microcluster formation, immunologic synapse formation or recruitment of GADS and pSLP76 to the plasma membrane. In contrast, LAT was required for anti-CD3 mediated microcluster formation and pSLP76 recruitment to the plasma membrane. However, LAT does appear to contribute to efficient synapse formation, PIP2 hydrolysis and IL-2 secretion when CAR+ JCaM2.5 or primary T cells are presented with Raji B cells, respectively. These data provide intriguing insights into the molecular requirements for third-generation CAR-T cell functions. The authors have developed quite a nice system to understand the molecular contributions for CAR-T function. A few suggestions are provided here to further enhance the accuracy and significance of the findings:

      1. The authors can address whether the LAT-independent effects are due to the attributes of third generation CAR-Ts with inclusion of CD28 and 4-1BB cytoplasmic domains or whether these differences are intrinsic to all CAR-Ts (e.g., first and second generation CARs).

      This is an excellent point. We have included new data showing LAT-independent cluster formation of the 1st generation CAR in both Jurkat and primary T cells (new Fig 4 and EV5). Therefore, we favor the second possibility as pointed by the reviewer that LAT-independent effects are intrinsic to CAR architecture.

      1. Since a first-generation CAR-T forms non-conventional synapses (Davenport, et al., PNAS 2018), the authors should consider more detailed kinetic analysis to understand the formation and dissolution of the constituents of the synapse with their third generation CAR. This should include measurements of the duration of microcluster and synapse formation as well as further analysis of c- and p-SMAC constituents (e.g., LFA-1, TALIN, LCK and pSLP76) over time.

      We agree with the reviewer on a more detailed characterization of the CAR synapse. We measured the duration of the unstable CAR synapse and time from cell landing to the start of retrograde flow (new Fig 2C). We also determined the localization of CD45, a marker for d-SMAC (new Fig 2D). We found that the formation of dSMAC is also not common in CAR T synapse, strengthening our conclusion that CAR forms non-typical immunological synapse.

      1. The authors utilize two different activation platforms. While using CD19/ICAM1 reconstituted bilayers, CAR+ JCaM2.5 or CAR+ primary T cells demonstrate no differences compared to wildtype JCaM2.5 cells in the parameters studied. However, when using Raji B cells, the CAR+ JCaM2.5 cells or CAR+ primary T cells demonstrate a more intermediate phenotype with respect to cell conjugate formation (Figure 3C) and IL-2 production (Figure 4D). The authors should analyze whether the differences attributed to the different outcomes may be due to the stimulation mode. For example, is c-SMAC assembly and GADS or pSLP76 recruitment to the plasma membrane still LAT-independent when activated with Raji B cells?

      As the reviewer suggested, we examined c-SMAC assembly in Raji B cells conjugated with CAR T cells. We found that the majority of CAR do not form cSMAC (new Fig EV4), which is consistent with the result from the bilayer activation system. Since both Gads and SLP76 are cytosolic proteins, they keep largely in the cytosolic pool which obscures their recruitment and clustering on the plasma membrane when imaged by confocal microscopy at the cross-section of cell-cell synapse.

      1. The authors should consider whether CAR expression level affects their observations. For example, do lower levels of CAR expression make the system LAT-dependent? Further, what is the level of the CAR relative to endogenous TCR expression on their primary T cells.

      We agree with the reviewer that it is informative to determine if LAT-independent signaling is dose dependent. We tried to measure the CAR concentration relative to the endogenous TCR/CD3z. By western blot using two different antibodies against CD3z, we detected TCR/CD3z expression, but found no bands corresponding to CAR. We believe this reflects a low expression of CAR in our system, which is confirmed by FACS. The general low expression of CAR makes it challenging to sort an even lower CAR-expressing population. Therefore, we sought alternative ways to determine the dose-dependence; we titrated the CD19 concentrations on the bilayer. As shown in the new Figure EV1, CAR formed microclusters similarly in the wild-type versus LAT-deficient cells in a wide range of CD19 concentration. Therefore, we conclude that the LAT-independent cluster formation is robust at low antigen density as well.

      Minor comment:

      1. Since JCaM2.5 has differences when compared to the parental Jurkat E6.1 T cell line, the authors should utilize JCaM2.5 reconstituted with wildtype LAT as a comparator.<br> Agreeing with this reviewer, we recognized that Jcam2.5 was generated by mutagenesis which may result in protein expression difference for genes besides Lat. As suggested by reviewer1, we used J.LAT, a genuine LAT knockout cell line that is generated by CRISPR-mediated gene targeting, to perform the clustering assay (new Fig EV2). Our results showed that, similar to Jcam2.5, CAR but not the TCR formed microclusters in J.LAT cells.

      Reviewer #3 (Significance):

      The mechanism(s) by which CAR-Ts function is of high significance from both scientific and clinical viewpoints. From a scientific viewpoint, it provides important basic mechanistic information of how T cells are being activated to kill tumor cells. By understanding the molecular requirements, additional generations of CARs can be designed to provide greater efficacy, overcome resistance and possibly less toxicity.

      This is an evolving field and little is known to date. Hence, this study could represent an insightful and important advance to the field.

      Audience is to both basic immunologist and cancer biologists.

      We appreciate this reviewer’s comments on the high significance of our work to the field of both basic immunology and clinical application.

      My expertise is in T cell signaling, T cell biology and immunotherapy.

    1. Wholly new forms of encyclopedias will appear, ready made with a mesh of associative trails running through them, ready to be dropped into the memex and there amplified.

      This sounds a lot like the personal knowledge management apps we have today, like Notion and Roam Research.

    2. Our ineptitude in getting at the record is largely caused by the artificiality of systems of indexing. When data of any sort are placed in storage, they are filed alphabetically or numerically, and information is found (when it is) by tracing it down from subclass to subclass. It can be in only one place, unless duplicates are used; one has to have rules as to which path will locate it, and the rules are cumbersome. Having found one item, moreover, one has to emerge from the system and re-enter on a new path.The human mind does not work that way. It operates by association.

      This describes why the bidirectional links in note-taking apps like Roam Research are so useful.

    3. The machines for higher analysis have usually been equation solvers. Ideas are beginning to appear for equation transformers, which will rearrange the relationship expressed by an equation in accordance with strict and rather advanced logic.

      Mathematica is probably one of the best example of this.

    1. John Berger in episode two of Ways of Seeing, he discusses the difference between nude and nakedness for a woman. He explains that nakedness is just the human female body without clothes. An appropriate example would be a woman in the shower by herself is naked. Nude is when there is an audience there to judge and sexualize the body of a female. This is significant in society as it is a reflection of inequality of women and how women are seen and viewed in western culture. I think this applies equally today as it did during the renaissance, or during the 70’s when this video was produced. I think we have substituted photography for the paintings. As he mentioned in his previous episode the camera, and as time has progressed technology has somewhat changed the perspective of the nude. I think the internet and social media has created a new version of the nude, that encompasses women who are not even naked. They may have a bikini or just a revealing outfit that is meant to entice the male sexuality, but the result is the same. The images have stripped that person of who they are, and replaced their identity with an image “that is first and foremost a sight to be looked at” as Berger states in his video.

      Excellent answer!

    1. While school closures may be necessary to slow the spread of the virus, they can adversely affect both parents, who might have to take off work to care for their child, as well as students, particularly low-income students, who rely on school meals for lunch.

      Many times we think about social distance, but we dont really think about kids who rely on lunch to have a proper meal for the day

    1. Note: This rebuttal was posted by the corresponding author to Review Commons. Content has not been altered except for formatting.

      Learn more at Review Commons


      Reply to the reviewers

      This preprint has been reviewed by subject experts for Review Commons. Content has not been altered except for formatting.

      Response to Reviewers

      We are grateful to the Reviewers for their thoughtful and helpful assessment of our work. Below we include a point-by-point response to the Reviewers' critiques concerning the interpretation of our results and the power of our system to elucidate key dynamics of fission yeast homology-directed repair (HDR). We appreciate that the Reviewers judged our assay to be a valuable new tool for studying DSB repair in S. pombe. In general, the Reviewers also felt that our data provides new insights into homology search during HDR in fission yeast, including 1) that multiple DSB-donor encounters often precede repair and 2) that the activity of the helicase Rqh1, which dissolves strand invasion structures, alters the kinetics and efficiency of HDR in S. pombe. The Reviewers also raised several concerns with regards to 1) some technical aspects of the experimental approach, 2) the display of the data, and 3) the interpretation of the data. The Reviewers requested additional experiments to address the efficacy of our 5 minute observational time window and the rate of spontaneous damage in the Rqh1 null background, which we are able to provide in a resubmission. We will also clarify experimental details that the Reviewers found confusing in the original text. Lastly, the Reviewers highlighted minor needed figure adjustments that we will incorporate.

      Point-by-point Response:

      *Reviewer #1 (Evidence, reproducibility and clarity (Required)):

      Vines et al adapted a system that has been used in S. cerevisiae to study the homology search and homologous recombination repair events by live cell imaging. The authors utilized a system they set up in a fission yeast strain that has a fluorescently tagged endonuclease induced DSB site and monitored RAD52 focus formation in both haploid and diploid cells. The main findings presented are that multiple strand invasion events occur during DSB repair and the role of Rqh1 in promoting these multiple events. For example, cells with Rqh1 loss either have a single strand invasion event that quickly leads to repair or a very long extensive repair time. Overall the results are intriguing with new insight into DSB repair being presented.*

      We appreciate the Reviewer’s recognition that our work provides new insights into homology-directed repair (HDR) in fission yeast.

      The manuscript would benefit from having another system to help to support or validate the key findings and/or the use of some mutants to help uncouple the different roles of Rad51 and/or Rqh1.

      While we agree with the Reviewer that using orthogonal approaches is always desirable, it is not clear what other experimental platform can address the dynamic events with single cell resolution that underlie our observations here; indeed, this was the motivation behind designing this new approach. However, we will provide additional, detailed context to support our findings in the revised manuscript that highlights how orthogonal experimental strategies (e.g. DSB repair outcome assays) already in the literature (e.g. Hope et al., PNAS, 2006) are consistent with our findings. Importantly, however, there is no other population-based system we are aware of that could demonstrate, for example, that Rqh1 shows two different behaviors in individual cells (repair failure and more rapid repair). See more in response to comment 7, below.

      \*Major comment:**

      1) In Figure 1C, and also Figure 2D, the RAD52 focus observed does not appear in the same location as the LacO cassette. I assume this is because of the way the images are cropped. It would be nice if the authors are saying that the RAD52 focus co-localizes with the inducible DSB location for this to be more readily apparent in the representative images. *

      Co-localization events, indicated with the yellow circles, are assessed within raw 3D data that is then flattened for representation in 2D in the figures. For Figure 1C, the two events in the example cell indeed overlap in 3D space. However, in Figure 2D (cells lacking Rad51) we do not observe any colocalization events in the example (and there are no time points annotated with yellow circles).

      2) In Figure 3A, the authors claim that the mean time to repair an endonuclease induced DSB is 50 min +/- 20 min. It is unclear whether or not this experiment is done in a diploid strain.

      We apologize if we were not clear. All experiments presented in the manuscript are carried out in diploid cells. What varies is whether there is a lac operator integrated at one copy of Chr II (all experiments except Fig. 2A) or on both copies (only Fig. 2A). This will be clarified in the revised text.

      3) In Figure 3, whether or not this experiment represents asynchronous cells can greatly influence the timing of DSB repair, as the cell cycle is a huge contributor to HDR repair.

      We agree with the Reviewer - the cell cycle has a critical influence on DSB repair mechanism. The diploid fission yeast in which we induced and observed DSBs are indeed asynchronous. However, in fission yeast, which spend over 80% of their cell in G2, we can assess cell cycle by morphology; cytokinesis coincides with the beginning of G2, which then persists until mitotic entry (which is also very obvious from the nuclear shape as visualized by Rad52-mCherry). Moreover, we previously found that HO endonuclease only induces DSBs during S phase (Leland et al., eLife, 2018). Given this, for individual cells we observe site-specific DSBs beginning in late S and early G2 phases and all of our analysis is done at this phase of the cell cycle. These observations are further validated by the observation that an HO-induced DSB undergoes very high rates of gene conversion in fission yeast (Prudden et al, EMBO J., 2003).

      4) In Figure 3D, since a major finding of the paper is that there are multiple invasion events, it would be nice to show some representative images of a few cells where multiple pairings occur.

      In Supplementary Figure 2A, we provided an example of a cell with multiple encounters between the DSB and donor. This will be more clearly highlighted in the revised text.

      5) It is known from Eric Greene's work that RAD51 mediated homology search can do multiple samplings of 8-9 nucleotide segments. Have the authors considered the area around the DSB site and how many potential pairing sites there might be in this region? Is it possible that having a LAC array with repeated segments might be influencing this the pairing since there would be multiple templates?

      We acknowledge that the homology of the region surrounding the DSB is important for faithful recognition of a homologous donor and that there could be many pairing sites surrounding our induced DSB after end resection. Such local sampling, however, would not be discernible due to the resolution of the light microscope (>0.2µm). We will address this noteworthy point during our discussion in the revision. Importantly, we placed the lacO array over 3 kb away from the locus where the HO recognition site is integrated on the homologous chromosome to attempt to avoid exactly the Reviewer’s concern.

      6) It would aid the reader if there were some picture schematics of what the authors think is occurring throughout the paper in the Figures. Since this is a results/discussion, this approach would be appropriate in lieu of a model figure at the end (which would also be very nice).

      We agree that diagrams would aid in communication of our hypotheses and interpretations, and these will be included in the revision.

      7) Since the multiple strand invasion events is a major finding of the paper, it is important to test the hypothesis that multiple strand invasion events are occurring a different way. A few ideas would be to examine Lorraine Symington's work on BIR where she observes multiple template switching events (Smith, CE, Llorente, B, Symington, LS (2007) Nature, 447(7140): 102-105) or something analogous to Wolf Heyer's recent study in Cell on template switching that the authors already cited. Another idea is to try a RAD51 mutant. For example, Doug Bishop's group has created a RAD51 mutant that uncouples the homology search from strand exchange, Rad51-II3A mutant (Cloud, V et al (2012) Science, 337(6099): 1222). Perhaps a mutant like this might be able to further support the key finding here.

      While our findings share parallels with the works raised by the Reviewer, we would argue that there is a fundamental difference between BIR-type assays and the one we present here, namely that we are visualizing multiple strand invasion events at the homologous chromosome in a normal, high fidelity repair event rather than multiple strand invasion events during BIR, which frequently result in translocations. Moreover, as the two chromosomes are perfectly homologous in our assay, we cannot leverage sequencing to reveal past strand invasion events that took place during HDR. We also cannot, unfortunately, access multiple simultaneous strand invasion events due to the diffraction limit of the light microscope. We concede that it would be informative to further dissect strand invasion using tools such as the Rad51-II3A mutant described in budding yeast in work referenced above by Reviewer #1 and developed in fission yeast by Sarah Lambert’s group (Ait Saada et al., Mol. Cell, 2017). However, with the present limitations on our laboratory access and the timeline necessary to carry out this experiment, we feel this is currently beyond the scope of this work.

      8) It is surprising that Rqh1 doesn't have a role in DNA end resection since this is a conserved function from budding yeast to man. Would similar results to what is observed in Figure 4 be observed in a Dna2 or Exo1 mutant?

      We acknowledge that Rqh1 orthologs in other organisms (BLM/Sgs1/etc.) have been shown to contribute to DSB end resection. However, previous work from our group indicates that Rqh1 is entirely dispensable for long-range resection in fission yeast (Leland et al., eLife, 2018). Interestingly, in this work we also demonstrated that it is only upon loss of either the 53BP1/Rad9 orthologue Crb2 or Rev7 that Rqh1 is able to compensate for loss of Exo1. It remains unclear whether this is a peculiarity of fission yeast (perhaps because they rely heavily on HR due to extensive time in G2) or if it is a direct consequence of the long G2 itself. Regardless, we demonstrated that cells lacking Exo1 cannot generate sufficient ssDNA tracts to load visualizable Rad52-mCherry (Leland et al., eLife, 2018). Given this, we cannot address this genetic background in this assay. The essential role for Dna2 in replication has also precluded its analysis.

      \*Minor comment:**

      1) As mentioned in the first line of the abstract, HDR is generally considered error-free as opposed to a pathway that "can be" error-free. *

      We acknowledge that HDR (and more specifically HR) is often error-free, but there are notable exceptions such as when a non-homologous donor is utilized for repair or when the polymerases engaged during repair incorporate errors (work from Haber and colleagues). We will expand and clarify this sentence in the revision.

      2) In Figure 2D, it is unclear whether this experiment is done in diploid cells. The rest of the figure is in diploid cells but two LacO cassette are not present past the first frame. Please clarify in the legend and/or figure panel. As mentioned above, this is also confusing in Figure 3.

      As above, we monitored repair events in diploid cells only – this will be clarified in the revised text.

      *Reviewer #1 (Significance (Required)):

      The most important advancement in this paper is that multiple strand invasion events occur during homologous recombination and the role of the Rqh1 in this process. Rqh1 is important protein whose mutation is implicated in human disease such as Bloom syndrome and cancer. In addition, misregulation of double-strand break repair and particularly of Rad51 is associate with cancer. Therefore, understanding the basic mechanisms of how Rad51 mediates double-strand break repair and the role of Rqh1 in this process is critical for understanding fundamental aspects of cancer development. * We appreciate the Reviewer’s assessment of the impact of this work.

      *Reviewer #2 (Evidence, reproducibility and clarity (Required)):

      In this study, Vines et al developed a microscopy-based assay to determine the kinetics of a site-specific interhomolog repair event, in living fission yeast cells. They detect efficient homology search and homology-directed repair in the system. They also observe that repair is likely to involve multiple site-specific and Rad51-dependent co-localization events between the DSB and donor sequence, suggesting that efficient inter-homologue repair involves multiple strand invasion events. Loss of the RecQ helicase Rqh1 leads to repair through a single strand invasion event. However, failure to repair is more frequent in rqh1 mutants, which could reflect increased strand invasion at non-homologous sites.

      Overall, I find the approach to investigate homology search and homology-directed repair using live cell imaging interesting and potentially very informative. The ability to observe the process in living cells, and with high temporal resolution, complements a variety of previous studies that employ more indirect approaches to invoke similar models. In particular, previous work by the Heyer, Lichten and Hunter laboratories, in budding yeast, has established that Sgs1 promotes non-crossover recombination by acting as a quality control in the maturation of HR intermediates. In this sense, while newly described here for fission yeast, it is not unexpected that homology-directed repair involves multiple strand invasion cycles. In my opinion, the strength of the work is the method/approach, rather than the specific conclusions made (even though I think that it is important to know how fission yeast cells perform homology search).*

      We thank the Reviewer for their appreciation of the value that cell biology can bring to the study of homology-directed repair. We wholeheartedly agree that this work is consistent with prior work on Sgs1. With regards to multiple strand invasion cycles, while we agree that there may be many in the field who could be unsurprised by this result, we would argue that 1) demonstrating this by direct visualization of individual DNA repair invents has clear inherent value and 2) many studying homology search itself (or who have modeled homology search in silico, for example) do not incorporate multiple strand invasion cycles in their thinking. Thus, we would argue that this work goes beyond a technical feat and will have impact beyond the approach.

      *However, for the reasons detailed below, my general impression is that it isn't clear how robust the method is at delivering unambiguous information on the important questions asked:

      1) The authors state that they have developed a system to monitor the 'dynamics and kinetics' of an engineered, inter-homologue repair event. With this in mind, I was expecting a more detailed exploration of the process of homology search. For example, what happens at shorter time scales? Is it possible that by imaging at every 5 minutes many of the events are missed? Could the authors be missing very transient events (especially in rqh1 mutants) by using an inappropriate time scale? *

      We acknowledge that it would be ideal to observe DSB repair across a range of time scales in our system. For practical reasons we found it most valuable to choose the 5 minute time window since it was most amenable to observing the entire course of repair as often as possible in an asynchronous cell population (see our response to Reviewer #1’s comment 3 above) while mitigating photobleaching. However, we recognize that we sacrificed time resolution between acquired frames in order to do this. Like the Reviewer, we were also concerned that we were missing transient events due to an inappropriate timescale.

      To address this, we acquired additional data in WT cells with greater time resolution with a focus on encounter frequency rather than time to repair (as the overall length of the usable movie that we can obtain is shorter). When imaging WT cells with a site-specific DSB at 2 minute intervals (2.5 times more frequently), we do observe a shift (of ~ 1 encounter per 30 minute window) toward more colocalization events with the donor sequence. We also observe, however, that more sampling leads to an increase in random encounters as revealed by similar analysis of the two lacO control strain as described in the manuscript. These data will be included in the revision and suggest that we may be missing some transient encounter events while using 5 minute time points. As noted by Reviewer #2, this could account for repair in the subset of WT and Rqh1-null cells in which we observed no encounters. We will acknowledge these caveats in the revision but would argue that our data support the conclusion that loss of Rqh1 decreases the number and/or lifetime of strand invasion events.

      2) Another point relates to the Rad52 signal/foci, which is central to the study. While it is clear to me what the authors consider to be a focus of Rad52, I am not sure how to interpret what has happens when Rad52 is as enriched throughout the entire nucleus as it is in the repair focus in the still before. For example, Figure 1C, 40 min vs 45 min. How do the authors interpret what is being visualised? Similarly, is the level of colocalization at 90 min really reflecting a specific enrichment of Rad52 at the DSB site? Much more of the Rad52 signal is away from the DSB. In other words, are quantitative criteria being used to assign colocalization events?

      As described in our Methods and the text, we used specific criteria to define 1) whether DSBs are site-specific and 2) whether they are colocalized with the donor site. In the images indicated as “contrast adjusted” we have scaled each panel time point individually with respect to the pixel intensities (that is, the least and most intense pixels have been set the same value for each). This strategy allows us to convey relatively dim Rad52-mCherry foci, particularly early after DSB end resection. A consequence of this is that the apparent background for panels in which there is not a strong Rad52-mCherry focus will appear higher, while the background will appear relatively less at time points with a strong Rad52-mCherry focus. For this reason we also present the raw image (found above). It is important to emphasize that when we are applying co-localization criteria, we do so within a 3D stack of images to ensure that the Rad52-mCherry signal and lacO array GFP signal coincide. In 2D representation, however, we understand that this may appear less clear.

      In the particular case of the colocalization in Figure 1C at 90 minutes that the Reviewer points out, it is more evident in the 3-D Z stacks that the surrounding mCherry signal apart from the colocalization with the lacO array is due to inhomogeneity in the background signal. Another contribution is that the lacO array signal often becomes delocalized during colocalization events (as evident in that 90 minute time point). Although this is an interesting observation, we are still investigating what activity may explain this response. We will address the caveats of our colocalization analysis more fully in the revision.

      3) In the system described here, Rad52 foci form in only ~15% of cells. I think it would be important to rationalise this low number in the manuscript. Moreover, G2 Rad52 foci still form at considerable rates in cells without HO. I think it would be important that the authors provide some explanation on what this might reflect.

      There are several considerations that we believe contribute to this observation, which we also documented previously in haploid cells (Leland et al., eLife, 2018). First and foremost, this assay is quite different from endpoint assays that involve induction of HO nuclease because we analyze only those events that happen immediately after additional of uracil to elevate HO endonuclease expression under the control of the urg1 promoter. Combined with the efficient repair of any DSB induced by leaky HO expression (taking less than an hour according to our data), we likely miss events that have already taken place or would take place later in other assay systems. Lastly, it is established that nucleosomes can prevent HO cleavage in its intrinsic role in budding yeast (Laurenson and Rine, Microbiol. Rev., 1992; Haber, Ann. Rev. Genet., 1998); we cannot rule out that cleavage at this particular site is less efficient due to intrinsic nucleosome stability. With respect to spontaneous DNA damage, most of this is short-lived and occurs in S-phase, likely due to replication stress, although we occasionally observe long-lived Rad52 foci in a sub-population of cells – this is in line with previous publications (Coulon et al., MBoC, 2006; Lorenz et al. Mol. Cell Biol., 2009; Sanchez et al., Mol. Cell Biol., 2012; Schonbrun et al., J Biol. Chem., 2013). We will provide a greater explanation of the observed induction rate in the revision.

      \*Other issues to consider:**

      4) In Figure 2D, the overlay does not show any green. It is possible that the green channel was not overlaid with the pink? *

      We apologize for this error and very much appreciate the Reviewer noticing that it is missing from the merged image. This will be corrected.

      5) In Figure 2D, the unadjusted images for Rad52 are very sharp. Did the authors perform contrast adjustment in the top panels? If so, this should be indicated. My current impression is that the data was duplicated by mistake.

      The Rad52-mCherry data in Figure 2D was labelled correctly and not duplicated. Because cells lacking Rad51 accumulate extensively resected DSBs (and therefore abnormally high levels of Rad52 loading), the intensity of Rad52-mCherry is very high. For simplicity we will remove the contrast-adjusted Rad52-mCherry images in the revision.

      6) I don't understand why is the time since nuclear division different is every single figure. For simplicity, it would be much better to start every figure at T=0.

      We agree with the Reviewer. In the revision we will normalize all kymographs to begin at t=0 with the exception of the Fig. S1D (where we are visualizing the subsequent division).

      *Reviewer #2 (Significance (Required)):

      see above. Reviewer #3 (Evidence, reproducibility and clarity (Required)):

      In this manuscript, the authors describe a system to monitor an inducible site-specific double-strand break (DSB) and the undamaged homologous locus during homology-directed repair in S. pombe cells. The authors show that the Rad52 focus on the induced DSB is more persistent than spontaneous Rad52 foci that form throughout the cell cycle. The persistent Rad52 focus intermittently colocalizes with the donor sequence labeled with LacI-GFP, reflecting multiple strand invasion events, and this colocalization requires the Rad51 recombinase. The authors report that the time to repair is dependent on the number of strand invasion events (colocalization of Rad52 and homolog), and that the initial distance between the induced DSB and the homolog predicts the time to their first contact, but does not predict the time to repair. Lastly, the authors claim that repair in rqh1Δ cells is bimodal, either failing to repair within the experimental time frame, or being more efficient than WT cells (which often involves a single colocalization event).

      **These claims are supported by the data:**

      1) Rad52 focus on the induced DSB is more persistent than spontaneous Rad52 foci that form throughout the cell cycle.

      2) Multiple colocalization events between Rad52 focus and the donor sequence are frequent, and this colocalization is dependent on Rad51, which reflects multiple strand invasion events.

      3) rqh1Δ cells have a lower rate of productive repair compared to WT cells. *

      The key concern I have for this section is the noise in Rad52 images. For example, in Fig. 1C at 15 minutes, it looks like there is a Rad52 focus both before and after adjustment but the time point is labeled as not having a Rad52 focus. Conversely, in Fig. 2D at 60 minutes, it looks like there isn't a Rad52 focus but the time point is labeled as having a Rad52 focus. How did the authors determine the presence of a Rad52 focus? Additionally, it is difficult to assess colocalization of Rad52 and LacI-GFP in merged images (hard to see Rad52 focus in Fig. 1C merged and LacI-GFP in Fig. 2D merged).

      The criteria that we established to indicate a Rad52-mCherry focus (as annotated by a pink circle and as explained in the Methods) is that it persists for at least three frames (>15 minutes). This was chosen because it is a characteristic of the HO-induced DSB but not of spontaneous DNA damage that occurs frequently during S-phase. Indeed, the numerous, small, and short-lived foci at the 15 minute time point in Fig. 1C referred to by the Reviewer occurs just 15 minutes after nuclear division and is perfectly characteristic of replication stress that is independent of HO endonuclease expression. Thus, the pink circles indicate a specific type of Rad52-mCherry focus that is relevant for the assay. We agree that the Rad52-mCherry focus in Fig. 2D at ~60 minutes is poorly visualized in the flattened image, but would like to emphasize that we assess the foci in the true 3D volume. With regards to the merged images, we will adjust the individual signals to make it easier for the reader to assess colocalization in the revision.

      \*These claims are supported by weak data:**

      1) The initial distance between the induced DSB and donor sequence predicts the time to their first physical encounter (Line 60). *

      We agree with the Reviewer that our word choice (“predicts”) suggests a stronger relationship than is supported by the data. However, we also argue that there is nonetheless a meaningful correlation. We believe this is an important point to make because it supports prior work in budding yeast suggesting that relative position affects donor choice preference. We will edit this language in the revised text.

      2) Repair efficiency is dictated by the number of strand invasion events (Line 61-62). Figures 3E and 3F technically have positive correlations that support the authors' claims but there is a lot of noise. I think the data needs to be more robust, especially considering the strong wording used to describe the data. A minor comment on Fig. 3F: why is there a data point with 3.5 encounters?

      Again, we agree with the Reviewer that our word choice (“dictate”) is too strong given the data and we will edit the text accordingly. We thank the reviewer for noticing the error in Fig. 3F, which will be corrected.

      \*These claims are not supported by the data:**

      1) In the absence of Rqh1, successful repair requires a single strand invasion event (Line 63). *

      We acknowledge that this is too strong a claim to make based on our data and will amend this language in the revision text. Specifically, and as outlined in our response to Reviewer #2 with regards to our imaging frequency, we will revise the manuscript to state that cells lacking Rqh1 are more likely to repair without a visualized colocalization event and/or they possess shorter lived strand invasion events. Importantly, repair outcome assays indicate that cells lacking Rqh1 display elevated gene conversion rates rather than non-HDR-mediated repair (Hope et al., PNAS, 2006). Thus, we do not expect that the lack of colocalization reflects NHEJ but rather our inability to “catch” the colocalization event with the temporal resolution we can achieve.

      2) rqh1Δ cells that complete repair are more efficient than WT cells and often involve a single colocalization event (Line 178-179).

      As for the above, we agree that our claim that rqh1Δ cells “often” involve a single colocalization event is too strong a claim based on our data. We will amend this language in the revised text.

      Fig. 4A shows an example of a rqh1Δ cell with productive repair but without any colocalization with the homolog, which contradicts the statement that successful repair requires a single strand invasion event in the absence of Rqh1. If the authors interpreted the single continuous presence of Rad52 focus during time-lapse as evidence of a single strand invasion event, then it would nullify using multiple colocalization events as evidence for multiple strand invasion events. In other words, the data in Fig. 3D that clearly displays multiple colocalization events in individual cells during repair can no longer be evidence of multiple strand invasion events since those cells all had one continuous presence of Rad52 focus.

      We believe that we understand the confusion that the Reviewer is articulating in their comment and apologize that we have not been clearer in explaining our interpretation. For this site-specific DSB to be repaired, we expect that it must either 1) engage with the homologous chromosome to be repaired by HR/BIR or 2) be repaired through an alternative pathway – at this non-repetitive, resected locus this would likely be a microhomology-mediated (alt-) NHEJ mechanism. However, prior analysis of repair outcome in a model of interhomologue repair in the absence of Rqh1 (Hope et al., PNAS, 2006) demonstrates an increase in cross-over HR events rather than end joining events, arguing that interhomologue HR still dominates (and with increased CO to NCO frequency). We interpret the continuous presence of a Rad52 focus to only reflect that a DSB has been subjected to resection and has not yet been repaired. Taking these two points together, within the lifetime of a Rad52-loaded DSB it can either 1) never colocalize with the donor sequence and fail to repair (as in cells lacking Rad51, Fig. 2D-F) or 2) undergo strand invasion (and therefore colocalization) at least one time (but possibly multiple times) to allow for HDR to occur. However, we agree (and must clarify in the revision) that we often infer that at least one strand invasion event has taken place to support successful HDR when we do not capture the event at our experimental time resolution. Based on the additional data at shorter timescales that we will add to the revised manuscript (as outlined in the response to Reviewer 2, point 1), which demonstrates that we may in some cases be undercounting relevant colocalization events that are too brief to be accurately captured with 5 minute time resolution, we think the most parsimonious explanation is that cells lacking Rqh1 spend less time with the DSB and donor sequence colocalized prior to repair. We agree with the Reviewer, however, that we cannot say whether this reflects a shorter duration of interactions and/or a fewer number of interactions. We will therefore revise the manuscript to acknowledge this point.

      Regarding the second claim, I think Fig. 4D only shows rqh1Δ cells with successful repair (since the longest repair time is 55 minutes, but it is not clear from the figure legend). It is not shown how many colocalization events these cells had in Fig. 4D, but there are 16 cells in Fig. 4D while there are only 2 cells with a single encounter (shown in Fig. 4F). With these numbers, it seems like rqh1Δ cells that complete repair are more efficient than WT cells but only few of these cells involve a single colocalization event.

      The Reviewer is correct, Figure 4D does indeed show only rqh1Δ cells with the site-specific DSB that successfully repair – this will be clarified in the revision text. As described above in our response to Reviewer #2’s comment 1, it may be that we are missing colocalization events in rqh1Δ DSB cells. However, we would argue that our data do support that, for cells lacking Rqh1 that execute repair, there are fewer and/or shorter-lived colocalization events. Again, this will be made clear in the revision.

      Also, how often do Rad52 foci form spontaneously in rqh1Δ cells and what is the duration? This data was provided for WT but not for rqh1Δ.

      We agree that increased levels of genome instability (and therefore Rad52 foci) would present an issue – and indeed this has prevented us from analyzing some genetic backgrounds. However, we do not observe a significant increase in spontaneous Rad52-mCherry focus formation in rqh1Δ cells. This data will be included in the revision.

      All of the data would have been more supported if the homologous chromosome would have been tagged. Such a configuration would really have helped the interpretation of the rqh1∆ data.

      We agree that in theory it would be advantageous to have both copies of the chromosome tagged. Indeed, we attempted to leverage a different version of this experimental system with lacO arrays on both copies while inducing a DSB. However, the complexity of monitoring (and keeping the identity clear) for the two copies presented major challenges. Better would be two distinct arrays – an approach that has been used in budding yeast. However, to date many groups, including ours, have been unable to get TetO-TetR arrays to perform well in fission yeast.

      * Reviewer #3 (Significance (Required)):

      The significance of this work is the conceptual advance in the field of DNA repair. Homology search is an important process in homology-directed repair and is not fully understood. This study reports time-lapse data on the interaction between a DSB and its donor template during repair and provides insight into the kinetics of homology search. The audience for this manuscript is the field of DNA repair, and to a lesser extent, field of live-cell imaging.*


    1. Promotion of Employees in an Organization Leave a Comment / Blog Contact Promotion or career advancement is a procedure through which a worker of an organization is given a higher portion of obligations, a more significant compensation scale, or both. Advancement isn’t only gainful for representatives but at the same time is exceptionally essential for the business or entrepreneurs. It supports the confidence of advanced representatives, expands their efficiency, and henceforth enhances the general benefits earned by the organization. A Promotion is above and beyond that, a worker takes while working in an association to the extent his/her work, rank or position is concerned. Each association or work environment has a specific activity pecking order structure as per which a worker propels in that association and gets advanced. Promotion isn’t only a prize that a worker is given for his/her proceeded with great execution, however, it is the verification that a business imagines that the time has come to add more obligations to a representative’s current arrangement of duties. There are numerous kinds of Promotion – while some assurance headway in the activity position or rank, others may ensure an expansion in the compensation or pay. In some association, an advancement may bring about a simple change of obligations and duties. Advancing representatives for their greatness in execution is essential to esteem their endeavors and keep their assurance up. Significance and Benefits of Employee Promotions An Promotion isn’t just an approach to add more obligations to a worker yet is a significant type of boosting representative inspiration and assurance. This outcomes in high profitability and keeps your organization from losing its significant and significant workers. There are numerous advantages and preferences of advancing representatives normally for their endeavors, and coming up next are a portion of the primary ones: Perceives and Improves worker execution, desire, and difficult work: One of the most significant advantages of representative advancement is that it causes you to perceive and recognize the presentation and difficult work of your workers and therefore causes them to feel esteemed. At the point when a representative’s endeavors are esteemed, he/she will in general work harder and attempts to improve his/her presentation further which thus works for the business. Advancement likewise improves the desire of representatives and lifts their energy for arriving at their objectives just as the destinations of your association. Lifts inspiration and expands unwaveringness of representatives: A worker who gets elevated will in general feel increasingly enlivened and roused to keep accomplishing the great work, and this is one more motivation behind why advancements are so significant. This expansion in inspiration prompts better efficiency and execution that thusly works in the kindness of your association. In addition, an advancement likewise improves and builds faithfulness among the representatives since they will in general feel a feeling of having a place in the working environment and begin thinking in support of its. Energizes maintenance: When the correct ability is recognized by the administration and opportune advancement is drilled, the meriting and gifted representatives may not be driven away from the working environment. This improves the maintenance of the gifted and skilled representatives and in this manner benefits the work environment much further. By holding top-level specialists and workers, your association forestalls the requirement for recruiting new ability and afterward preparing them once more, along these lines setting aside time and cash. Creates serious soul at the work environment: When the better group individual gets advanced, the other colleagues might be charged and propelled to perform superior to them, and this assists with building up a sound serious soul at the work environment. This sort of a soul is useful for an association since it guarantees great exhibitions all around and consequently a general improved efficiency. Grooms pioneers for the future: When the administration chooses to advance representatives for their difficult work, they are fundamentally perceiving ability and discovering future pioneers. This recognizable proof of future pioneers empowers them to prepare representatives for the future and causes them to investigate their aptitudes and ability further. Preparing pioneers for what’s to come is gainful for the association and assists with improving the general efficiency. Decreases representative opposition and discontent: If workers continue buckling down with no gratefulness, applause, or evaluation, a feeling of discontent and obstruction may manifest, and this may not be useful for the association. This can even cause their exhibition to go down, consequently demonstrating negative for the organization. In any case, inside advancement can assist with lessening worker opposition and discontent. When to Promote Employees? For an association, choosing whether or not a representative is prepared for advancement can be a test. Not all representatives might be prepared for an evaluation or an advancement because of absence of the length of administration, the correct number of accomplishments just as absence of the required abilities and involvement with the given position. An advancement is a significant choice since it doesn’t just include change in rank yet additionally more obligations and duties and thus should be taken after cautious appraisal and assessment of one’s abilities, execution and a few different components, that have been given underneath: Execution — One of the most significant focuses for assessing when is the ideal chance to elevate a representative is to survey or assess his/her presentation over a given timeframe. Length of administration (status) – Another thing to think about for knowing when the worker is prepared for advancement or not is the length of administration. The quantity of years that one has additionally served decides the hour of advancement. Legitimacy and capacity – The legitimacy and the capacity of the individual, is additionally an extraordinary apparatus to gauge the general value of the worker’s advancement. Instructive/specialized capabilities – The instructive and specialized capabilities is additionally a measure or dynamic apparatus undoubtedly. Appraisal of potential – The measure of potential one has in him to act later on or at another position ought to likewise be surveyed before choosing about advancements. Dividing of the advancement and profession length of the individual – Another thing that must be viewed as while considering the advancement for a representative is the time since the last advancement. Preparing – The measure of preparing a worker has been given or the level to which he/she has likewise been prepped goes about as a central factor. The Most Effective Method to Promote Employees Since you realize how to distinguish whether a representative is prepared for promotion or not, the time has come to make sense of how you would advance him/her. Promotion isn’t only the upward or forward reassignment of a representative of your work environment however is an appropriately organized procedure that must stay inside the limits of the association’s chain of command. Consequently, an appropriate procedure of promotion of representatives must be followed which is reasonable for all and one-sided for none. The accompanying given data will assist you with realizing how to advance your representatives: Models and plans of promotion There are a few unique plans and models of promotions. Each association either picks one of these strategies or applies a mix of at least one such model to advance their representatives. How about we investigate the absolute generally utilized and well known strategies: Non-competitive model of promotion Promotions that depend on the standard arrangement of accomplishments and achievements of workers are known as noncompetitive promotions. In such promotions, representatives are advanced once they arrive at a specific achievement in their vocation venture, regardless of variable highlights like execution and devotion, and so forth. These kinds of promotions work flawlessly for all the representatives, and there is no degree for a predisposition or uncalled for examination. Time scale promotion (Based simply on status) A period scale promotion is a sort of a noncompetitive promotion that happens after the individual has spent a norm or fixed number of years working for the association or working at a specific situation in the association. Accumulation of Duties For the situation when an individual must be elevated because of an expansion in his/her remaining task at hand or obligations is additionally a non-competitive strategy for promotion. Adhocism now and again, an association may need to advance somebody without following any approach however just to address the issues of critical or dire occasions or circumstances. Serious model of promotion A serious model of promotion is a strategy wherein workers need to demonstrate their value to managers so as to be elevated to a higher position or position. In contrast to the noncompetitive strategy, this technique considers representative presentation, efficiency, aptitudes, experience, information and other such factors. There is a correlation of representatives based on their difficult work and execution in this model. Legitimacy based promotion – Merit-based promotions are serious kinds of promotions in which an individual is advanced based on their aptitudes, execution, information, difficult work, and capabilities. Legitimacy cum-rank – Merit-cum-position promotions are those sorts of promotions where harmony between the legitimacy just as the status of the individual is remembered before thinking about a promotion. Up-degree promotion or Out promotion This is the sort of a promotion technique where when the measure of boosts in salary without an adjustment in the activity or rank. If there should be an occurrence of Out promotion, a representative looks for work elsewhere as a result of being offered a superior compensation scale. Dry promotion For this situation, a worker is elevated to a higher position yet without an expansion in the pay. Paper promotion Paper promotion happens in government occupations that comprise of a few divisions. These kinds of promotions occur based on the rank of a worker and are the ones where the representative having a place with a parent office is given a promotion by another office from which it gets the climb in pay.

      Promotion is an achievement that only the best employee deserves. As an employer you must give the promotion as a token of appreciation. Find out how.

    1. Note: This rebuttal was posted by the corresponding author to Review Commons. Content has not been altered except for formatting.

      Learn more at Review Commons


      Reply to the reviewers

      We thank the reviewers for their comments and outline below how we plan to address them.


      Reviewer #1 (Evidence, reproducibility and clarity (Required)): **Summary:** Provide a short summary of the findings and key conclusions (including methodology and model system(s) where appropriate). The authors here describe a method to modify bacterial artificial chromosomes (BAC) harbouring gene loci from eukaryotes. When wanting to modify a BAC an antibiotic selection cassette is often included alongside the desired mutation/modification to increase the number of successful recombinants in E.coli. Traditionally, this is removed in a second recombination process to leave only the desired modification. The novelty in the procedure described herein is to add a synthetic intron consensus sequence around the selection cassette, which eliminates the need for the subsequent removal of the antibiotic cassette from the BAC before transfection into mammalian cells, saving time and resources. The technique is clever in its simplicity and appears to function for a number of gene loci. The authors validated the correct functioning of the modified BACs for a number of genes using three main assays - transcript level, protein level and localisation. **Major comments:** *Are the key conclusions convincing?* The conclusion that the method described generates functional modified BACs is valid. *Should the authors qualify some of their claims as preliminary or speculative, or remove them altogether?* While the method is successfully employed in this study, its efficiency is not quantified in relation to the state-of-the-art as described in the introduction. One assumes it would be more efficient, but this has not been tested empirically in the paper. Does the inclusion of the synthetic intron sequence have an effect on the efficiency of modifying BACs compared to a more typical two-step positive/negative antibiotic selection cassette? *

      • *

      This is a good point that we did not directly address. In general, the efficiency is similar to that of integrating any cassette with selectable marker, as has been published (Poser et al 2008), and therefore also higher than the two-step counterselection method, which requires such a cassette integration in the first step alone. We will include new data specifically addressing the efficiency of our new method (see specifics below)

      The functionality of this approach rests entirely on the ability of the target cell to correctly splice out the synthetic intron. The authors are aware of this potential problem as highlighted in the lines below, but do not make efforts to explicitly test splicing. On lines 224-225, the authors state "We cannot exclude that a small portion of synthetic introns within individual cells are misspliced". On lines 230-231 it is stated that "mis-spliced mRNAs are probably minimal and degraded by nonsense-mediated decay". On lines 215-217, the authors describe an "investigation of transgenic lines at the single-cell level" that suggests "the synthetic intron is correctly spliced out in all the cells of the population". How do the authors reach this conclusion? U2OS and HeLa cells are considered very "robust" and may not show detectable consequences when stressed with an increased level of nonsense-mediated decay. Further, many genes maintain a high level of expression that buffers them against small changes in transcription/splicing. The synthetic intron might have a bigger impact on more tightly regulated genes, so assessing the splicing rate would be essential if the authors wish to advocate their technique as generally applicable.

      • *

      We will assay for splicing efficiency as outlined below.

      The ability of the synthetic intron to be removed from final transcripts depends on functioning splicing machinery. The authors might emphasise this issue, as spliceosome mutations are important fields of study and might not be compatible with this method.

      • *

      We can add this in the text

      The authors used un-directed integration of each BAC under study. Therefore, it is hard to assess what effect the synthetic intron has, as the authors only ever assess the downstream levels of the correctly spliced, translated and localised protein. The authors themselves state that this can lead to clonal variations in expression of up to 2-fold and on line 250 that this variation "could compensate for synthetic intron effects", but make no effort to test this. Again, lines 267-268 highlight the potential dangers of potential effects of the synthetic introns, but do not test these. \Would additional experiments be essential to support the claims of the paper? Request additional experiments only where necessary for the paper as it is, and do not ask authors to open new lines of experimentation.* If not already performed, a large number of bacterial colonies should be screened for the correct modification and frequency of correct ones reported. This frequency - reported for at least three different modifications - would estimate what sort of efficiency this method provides. The modified region of each BAC should be sequenced and the results reported. The rate of exactly modified clones is important, in case of spontaneous or low fidelity integration of the antibiotic cassette. The percentage of transcripts that have the synthetic intron correctly spliced out should be measured for some of the BAC constructs used in the study. A direct head-to-head comparison of this newer method compared to other techniques, or even the authors' own previous two-step approach is necessary to assess the benefits of this method. Preferably, the experiment would be run in parallel with and without antibiotic selection applied, to show that it drastically improves chances of finding a correct clone. *

      We will generate 3 new mutations in BACs and analyze both the efficiency of integration by PCR and accuracy via sequencing. In practice, we have observed that the efficiency is similar to any other cassette integration, such as a GFP tag (Poser et al Nature Methods 2008) or a counterselection cassette (Bird et al Nature Methods 2012) (80-90%). Integrating a mutation via the second step of the counterselection method introduces a further 20% decrease in efficiencies on average.

      \Are the suggested experiments realistic in terms of time and resources? It would help if you could add an estimated cost and time investment for substantial experiments.* Repeating the transformation of the BAC and targeting cassette and assessing the recombination efficiency and sequencing should only require existing reagents and take less than a week or two to complete. Quantitative RT-PCR to assess the percentage of transcripts that have the synthetic intron spliced out would take a little more work. However, this should not be a considerable investment in time or resources for a standard microbiology laboratory and could be completed within a few weeks using modern techniques, such as that described in Londoño et al. 2016. Repeating all the experiments in parallel would be considerable work and would only be strictly necessary if the authors wish to emphasise the benefits of their method over the many others already in wide use. *

      • *

      We will use quantitative PCR to estimate the fraction of transcripts that correctly splice out the artificial intron for two clonal cell lines characterized in the study: RNAi-resistant AurA-GFP (Fig 4), and GTSE1-14A (newly introduced; see below). While the exact method described in Londoño et al 2016 will not be applicable due to the larger size of the artificial intron, we believe we can adapt it to detect different splicing events.

      \Are the data and the methods presented in such a way that they can be reproduced?* Barring the omission of Table S1, which presumably includes exact information on the BACs modified and sequences used etc., there is sufficient other data and methods to allow the experiments to be repeated. Targeting the ESI procedure to the middle of exons is likely to have a bigger impact for smaller exons as the authors mention on lines 99-100. Making it clear which exon sizes for each gene were successfully targeted in this study would help give some idea of how significant a problem this might be. Perhaps Table S1 contains this information, but it was not provided. It would also help reviewers check the design strategies. *

      We apologize for inadvertently failing to upload Table S1 on bioRxiv. It has been uploaded now as part of this submission process. This table indeed contains BAC and target sequence information, including the size of the targeted exon (and the 2 “new” resulting exons). Targeted exons range in size from 138bp to 1537bp, and “new” exons are as small as 48bp.

      \Are the experiments adequately replicated and statistical analysis adequate?* The replication and statistically analysis of the data as presented appear adequate. Figure Legends should state the statistic used to generate error bars. *

      This will be updated

      \*Minor comments:** Specific experimental issues that are easily addressable. Are the promoters used in the vectors described universally functional? For example, is the PGK promoter functional in yeast? *

      • *

      The PGK promoter contained in the cassettes is a mammalian promoter, which has also been reported to work in flies.

      \Are prior studies referenced appropriately?* The manuscript may benefit from the referencing of BAC modification techniques from a wider variety of groups, such as those using CRISPR-guided recombineering (Pyne et al. 2015). *

      We will add citations of more techniques

      \Are the text and figures clear and accurate?* The body text is very clear save minor typographical or grammatical errors. Regarding figures, some of the coloured text in Figure 1 is somewhat illegible when printed in grayscale. Line 278 - The acronyms LAP and NLAP are not defined/explained. Antibody section starting Line 282 may fit better next to Western Blot section. Figure 2C - The blot images would benefit from arrows to indicate expected sizes of proteins. Figure 3A - the graph may benefit from a dashed line at 100% to highlight that values are normalised to controls. Figure 4 - The differences between panels B & C are unclear. Figure 4E - The legend could provide a little more detail on cell cycle stage/status of the captured cells. *

      All of the above will be addressed accordingly

      \Do you have suggestions that would help the authors improve the presentation of their data and conclusions?* Lines 23-27 are somewhat unclear and feel out of context. Perhaps the authors could clarify this as a further advantage of using BACs instead of endogenous gene modifications. *

      Thanks for the input, we will clarify this.

      While not affecting the factual content of the paper, I would advocate that the authors format the method described in Figure S3 into a more detailed text based layout similar to that seen in a typical Nature Methods article. However, this may depend on the format required by any eventual publishing journal.

      • *

      We prefer the graphical protocol, but will discuss whether to add a text protocol with the journal editor.

      That all of the work the paper was carried out in human cell lines and using human genes is a further caveat, but the authors admit this in the discussion and one would assume that most mammalian cells would respond similarly in their ability to splice out the synthetic intron. Reviewer #1 (Significance (Required)): \Describe the nature and significance of the advance (e.g. conceptual, technical, clinical) for the field.* This work is a formal description of a newer method that could be useful for many of those employing bacterial artificial chromosomes in numerous studies, such as gene regulation. *Place the work in the context of the existing literature (provide references, where appropriate).* This work builds on methodology previously published by the authors - a counter-selection two-step procedure (Bird et al. 2011). It sets out to formally describe a method merely mentioned as "BAC intronization" in a later paper by some of the authors (Zheng et al. 2014). Other alternative one-step procedures are also available, but present a different set of challenges (Lyozin et al. 2014). Some newer approaches, such as those using CRISPR-guided recombineering (Pyne et al. 2015) or systems that combine CRISPR and positive/negative selection cassettes (Wang et al. 2016) may be slightly more efficient, but are also more complex in their design. Bird et al. 2011 DOI: 10/dv776q Pyne et al. 2015 DOI: 10/f7jx92 Wang et al. 2016 DOI: 10/f89db5 Zheng et al. 2014 DOI: 10/f5pkr6 *State what audience might be interested in and influenced by the reported findings.* As a technology paper this work should have interest from a broad field of research. While the use of BACs could sometimes be considered more traditional in light of the explosion in CRISPR-based genome editing capabilities, it is definitely seeing a resurgence as the limitations of CRISPR in modifying large regions of genome become more apparent. Therefore, technologies that accelerate the modification of BACs could prove increasingly useful. As category of audience, all those involved in significant recombineering or gene/genome engineering would potentially benefit. *Define your field of expertise with a few keywords to help the authors contextualize your point of view. Indicate if there are any parts of the paper that you do not have sufficient expertise to evaluate.* Synthetic genomics, synthetic biology, cancer cell biology, gene and genome engineering REFEREES CROSS COMMENTING I would agree with reviewer two's assessment that we both view the paper in a similar light. Reviewer #2 (Evidence, reproducibility and clarity (Required)): This is a methods-focused paper that presents a strategy to efficiently introduce mutations into a bacterial artificial transgene using synthetic introns. BAC-based methods have been an effective strategy for introducing trans genes into human cells to achieve near-endogenous expression, including extensive work from these authors. However, generating mutations and changes within the internal coding sequence presents some challenges for how to target these mutations and select for the mutated form. Here, the authors describe a way to overcome this by introducing synthetic introns into an adjacent sequence. This allows them to introduce a selectable marker and conduct the molecular biology without creating complications downstream for the functionality of the protein. This method is carefully described and presented. The authors also provide clear validation by using this to create RNAi-resistant versions of multiple different mitotic factors as well as creating targeted mutants that alter the functional properties of a protein. This work clearly takes advantage of other ongoing studies from these labs (including mutants and cell lines that appear to also have been described elsewhere), but the ability to combine these in a single paper and clearly describe the method provides a helpful advance and validation. Based on the description and data presented, I think that things are clear and carefully validated. As such, I do not have technical comments or concerns and I would be comfortable with this paper appearing in an appropriate journal in its present form. Reviewer #2 (Significance (Required)): This is a solid methods paper, but for considering the nature of the impact and significance of this paper, there are several things to note: 1.The BAC-based method does appear to be a powerful and effective strategy. However, beyond the work of Mitocheck and the authors that are part of this paper, this has not seen widespread adoption. It is possible that this current method may increase its usage due to the value of the targeted mutations within the coding sequence, but at present it is not a broadly used strategy. *

      We agree that using BACs as transgenes has not seen widespread adoption as a tool on the broader cell biology community (although certainly beyond members of the Mitocheck consortium). This is likely because many erroneously think that it is a technique for specialist laboratories. We are trying to change this! For reasons outlined below, there is still an increasing desire for conditional analysis of mutated genes under physiological expression/regulation frequently not attainable via directed Cas9-based mutation. A major aim of this paper is thus to further simplify the methods for generating modified BAC transgenes.

      2.This BAC-based approach (and also RNAi) are becoming increasingly replaced by the use of CRISPR/Cas9 genome editing. The absence of Cas9-based strategies in this paper limits the potential impact and reach of this paper. The authors do mention the possibility of using a similar synthetic intron strategy for use with Cas9 in the Discussion, and appear to have conducted some experiments. If possible, it would substantially increase the value of this paper if this data and strategy were also included in the Results section (acknowledging that this may still be a work in progress).

      While some uses of BAC transgenes are in some cases better replaced by CRISPR/Cas9 techniques (i.e. GFP tagging), there are several occasions where using BACs are preferable: As stated in the text, RNAi-resistant BACs allow for conditional analysis of recessive mutations. Mutations in essential genes that are lethal will prevent growth and recovery of viable cells if integrated into the genome via Cas9. Additionally, deleterious mutations are prone to accumulate suppressive changes in chromosome integrity or gene expression during the procedure of selecting and expanding Cas9-modified cells for analysis, particularly in the genomically instable cancer cell lines frequently employed.

      We use both BACs and CRISPR/Cas9 in our lab according to our needs.

      We do have an ongoing project to apply this intronization technique to enable more efficient selection of CRISPR/Cas9 integrations. Preliminary results suggest that it works to allow selection of point mutations, but it is still being optimized, including a redesign of the cassette, and is not ready for publication.

      3.The method is solid and well-validated, but there are no new results or insights presented in this paper from the work that is described (this is fine, just commenting for considering the right journal fit).

      As “biological insights” gained as a result of this technique we had cited a couple studies that made use of the technique already (to functionally analyze a microcephaly-associated mutation in the centriolar protein CPAP at the single cell level in HeLa cells and neural progenitor cells (Zheng et al 2014, Gabirel et al 2016)). As a response to this critique to include “new biology” in this paper, we will add new unpublished data investigating a specific question: Is the cell-cycle-regulated disruption of the EB1-GTSE1 (microtubule plus-end tracking proteins) interaction in mitosis required for chromosome segregation fidelity? We have generated a GTSE1 mutant with 14 phosphosites mutated to alanine using this technique. We will present the effect on chromosome segregation.

      REFEREES CROSS COMMENTING It appears that both reviewers are largely on the same page regarding this paper.

  6. Jun 2020
    1. Reviewer #2

      In this paper, Benwell and colleagues present a perceptual decision-making task with confidence ratings in human subjects to investigate choice and metacognitive history biases. The statistical analyses are solid and thorough. The mutual information analysis is a valuable complement. The paper is comprehensive, clearly written and the figures are informative. It would help if the authors could emphasise their reasoning across the paper: why selecting this particular paradigm to study history biases, and what is the added value of their findings beyond previous work? Many similar data sets (perceptual decision & confidence) have been published and are open. Moreover, it seems important to better justify their hypotheses and motivate the analyses, particularly the final focus on repeat vs. alternation in a paradigm in which these were not manipulated explicitly by the experimenters.

      The authors suggest that history biases are adaptive (building on the stability of real-life environments), whereas at other times, that they are maladaptive ("irrelevant factors such as previous confidence reports"). It would be helpful to explicit the arguments in favour of either interpretation, and to clarify what computational interpretation the present findings favour, if any. There is already a little bit about why it may be advantageous to assume stability. Are there other reasons to think of choice/metacognitive bias as helpful vs. maladaptive? In which contexts? If repeating were a more prevalent bias than alternating in the population, why would this be useful? Relatedly, it would be helpful to further clarify for readers why it is relevant to study choice and confidence history biases, i.e. explain why it is not a simple by-product of experimental designs where experimenters artificially present multiple times very similar decisions.

      It is interesting that the authors comment on the prevalence of each result in their sample, instead of simply reporting statistics on group means, to get a better sense of the strength of the findings. However, it is a bit difficult to generalise about "population prevalence" unless larger samples than the current n=37 are used. Because the experimental design overlaps with previous work, most of these analyses could be re-done on other datasets to address discrepancies between the present findings and that of Urai et al. The confidence database (Rahnev et al., 2020) may provide a useful resource for future work (especially for drawing conclusions about population prevalence based on the current sample of 37 subjects).

      Technical suggestions/comments:

      Could the authors indicate the proportion of errors vs. correct trials on previous repeating vs. alternative trials? If there were more errors on alternating trials, could it be that due to a post error slowing mechanism whereby subjects became more accurate after an alternate, hence the increase in d'? Regarding Fig S1 and the comparison with previous studies, it would be worth discussing the results in relation to a related study in rats (Hermoso, Hyafil et al., 2020 ncomms), for instance examining if the choice bias was overall driven by correct trials and absent after errors (Fig 2A). Finally, in Fig S1 the authors show a choice bias present for both high and low confidence at t-1. Would it be more precise, for concluding about a lack of an influence of confidence, to perform an ordinal regression analysis using the 4 levels of confidence available?

      In Fig 1F, 1G, 1H, could the authors perform psychometric and statistical analyses to actually demonstrate the findings that the authors describe, or rephrase that the confirmation of model predictions are qualitative only? (For instance, showing quantitatively that the slopes are different for high and low confidence in Fig. 1F)

      In particular, when comparing Fig 1F and 1G, are the patterns for confidence and RTs identical with respect to absolute orientation? If so, is this an issue for interpreting confidence data (supposedly not only a strict reflection of RTs but also incorporating information about accuracy)?

      Could the authors comment on (even briefly) the other psychometric parameters (stimulus independent lapse rate and slope)? Why do the stimulus-independent lapses fixed and not fitted with the two other psychometric parameters? Does it change the conclusions if they are fitted? It would be worth checking parameters of the sigmoid psychometric function in Fig S1 (right panel red curve), because the psychometric function looks unusual with an increase at highly positive orientations.

      It is reassuring to see that the correlation results in Fig. S4 are reproduced using an alternative metric of metacognitive efficiency (meta-d'/d'). However, could the authors provide this measure for all other analyses based on meta-d'-d'? I am not asking for a detailed breakdown or new figures, but at least in the text specify whether findings are maintained using this alternative metric.

      Could the authors argue that here we have a true metacognitive history bias, and not a bias due to low-level effects e.g. motor anchoring, use of scale? (See e.g. Foda, H., Barger, K., Navajas, J., & Bahrami, B. (2017). Domain-general idiosyncratic anchoring of metacognition.)

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      Reply to the reviewers

      We thank all reviewers for their comments and suggestions, which will make our manuscript a much better one. Accordingly, we have already made changes to the manuscript (marked in yellow) and we will perform all the experiments requested. Below, we answer the reviewers point by point.

      Reviewer #1 (Evidence, reproducibility and clarity (Required)): This study provides solid evidences showing a role for the spectraplakin Short-stop (Shot) in subcellular lumen formation in the Drosophila embryonic and larval trachea. This subcellular morphogenetic process relies on an inward membrane growth that depends on the proper organization of actin and microtubules (MTs) in terminal cells (TCs). Shot depletion leads to a defective or absent lumen while conversely, Shot overexpression promotes excessive branching, independently on the regulation of centrosome numbers previously shown to be important for the regulation of the lumen formation process (Ricolo, D., Deligiannaki, M., Casanova, J. & Araújo, S. J. Centrosome Amplification Increases Single-Cell Branching in Post-mitotic Cells. Current Biology 26, 2805-2813 (2016)). Shot is rather important to regulate the organization of the cytoskeleton by crosslinking MTs and actin. Shot expression in TCs is controlled by the Drosophila Serum Response Factor (DSRF) transcription factor. Finally Shot functionally overlaps with the MT-stabilizing protein Tau to promote lumen morphogenesis. The figures are clear and the questions well addressed with carefully designed and controlled experiments. However, I would have few suggestions that will hopefully make some points clearer. **Major comments:** -Statistical analyses should be added for comparisons of proportions, including Fig. 1E, 1L, Fig. 2G-I, Fig. 6L, Fig. 7K, Fig. 8C-D and Fig. 9G.

      We agree with this and have now redone all graphs and revised all quantifications from this study. We have added error bars in all above mentioned graphs and have provided statistical analysis where appropriate. We have also redone all graphics and phenotype reporting, which is done now in relation to total TCs (rather than embryos or GBs and DBs TCs). This was suggested also by reviewer #2 and we agree because this is a more stringent and comparable way of quantifying our results.

      -It is not always clear what genotype has been used as the "wt" genotype, as in Fig. S2 or Fig. 3 for example, this should be added to figure legends.

      We have now clarified which flies are used as controls in each experiment throughout the paper. We have left wt where flies were wt, and changed all other cases to either the genotype or “control”.

      -Live imaging of Shot has been performed with ShotC-GFP, that cannot bind actin. Don't the authors think ShotA-GFP would reflect more accurately Shot endogenous behavior as it interacts both with actin and MTs? It would be better to show this, even if the results shown here tend to be consistent with Shot endogenous localization shown with Shot antibody staining.

      We agree and we will analyse movies with both ShotC and ShotA and present them in the revised version.

      -It is of course not possible to generate CRISPR mutant flies with mutations in putative DSRF binding sites in a reasonable amount of time, to confirm that Shot transcription is controlled by DSRF. It would thus be nice to reveal shot mRNA expression with in situ hybridization experiments in wt vs. bs embryos. This would confirm that Shot mRNA is downregulated upon DSRF inhibition and rule out a possible indirect effect on Shot protein stability for example.

      We believe the presented 3-way approach (in silico, protein quantification and phenotype rescue) is sufficient to show that Shot expression is regulated by DSRF. It is unlikely that we are dealing with protein stability or other issues, because we can rescue the lumen elongation phenotype by solely expressing Shot in TCs. However, we agree it would be nice to show this in an in situ hybridization experiment, and we will try to provide a conclusive one for resubmission. In situ detection methods, however, may not be accurate enough to detect such differences in single-cells.

      -In the same figure, it would also be interesting to show what happens to actin and MTs in bs TCs and to which extent their organization is rescued by Shot overexpression.

      We are working on this for resubmission. These experiments were frozen by the current COVID-19 pandemic and this is why they were not submitted with the first version.

      -UAS-EB1GFP does not seem to be an appropriate control in Figure 9 (A and B) since it can affect MT dynamics (Vitre, B. et al. EB1 regulates microtubule dynamics and tubulin sheet closure in vitro. Nat. Cell Biol. 10, 415-421 (2008)). Why not simply use an UAS-GFP?

      We have not detected any notorious larval TC phenotypes by overexpressing UASEB1GFP in TCs. Their branching is comparable to that in previous studies (for example Schotenfeld-Roames, et al Current Biology 2014) and there were no detectable luminal branching phenotypes. However, we agree it is more correct to analyse cells with a plain GFP and have repeated the controls for this experiment using DSRFGAL4UASGFP. This is now shown in figure 9.

      -Shot and probably Tau crosslinking activities are important for lumen morphogenesis with a striking increase in the number of embryos without lumen in shot3 and shot3 tauMR22 mutant embryos. The rescue experiments clearly show that Shot binding to both MT and actin is essential for efficient rescue. The same might apply to Tau since it is able to crosslink actin and MTs (Elie, A. et al. Tau co-organizes dynamic microtubule and actin networks. Sci Rep 5, 1-10 (2015)). I believe showing actin and MTs organization in these rescue experiments would be necessary.

      We agree and we will provide these experiments upon resubmission.

      Second, the overexpression experiments indicate that Shot is able to induce extra lumen formation even when unable to bind actin as shown with the increase in the number of supernumerary lumina (ESLs) under overexpression of ShotC and ShotCtail to a lesser extent. This phenotype is also observed under Tau overexpression. This suggest that not crosslinking anymore but rather making MTs more stable could be sufficient to promote extra lumen formation in a wt context. Stabilising MTs by treatment with Taxol might thus be sufficient to promote ESL formation. I am fully aware of the difficulty of treating Drosophila embryos with drugs, making this experiment hard to do, but I think this dual function of Shot and Tau (crosslinking actin and MTs to promote branching vs. stabilizing MTs leading to excessive branching) should be discussed.

      In Figure 2 we show not just that UASShotC is able to induce ESl but also that UAS-ShotCtail containing only the MT binding domain of Shot is enough to induce ESLs in TCs, whereas UAS-deltaCtail is not. We agree Taxol treatment would be a nice experiment to do, however we also think we provide enough evidence that MT stability is enough for ESL whereas de novo lumen formation requires crosslinking of MTs to actin. As advised, we will discuss better both Shot and Tau dual function in ESL generation and de novo lumen formation for resubmission.

      **Minor comments:**

      We have already addressed most these minor comments in the manuscript (text revised and changes in yellow). And we provide answers to some of the comments below.

      -p2 line 1: 'acentrosomal luminal branching points' may be better than 'acentrosomal branching points' to describe the phenotype. -p4, line 16: the reference 23 is not properly inserted (should be after 'closure'). -p5, line 16: Please mention what the abbreviations Bnl and Btn stand for. -p5, line 20: these 80% of TCs cells with defects in subcellular lumen formation should appear on the graph in Fig. 1E (as shown in graph 1L).

      We have added shot RNAi results to graph E in figure 1.

      -p5, line 26: this 36% value does not seem to correspond to anything on the graph in Fig. 1N. According to the figure legend, 20% of TCs did not elongate at all and the lumen was completely absent (class IV), which is consistent with the result shown in Fig. 1L. Also, I am not sure why only 25 TCs were analysed in Fig. 1N while there are the data to analyse more as shown in Fig. 1E (400 TCs), this would make the graph more representative.

      Figure 1 N represents a detail of the different phenotypes present in shot mutant embryos. Whereas for most of the paper we consider only complete lack of TC lumen, here we show the different types of affected TCs and not just the ones with a complete lack of subcellular lumen. We apologise because it was not explained in the original manuscript that types III and IV are the “no lumen” class (they were subdivided into 2 classes because they have different cell enlongation phenotypes). 36% of the total of affected TCs displayed the lack of lumen phenotype (this means a 22,5 % of the total number of TCs, because total affected TCs are 62,5% only). Numbers are similar but not exactly the same because this analysis was done using confocal microscopy and cells analysed one by one in detail, which is not possible using colorimetric methods and only luminal markers. This is also the reason we only analysed 25 TCs in this case. We thank the reviewer for pointing this out and have better described it in the manuscript.

      -p6, line 8: ShotA-GFP is indeed a long isoform but is not the full-length Shot, as it does not contain the plakin repeat exon which would add another ~3000aa.

      We have corrected this.

      -p6, lines 21-23: ShotA-GFP localisation is not shown in FigS1. The authors should refer to Fig. 2. Enlarged areas/arrows might help the reader to better visualise the different localisations of ShotA-GFP and ShotC-GFP.

      We thank the reviewer for this request and we will change the figure providing enlarged areas upon resubmission. In this version of the manuscript we have already changed the error in figure referral in the text.

      -p7, line 23: Rca1 mutants should be better introduced here.

      We have added one sentence of introduction to the Rca1 phenotype.

      -p8, line 6: Shot colocalizes/associates with stable MTs and actin would be a more appropriate title for this paragraph.

      We thank the reviewer for this alternative, and we have changed this title in the manuscript.

      -p16, line 18: 'Shot is able to mediate crosstalk' would be better than 'Shot is able to crosstalk'. -p40, lines 6 and 7: L, M and N should be K', K' and K' respectively. -p41, Fig 10D: It is quite hard to see on the cartoon what the phenotype is for Shot OE.

      We will make this clearer for resubmission.

      -The following reference shows an important role for Shot in crosslinking actin and MTs during morphogenesis of the Drosophila embryo and should be cited in this manuscript (Booth, A. J. R., Blanchard, G. B., Adams, R. J. & Röper, K. A Dynamic Microtubule Cytoskeleton Directs Medial Actomyosin Function during Tube Formation. Developmental Cell 29, 562-576 (2014)).

      We thank the reviewer for pointing this out, because this is of course an important reference known to us, which we forgot to add. We have now added this to the manuscript.

      -FigS3. It would be good to add the labels on the figure (ShotC-GFP in green, and MoeRFP/lifeActinRFP in Magenta).

      We will do this for resubmission.

      Reviewer #1 (Significance (Required)): The findings shown in this manuscript shed an important light on the way subcellular morphogenesis occurs. It was known that both actin and MTs were required in this process, particularly during the formation of Drosophila trachea (JayaNandanan, N., Mathew, R. & Leptin, M. Guidance of subcellular tubulogenesis by actin under the control of a synaptotagmin-like protein and Moesin. Nature Communications 1-10 (2019). doi:10.1038/ncomms4036; Gervais, L. & Casanova, J. In Vivo Coupling of Cell Elongation and Lumen Formation in a Single Cell. Current Biology 20, 359-366 (2010)). This work provides additional molecular insights into the way branching morphogenesis from a single cell occurs in vivo, clearly demonstrating a requirement for actin-MT crosslinking mediated by Shot and Tau. This could be of great interest in the field of branching morphogenesis and lumen formation, not only in invertebrates but also in vertebrates where such a crosslinking might occur in the vasculature, the lung, the kidney or the mammary gland for example (Ochoa-Espinosa, A. & Affolter, M. Branching Morphogenesis: From Cells to Organs and Back. Cold Spring Harb Perspect Biol 4, a008243-a008243 (2012)). *Field of expertise:* morphogenesis, Drosophila, cytoskeleton, microtubules. Reviewer #2 (Evidence, reproducibility and clarity (Required)): **Summary:** The development of branched structures with intracellular lumen is widely observed in single cells of circulatory systems. However the molecular and cellular mechanisms of this complex morphogenesis are largely unknown. In previous study, the authors revealed that centrosome as a microtubule organizing center (MTOC) located at the apical junction contributes subcellular lumen formation in the terminal cells of Drosophila tracheal system. The microtubule bundles organized by MTOC are suggested to serve as trafficking mediators and structural stabilizers for the newly elongated lumen. In this manuscript, they focused on a Drosophila spectraplakin, Shot, which have been reported to crosslink MT minus-ends to actin network, in the subcellular lumen formation. The paper started by description of lumen elongation defect of the tracheal terminal cells in the shot[3] null mutant. The overexpression of full-length and series of truncated form of shot exhibited extra-subcellular lumina (ESL) in TCs, suggesting that Shot is required for the lumen formation in dose dependent manner. They next addressed whether Shot overexpression induces ESL through the supernumerary centrosomes as in Rca1 mutant, however the number of centrosomes was not affected. Moreover, the ESL were sprouted distally from the apical junction, suggesting that Shot operate in different way from the Rca1-dependent microtubule organization. To get mechanistic insight of Shot in the luminal formation, they checked localization of the Shot and found it localized with stable MTs around the nascent lumen and with the F-actin at the tip of the cell during the cell elongation and subcellular lumen formation. In shot[3] mutant, the MT-bundles were no longer localized to apical region and the actin accumulation at the tip of the cell was also reduced. The rescue experiments using several truncated forms of Shot, and well-designed genetic analysis using various shot mutants revealed that both MT binding domain and actin binding domains are needed to develop the lumen. The expression of shot was under the regulation by terminal cell-specific transcription factor bs/DSRF, and the overexpression of shot in bs LOF mutant suppressed its phenotype, indicated that part of the luminal phenotype of bs mutant in terminal cells are due to lower levels of the activity of shot. Finally, they checked whether Tau can compensate the function of shot in the subcellular lumen formation. The lumen elongation defect in shot mutant was suppressed by tau expression, and tau overexpression phenocopied the shot overexpression-induced ESL. Although tau mutant did not show the lumen formation defects, the double mutant of shot and tau exhibited synergistic effect. Shot was also required for subcellular luminal branching at larval stages. Overall, this work highlighted the importance of Shot as a crosslinker between MT and actin that acts in downstream of the FGF signaling-induced bs/DSRF expression for the subcellular lumen formation. An excess of Shot is sufficient for ESL formation from ectopic acentrosomal branching points. Furthermore, the Tau protein can functionally replace Shot in this context. **Major comments:** *- Are the key conclusions convincing?* *- Should the authors qualify some of their claims as preliminary or speculative, or remove them altogether?* The conclusions were basically supported by the set of data presented in this article, but following points need to be clarified. The truncated form ShotC lacks only half of calponin domain that are essential for the actin binding, thus it is still possible to bind actin to some extent. Although the actin binding activity is reported as "very weak" in the cited references, the quantitative analysis has not been done. Thus, the interpretation and claims based on the experiments using ShotC should be reviewed carefully.

      We agree with the reviewer and will revise all the text for resubmission in order to make this unambiguous. However, we would like to remark that our claims are not only based on UAS-ShotC but also in the shotkakP2 allele, which does not contain one of the calponin domains and in isoforms such UAS-Shot C-tail which do not have any ABD.

      Data set in some places seems fragmented. For example, overexpression study of shot constructs (Fig. 2) lacks phenotypic comparison of control (btl Gal4 driven control FP) to compare if phenotypes of shot constructs expression are different from control. Different methods of phenotypic quantification are employed. One was counting embryo number with at least one abnormality among 20 TCs of DB or GB, or the other counting every TC for the presence of lumen/branching conditions. The latter is more stringent measure and is more appropriate for the study of single cell morphogenesis.

      We totally agree with the reviewer. We have now revised all quantifications and graphs:

      1) We have used btl>GFP as control to all overexpression experiments in embryos and DSRFGAL4UASGFP in control larvae.

      2) We have made the paper uniform regarding quantifications, which are now all done in relation to total TCs and not embryos.

      For this reason, many of the graphs, figure legends and quantification values in the the manuscript text are now changed.

      *- Would additional experiments be essential to support the claims of the paper? Request additional experiments only where necessary for the paper as it is, and do not ask authors to open new lines of experimentation.* The all movies were using ShotC isoform which lacks half of the actin binding domain. The truncated isoform is not suitable to observe the localization, especially the colocalization with actin. The movies need to be retaken using full-length Shot at the dosage that does not interfere with normal TC development.

      We agree and we will analyse movies with both ShotC and ShotA for resubmission.

      Some statements on Moesin and Tau localization sound as if the authors studied Shot interaction with nascent Moe and Tau molecules. This is confusing because fragments of Moe and Tau, but not functional full length proteins, were used.

      We will revise the text to make this unambiguous fir resubmission.

      *- Are the suggested experiments realistic in terms of time and resources?* It would help if you could add an estimated cost and time investment for substantial experiments. Because the transgenic fly is already present, we assume it would be done in 4 weeks. However, it would be influnced under social circumstances whether the lab facilities are able to access or not. *- Are the data and the methods presented in such a way that they can be reproduced?* *- Are the experiments adequately replicated and statistical analysis adequate?* The methods provided seem to be sufficient for reproducing the data by competent researchers, and most of the data are solid and the sample numbers are sufficient for the claims. However, the criteria for phenotypic evaluation differs among graphs and figures, that possibly confuse the readers. Standardized measurement methods are desirable. **Minor comments:** *- Specific experimental issues that are easily addressable.* In the rescue experiments shown in Figure 6, only full-length Shot rescued the subcellular lumen formation, but either of truncated Shot did not. The localization study of MT and actin in those conditions will reveal whether proper localizations of actin and MT are critical for the lumen formation.

      We are working on this for resubmission. These experiments were stalled by the current COVID-19 pandemic and this is why they were not submitted with the first version. We will provide MT and actin localization for the rescue experiments with ShotA and ShotC.

      *- Are prior studies referenced appropriately?* The references are cited appropriately. *- Are the text and figures clear and accurate?* There are several typos: Remodelling -> remodeling, signalling -> signaling. In the figure 2, G and H seem redundant. Scale bars are missing in Fig1 F-K, Fig2 K-L, Fig6 A-I, Fig7 E-J and Fig8 E-J.

      We have changed the graphs in figure 2. Typos have been corrected. We will provide errors bars for resubmission.

      The author often called shot+ genotype as "wild type". They are transgenic strains with some mutations, and cannot be found in the wild. They should be simply called with genotype or "control" for experiments.

      We thank the reviewer for pointing these typos and incoherences with control genotypes. We have partly revise the text and figures and will finish for resubmission.

      *- Do you have suggestions that would help the authors improve the presentation of their data and conclusions?* In Figure 4, as the localization of Shot is difficult to see in detail, enlarged insets might help. In addition, the green and cyan in C'-E' is difficult to distinguish.

      We will change this for resubmission.

      With Figure 5, the authors claimed that Shot LOF leads to disorganized MT-bundles and actin localization. We feel this is an overstatement and the Figure should be backed up with better data, or removed. F-actin and microtubule localizations are highly dynamic and the snapshot pictures are insufficient for demonstrating defective localization. It is also possible that (potential) difference in the marker localization is due to indirect effect of Shot LOF in cell shape.

      We agree with the reviewer that fixed samples are not the best to analyse cytoskeletal components, but we observe clear differences in MT bundles and specially in actin localization in shot mutants as compared to controls and we believe it is important to show these results. Cell shape might of course alter the analysis which is why we present 3 different cell shapes in Figure 5. In addition, there are many previous studies where localization of MTs and actin was done in fixed mutant embryos, where cell shape is also affected, and revealed important steps in TC formation (Gervais and Casanova, 2010; JayanNadanan et al. 2014).Nonetheless, we have revised the text in order to avoid overstatements.

      Reviewer #2 (Significance (Required)): *- Describe the nature and significance of the advance (e.g. conceptual, technical, clinical) for the field.* *- Place the work in the context of the existing literature (provide references, where appropriate).* In blood capillary and insect trachea, the branching process of single vessel cells involves sprouting of cell protrusions, followed by the lumen extension from the main vessels. The lumen formation involves assembly of plasma membrane components inside of the cytoplasm. Since the luminal membrane is associated with protein complexes common to apical cell membrane, lumen formation is believed to involve redirection of apical trafficking of membranes to intracellular sites (Sigurbjörnsdóttir, Mathew, Leptin 2014, 10.1038/nrm3871). The authors previously demonstrated that centrosome is an important link of preexisting lumen to de novo lumen formation, leading to the hypothesis that centrosome-derived microtubules organize lumen membrane assembly. *- State what audience might be interested in and influenced by the reported findings.* In this manuscript, the authors addressed this issue by looking at the function of Shot/Plakin that has both microtubule and actin binding activities. Shot is an ideal candidate for linking actin-rich cell protrusions in the leading edge to centrosome- associated lumen tip. Indeed the authors clearly showed that shot is required for lumen extension and overexpressed shot protein associates with intracellular tract rich in microtubules and F-actin. Their findings are definitely a progress in the field of Drosophila tracheal development. Having said that, how Shot links leading edge protrusions and centrosomes, how it is organized into pre-lumen tract, and how it contribute to further assembly of luminal membrane and directed secretion, are not well understood yet. Without clues to those fundamental questions, I believe this paper is most appropriate for experts readers of Drosophila cell biology and tracheal development. Finally I feel that the paper include many data sets and some pictures are not easy to grasp essential points, such as three movies showing localization of overexpressed shot-C, RFP-moesin, and Lifeact. *- Define your field of expertise with a few keywords to help the authors contextualize your point of view.* Drosophila, tracheal cell biology. *- Indicate if there are any parts of the paper that you do not have sufficient expertise to evaluate.* No Reviewer #3 (Evidence, reproducibility and clarity (Required)): **Summary** In their manuscript entitled "Coordinated crosstalk between microtubules and actin by a spectraplakin regulates lumen formation and branching" Ricolo and Araujo characterize the requirement for Short Stop (Shot) in the formation of subcellular tubes in tracheal terminal cells. The authors examined embryos homozygous for shot3, a presumed null allele of shot. They found an 80% penetrant defect in seamless tube formation or growth. The phenotype resembles that reported for mutations in blistered, which encodes the Drosophila SRF ortholog. The authors find that expression of SRF is not blocked by mutations in shot and later find that bs mutants have decreased levels of shot expression and that shot overexpression can partly suppress the bs tube formation defects. The authors then examine whether the requirement for shot is autonomous to the trachea and find that it is, as pan-tracheal shot RNAi replicates the seamless tube defects. The authors find that overexpression of various Shot isoforms results in the formation of ectopic seamless tubes within terminal cells. Using the various transgenic constructs available for shot, the authors show that the overexpression phenotype is dependent upon the interaction between Shot and microtubules, and is dose-dependent. Previous work had shown that ectopic terminal cell tubes also can arise due to increased centrosome number; the authors show that centrosome number is not altered in shot mutants. Shot has well characterized actin and microtubule binding functions, and the authors show that Shot localization overlaps both with microtubules and with actin, and that both cytoskeletal elements are aberrant in shot mutant cells. In a series of experiments utilizing various shot mutant backgrounds and shot transgenes, the authors identify requirements for both Shot-cytoskeleton interactions in the formation and branching of seamless tubes in terminal cells. Finally, the authors examine the requirement for Tau in the same processes. Tau and Shot had previously been found to work together in neurons, and this seems to be true in terminal cells as well. Tau overexpression induces ectopic seamless tubes and can partially suppress shot loss of function. Embryos mutant for tau showed seamless tube directionality defects, but not lumen formation or branching. Embryos doubly mutant for tau and shot showed a more severe seamless tube defect than shot mutants alone - an increase in terminal cells with no lumen from 22% to 85%. Authors also examined terminal cells in larval stages using dsrf-Gal4 to knockdown shot in terminal cells (rather than pan-tracheal knockdown with breathless). The authors conclude from their studies that Shot, through its interactions with microtubules and the actin cytoskeleton coordinate the outgrowth and branching of subcellular tubes. Overlapping function of Tau and possibly other additional MAPs also act in these processes. The work is largely well done and the conclusions are supported by the data. **Minor concerns:** -If one were to start this work today, crispr knockout and knockins would be preferred. While shot^3 is widely considered a null allele, there are indications that some shot function is still present in shot^3 embryos. This would also be relevant to the penetrance of the defects. The transgenes are useful, but given the dosage effects noted in various of the authors experiments, interpretation of some experiments is complicated as compared to a knockin. For overexpression experiments, landing site constructs would be preferable. I do not mean to suggest that the authors necessarily go this route, but am just pointing out a limitation of the approach.

      We agree, but we also think that with the amount of data and tools generated by other labs over recent years, regarding shot function in the nervous system (Voelzmann et al 2017), we are in a position to be able to take the conclusions of this work based on these transgenic and different shot alleles.

      -Insight into function at higher resolution than altered microtubule and actin organization would significantly increase the impact. -cell autonomy (line 19, p5) is not the correct term. Pan-tracheal knockdown tests tissue autonomy. Mosaic analysis or terminal cell specific knockdown would address cell autonomy.

      We have changed the manuscript accordingly.

      -line 14 p6 acting should be actin -dsrf-Gal4 transgenes were made by Mark Metzstein

      We have corrected these.

      -there also appears to be rescue of the fusion cell defects of shot by Tau overexpression. Authors should comment on this and what it means for the seamless tubulogenesis program in terminal cells vs fusion cells.

      We will reanalyse shot rescued with tau embryos focusing on fusion phenotypes and discuss this in the revised version.

      Reviewer #3 (Significance (Required)): The findings will be of interest to a broad cell biology community as they provide a conceptual advance and may help to focus future work on seamless tubulogenesis. The authors do a good job of placing the results in the context of previous studies. *Field of expertise:* Drosophila, tracheal tubulogenesis, developmental biology

    1. That is why it is ridiculous to expect college institutions to be radical.

      I think this is correct and tied to the neoliberal management style of the modern university. Individual faculty members may be "radical," (however you define that), but institutions as a whole tend to mitigate any attempt at change. How did we get here and how do we change it?

    1. As a member of this world we call a society, a mother, a daughter, and a sister it can give me the sense of drowning. I have all the responsibilities to those roles, some since the time I was born, so you would think I could balance it all well. Having a strong, local support system is vital to my well-being, but unpacking and examining our worldviews is important for my well-being too but can also have far reaching consequences. Am I making the right choices or creating a distorted reality for myself to escape to? Be it from church, family, friends, school, or coworkers I am still learning how to value and identify myself as well as the wider context around me.

      Your piece starts pretty abstract here with some big ideas. Consider maybe starting with something concrete--an image, an anecdote, etc. and then moving from concrete to abstract. Abstraction puts a heavier cognitive demands on the reader and may feel less engaging initially...

    1. We professors may have been energetic and eager about discussing the issues in our classrooms

      or maybe not? Do you think it's common for faculty to want to engage students beyond their discipline? Is this interest more common among faculty teaching certain disciplines (e.g. history, political science, philosophy) as opposed to faculty in other fields (e.g. science, engineering, education)? If faculty aren't inherently interested in teaching about civic engagement, should they be? Why?

    2. Fostering Civic Engagement across the CurriculumNow that media coverage has waned, it is the time for reflecting on last year’s highs and lows as we make improvements for the future. Extensively covered in the media, the midterm election cycle was a 2018 event for the history books. These developmentsstill presenta unique opportunity for teaching civic engagement to students. We professors may have been energetic and eager about discussing the issues in our classrooms. On the contrary, some of our students might have lacked the same enthusiasm. Othersmay have expressed outright anxiety regarding the political process. What can we do to help with such issues in the future?

      This is a timely and important topic. The piece is currently written through the eyes of a professor and is addressed to other professors. I think this is absolutely fine, but it may limit the potential audience. Is it only faculty that have the opportunity to help students gain a better understanding of and appreciation for the election process and the role of civic engagement? Would any of these strategies be useful to others, such as advisors, administrators, ...

    1. Effective Headhunting Techniques for Hiring Leave a Comment / Blog Contact Headhunters are individuals who are employed by an organization or enterprise to discover, vet, and present reasonable possibility for a vocation position. The headhunter’s main responsibility is to guarantee the competitor has the right range of abilities for a position and they’re frequently recruited to discover possibilities that have a one of a kind or elusive range of abilities. When in doubt, headhunters are self-employed entities or workers of an enlisting organization. They are recruited and appointed by an organization searching for top notch ability and regularly work for a few organizations at a given time. It’s normal for headhunters to have some expertise in a field, for example, tech or showcasing. Headhunting recruitment permits them to filter through resumes to find the most ideal contender for an occupation speedier and all the more proficiently. 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At the point when you figure out how to scout the correct way, finding the most qualified competitors is simpler than any time in recent memory. Here are five hints to help your area of expertise scout the best applicant. Have HR Gotten the Message Out With Workers To discover profoundly qualified contenders for your activity, start by tapping your current representative system. To do this, have your area of expertise spread some verbal data about the position that is open, the sort of up-and-comers you’re searching for, and the necessities for the post. With any karma, your worker system will impart the news to their certified companions with your area of expertise, which serves to bring you preferable ability over you could have in any case gotten to. On the off chance that you’d prefer to make this a stride further, think about posting updates on your accessible opening via web-based networking media. This makes it simple for your current representatives to impart the post to their certified loved ones. Your area of expertise ought to likewise make posts on LinkedIn and Twitter or head to a public exhibition, where your workers can get the message out of your situation to passers-by. Send Representatives into Your Trade Groups To enhance your scouting procedure, urge your HR division to scour the exchange bunches you take an interest in. Since you’ve just got these associations, presently is an excellent opportunity to benefit as much as possible from them. To begin, plan a portrayal of your activity and send it out to chiefs, thought pioneers and industry heads in your specialty. You may likewise decide to utilize phone systems administration to get the message out about your accessible position and get individuals intrigued. While organizing everywhere is a keen method to draw in more contender to your position, organizing inside your trade groups is exceptionally focused on method of arriving at new up-and-comers. Since trade groups draw in a quite certain gathering of individuals, they’re extraordinary spots to search for applicants with exceptional aptitudes or interests. This, thus, chops down the time you’ll spend scouting and guarantees you’ll locate a superior applicant quicker and simpler. Optimize Your Site Your site is your biggest resource, so why not use it to get the message out about your vacant position? In the event that you don’t have a Careers page, your area of expertise should work with the organization’s tech or IT group to make one at this point. In the event that you do have a Careers page, be certain it includes a compact, elegantly composed set of working responsibilities that will interest possible competitors. Recollect that there are many dry, slow sets of expectations out there, which implies you’ll do well to make yours drawing in and captivating – consider recruiting proficient journalists in the event that you need help. The more you can make your set of working responsibilities energizing, the almost certain it is you’ll draw in all around qualified, drew in, intrigued up-and-comers who will smooth out your scouting procedure and make it simpler to fill your vacant position a lot quicker. Catch Up With Intrigued Leads In the event that you have leads intrigued by your task, make certain to catch up with them in like manner. Keeping in touch with the leads who contact your organization about an occupation is a brilliant method to prop the energy up and guarantee these gifted candidates consider your to be as a drew in and dynamic business. While there are many approaches to stay in contact with intrigued up-and-comers, probably the most practical are welcoming possibility to buy into a bulletin, lining up after a meeting with email, and making a pre-programmed message that thanks intrigued contender for their applications. Guaranteeing your area of expertise is dynamic about getting the telephone and calling up-and-comers can help, too. Take advantage of Innovation There are many approaches to enroll new applicants utilizing the web. Notwithstanding making posts via web-based networking media, it’s anything but difficult to utilize the web to post positions on proficient scouting locales, merchant destinations and arranged advertisement locales. The web likewise gives a direct and smoothed out approach to check the viability of your post. In case you’re not getting the candidates you need, consider changing your methodology likewise. At the point when you outfit innovation in your scouting, it’s simpler to arrive at a more extensive choice of up-and-comers, guarantee your activity posting mirrors the quality and commitment of your organization and win yourself a situation among the positions of the most wanted managers. Being Conceivable This is a key point. When directing an official pursuit, high-esteem applicants don’t need their time squandered with theoretical openings for work. On the off chance that the selection representative doesn’t accept that the up-and-comer they’re reaching is directly for the job, in what capacity will the applicant ever trust it themselves? As selection representatives, we make it understood to every competitor we approach that they are an ideal fit. Furthermore, this isn’t simply a mystery. In light of our examination, we realize that each individual we converse with is an ideal choice for the activity. Advocating Straightforwardness We’ve all run over employments that look excessively great to be valid. This absence of genuineness, shockingly, prompts numerous up-and-comers being suspicious about jobs they’re advertised. Talent scouts get this, which is the reason they champion straightforwardness. It bodes well. You wouldn’t have any desire to be told about work that sounded intriguing however was totally unique when you discovered increasingly about it, okay? Since talent scouts are specialists in your industry, they are well prepared to introduce openings for work obviously and answer any inquiries precisely. This gives the competitor certainty and guarantees the enrollment procedure is a smooth one.

      Headhunting is a practice that involves prospecting a candidate for a specific position. using effective Headhunting techniques the hiring becomes effective.

    1. Who Are Your Learners? Before we begin teaching it is important to know who our learners are. They will come to courses with different goals, motivations, learning styles and prior experiences. One way for a new teacher to gain an understanding of their learners is to create a learner profile. This can be done using information from your student management system, student interviews or other such information sources. Below are some questions to consider as you think about yourself as a learner in this course: How do you learn? What type of learning environment do you feel most comfortable in? i.e. classroom, online, workplace, one to one training? What are some of the barriers to learning that you face? i.e. work, family, illness, transport, financial stress How can you overcome some of your barriers? Do you have a process to learning? Edit Edit settings Move right Move left Duplicate Assign roles Delete   Edit Edit settings Move right Move left Duplicate Assign roles Delete    Creating Your Own Learner Profile If you were unable to attend the Induction workshop, click on the web icon link below, read through the information and answer the questions in the Activity to create your learner profile. Add some of your adult learner characteristics to the discussion forum. As you do this think about how you would cater for all the differing characteristics that your learners may have. Edit Edit settings Move right Move left Duplicate Assign roles Delete     Edit Edit settings Move right Move left Duplicate Assign roles Delete     Click on the Web icon to open a Wiki educator page about Knowing the learner. Read through the web page then complete the activity below to develop your own learner profile. (Note: The link will open in a new webpage) Edit Edit settings Move right Move left Duplicate Assign roles Delete   Edit Edit settings Move right Move left Duplicate Assign roles Delete    Adult Learning Theory Adult learners may be concerned about participating and losing their dignity, may have a fear of being exposed or participating in group work, may be uncomfortable with authority, and may doubt their abilities. To teach effectively requires an understanding of how learners learn and we will be looking more deeply into adult learning theories in Module 2: Learning and Teaching. To gain further insight into our learners let's look to Malcolm Knowles (1980). He identified he needed to understand how adult learners learned and in response developed a theory of Andragogy. He claims that in virtually all adult learners there is a need to be self-directed and he made six assumptions about how adults constructed knowledge (Brennan Kemmis & Atkins, 2014): Need to know Foundation Self-concept Readiness Orientation Motivation

      Could this all go into Module 1 somehow? A new topic? Where does each part sit?

    1. Note: This rebuttal was posted by the corresponding author to Review Commons. Content has not been altered except for formatting.

      Learn more at Review Commons


      Reply to the reviewers

      The response to reviewers consists of three parts:

      1. A summary of the main points from the two reviews, and the authors' response to these points.
      2. A detailed revision plan for the preprint, taking into account both the main points of the reviews, and other comments made by the reviewers.
      3. A point-by-point response to the reviewers.

      For figure citations, OV = old version, i.e. bioRxiv preprint 2019-826180v2, and NV = new version, i.e. revised and re-submitted version.

      1. Summary of main points by the reviewers, and authors’ responses:

      • Both reviewers felt that the manuscript was overlong; Reviewer 1 recommended either shortening it or splitting it into two stories, while Reviewer 2 recommended cutting down the text.
        • We have considerably shortened the manuscript in accordance with this request (see revision plan below). We had already considered splitting the manuscript into two parts during the drafting stage, and had rejected this possibility as the data are intertwined - the retroactive validation of the dimer interface by the mutagenesis constructs (OV Fig. S3 [NV Fig. S4]) being a good example.
        • The revised manuscript features 7 main figures and 13 supplementals.
      • Both reviewers felt too much text and figure space was allocated to negative data, specifically the investigation of potential lipid binding by the TbMORN1 protein, and that there should be more focus on the positive parts of the story.
        • A key part of shortening the manuscript has been moving most of the negative data on lipid binding into the supplemental figures, and considerably shortening the associated text. This has allowed the main figures and associated text to focus more on the positive elements of the project, while still ensuring publication of all the data.
      • The reviewers appear to be in slight disagreement concerning discussion of the data. Reviewer 1 has encouraged more speculation on the physiological role of PE binding, a potential lipid transfer function, a role for calcium ions, the relevance of the observed disulphide bond, and the role of zinc ions in apicomplexan proteins; Reviewer 2 has recommended avoiding excessive speculation or inference.
        • Given that both reviewers have agreed that the original manuscript was overlong, we have implemented Reviewer 2's suggestion here and reduced the amount of speculation in the revised text.
      • The reviewers agreed that the technical quality of the data was high and that the conclusions drawn were robust.
        • We are glad that the reviewers were appreciative of the data quality. For this reason, we were reluctant to remove any of the data from the manuscript and would prefer instead to transfer it to the supplementals. We feel that the negative data still have considerable community value, given that they show that MORN repeats are not automatically lipid binding modules and can thus act as a caveat to other researchers.

      2. Detailed revision plan for the preprint:

      • We have implemented the reviewers' suggestions and substantially shortened the manuscript, primarily by trimming the (phospho)lipid-binding section, which contains a large amount of negative data. The following main figures have been moved into the supplemental section:
        • OV Fig. 2 ("TbMORN1 interacts with phospholipids but not liposomes") has become NV Fig. S2
        • OV Fig. 4 ("TbMORN1(2-15) does not bind to liposomes in vitro") has become NV Fig. S6
        • OV Fig. 8 ("Conservation and properties of residues in TbMORN1(7- 15)") has become NV Fig. S11
      • This has left a total of 7 main figures and 13 supplementals.
      • The text associated with the entirety of the lipid-binding part (OV lines 210- 530, OV Figs. 2-6 [NV Figs. 2-4, S2, S6], OV Supplemental Figs. 2-6 [NV Supplemental Figs. S3-S5, S7, S8]) has been condensed. The focus of this section is now on the positive parts of the data: the PE association (OV Fig. 3 [NV Fig. 2]) and the in vivo work (OV Figs. 5, 6 [NV Figs. 3, 4]).
      • We have additionally limited the amount of inference and speculation in the manuscript.

      3. Point-by-point responses to the reviewers

      Reviewer #1 (Evidence, reproducibility and clarity):

      MORN (membrane occupation and recognition nexus) repeat proteins are found in prokaryotes and eukaryotes. They feature characteristic repeats in their primary sequence, have been assumed to play a role in lipid binding, but remain poorly characterized on the functional and structural level. This manuscript tries to address both these questions and is organized in major parts. In the first part the authors characterize a putative role of MORN repeat proteins in lipid binding and membrane association. In the second part, the authors use X-ray crystallography to establish the structure of MORN repeat proteins and to investigate the dimerization.

      As a cleverly chosen point of departure, they focus their study particularly on MORN1 from Trypanosoma brucei (TbMORN1), which is composed solely on MORN repeats. The structures of MORN repeats (from several species) in part two provide interesting insights into their mode of homotypic interactions and their role as dimerization or oligomerization devices. The lipid binding and membrane association of MORN proteins in the first part remains somewhat confusing and unclear, despite the use of a whole battery of techniques.

      We anticipate that the shortening and refocusing of the lipid binding data has addressed this issue.

      It is questionably, why the authors invest so many figures and words to inform the reader on negative results.

      We have chosen to publicise our negative data in full because, as noted in the manuscript, there is a widespread and erroneous assumption that MORN repeats are lipid binding modules. We feel that publishing these data will allow them to act as a caveat to other researchers working on MORN repeat proteins. We have, however, addressed the reviewer's request in that we have considerably shortened the text associated with these data and have moved the corresponding figures into the supplementals.

      The authors suggest that MORN proteins can bind to lipids via their hydrophobic acyl chainswhich is 'very hard to imagine under physiological conditions unless TbMORN1 is a lipid carrier and not a membrane-binding proteins. Unfortunately, a role as lipid carrier has not been rigorously tested.

      The reviewer is correct that we have not specifically tested for a function as a lipid carrier protein and although this was only speculation, it has been toned down accordingly.

      In this sense the first part remains somewhat immature and incoherent. Furthermore, they suggest based on the lack-of-evidence that MORN proteins do not bind membranes in vivo and in vitro.

      We are not clear where this suggestion was made. Our data indicate that TbMORN1 does not directly bind membranes in vivo or in vitro, and we therefore noted that putative lipid binding by other MORN repeat proteins should be viewed with caution. Specifically, we stated in the Discussion (OV lines 955-956) that "the presence of MORN repeats in a protein should not be taken as indicative of lipid binding or lipid membrane binding without experimental evidence". Again, our expectation is that the major changes planned for the data presentation in this section will make it more coherent.

      The main issue of this manuscript is, in my view, the way the data were presented.The manuscript is generally well-written, but much too long. The structural work is important and concise.

      We have considerably shortened the manuscript as per the reviewer's request, and especially the section on lipid binding.

      The first part, however, reports in five separate figures on a lack of membrane binding by a MORN protein and its ability to bind individual lipids. The physiologically relevance of this lipid binding is questionable as acknowledged by the authors.

      We have moved two of these figures (OV Figs. 2, 4) into the supplementals section [NV Figs. S2, S6], shortened the associated text, and limited the amount of speculation.

      Even though I find it important that the membrane/lipid binding ability of MORN proteins is rigorously tested, I would highly recommend to separate the current manuscript in two independent stories. Alternatively, I would recommend to reduce the first part into a single figure and to remove the most artifactual assays.

      We have implemented the second of these two suggestions for the manuscript. We had already considered splitting the manuscript during the drafting stage, but rejected this possibility as the data were too intertwined. Consequently, we have opted to considerably reduce the first part, and moved OV Figs. 2 and 4 into the supplementals [NV Figs. S2, S6]. We would prefer not to remove data altogether as they are likely to have community value even if they are negative and as noted, they are of good quality.

      In the current form, the first part and the second part of the manuscript remain somewhat detached from each other. The characterization of the lipid binding/membrane binding properties has a number of substantial weaknesses (e.g. use of quite different, nonphysiological buffers for membrane binding assays; use of deletion mutants for the binding assays, which do not show the full potential of oligomerization). This which makes it hard to read and confuses the reader. Even though I have no reason to doubt the conclusions by the authors, I do not think that all necessary caution has been invested to rule out other possibilities.

      We believe that the shortening and refocusing of the manuscript should address these issues. For consideration of the buffer and deletion mutant points, please see responses to Major Points below.

      In summary, even though the technical quality of the individual performed assays is high, there are some conceptual issues that make it hard to make a strong case based on a collection of individual, clear datasets. Even though I find the structures of the MORN proteins important, timely, and interesting, I would not recommend this study for publication in its current form. The manuscript would be more fun to read if both of the parts would be shortened substantially and more focused.

      We have implemented this suggestion: the manuscript has been considerably shortened (from 20,489/135,073 to 18,555/103,988 characters/words, focused on reducing the negative lipid-binding results).

      While I agree that most evidence provided on lipid/membrane binding of TbMORN1 argue against a direct role of MORN proteins in membrane binding, I feel that the experimental approach is not coherent enough. See a few major points of criticism below.

      Major Points:

      1. The authors decide to characterize the membrane binding of a MORN repeat protein using a deletion variant that lacks the N-terminal repeat. However, in Figure 1B they show that the N-terminal repeat is important for the formation of higher-order oligomers. While I fully understand that the presence of the most N-terminal repeat does hamper the structural work, I find it problematic to remove it for the lipid/membrane-binding assays. The formation of higher oligomeric species beyond the dimer, may be important for membrane binding/recruitment (avidity effects).

      As we explained in the manuscript, the reason for not using the full-length protein for in vitro work was because it was polydisperse, and that the yields were extremely low. See OV lines 178-179 ("The yields of TbMORN1(1-15) were always very low, making this construct not generally suitable for in vitro assays".) and OV lines 411-414 ("...TbMORN1(1-15), which was polydisperse in vitro and formed large oligomers (Fig. 1B). The membrane-binding activity of these polydisperse oligomers was not possible to test in vitro, as the purification yields of TbMORN1(1-15) were always low."). Consequently, we used the longest construct that was suitable in terms of chemical and oligomeric homogeneity. Using the full-length protein would have had inherent problems with aggregation, and consequently would have compromised the data and derived results. In order to make this clear in the manuscript we edited the sentence mentioned above as follows:

      “It was not possible to test the membrane-binding activity of these polydisperse oligomers in vitro however, as the purification yields of TbMORN1(1-15) were always low. As an alternative, the possible membrane association of TbMORN1(1-15) was examined in vivo."

      2) (Related to point 1) I do not understand the choice of the buffers used for some of the assays. The use of pH 8.5 and NaCl concentrations of 200 mM are non-physiological.

      These were the buffer conditions required to retain the protein in a monodisperse state, suitable for in vitro assays.

      For CD spectroscopy, a high ionic strength was obtained by the use of 200 mM NaF. If a high ionic strength is required to prevent the formation of higher oligomers of MORN, it raises the question if the formation of higher oligomers (under physiological conditions) may also contribute to their function.

      The oligomers of TbMORN1 may indeed be the most functionally relevant form of TbMORN1 but we do not currently have a means of testing this in vitro, as acknowledged in the text (OV lines 411-414, quoted above). The aim of CD spectroscopy was to assess fold integrity and stability of different constructs; we used buffers as recommended for the CD spectroscopy experiments by Kelly et al, 2005 (doi:10.1016/j.bbapap.2005.06.005) (Table 1 and section 4.2). Furthermore, the CD spectra of TbMORN(1-15) and TbMORN(2-15) (OV Fig. S1E [NV Fig. S1E]) are basically superimposable, suggesting identical secondary structure content at the concentration used for these experiments.

      It is unclear, in which buffer the fluorescence anisotropy measurements were performed.

      We have provided details on the buffer conditions for the fluorescence anisotropy experiments in the Materials and Methods section, NV page 23, lines 962-963.

      The sucrose-loaded vesicles were hydrated in a 20 mM HEPES pH 7.4, 0.3 M Sucrose. The composition of the buffer after the addition of MORN proteins is not clear.

      The Materials and Methods are now unambiguous on this point. Please see NV lines 1036- 1046: "6 μM Rhodamine B dihexadecanoyl phosphoethanolamine (Rh-DHPE) was added to all lipid mixtures to facilitate the visualisation of the SLVs. The lipid mixtures were dried under a nitrogen stream, and the lipid films hydrated in 20 mM HEPES pH 7.4; 0.3 M sucrose. The lipid mixtures were subjected to 4 cycles of freezing in liquid nitrogen followed by thawing in a sonicating water bath at RT. The vesicles were pelleted by centrifugation (250,000 × g, 30 min, RT) and resuspended in 20 mM HEPES pH 7.4, 100 mM KCl to a total lipid concentration of 1 mM. SLVs were incubated with 1.5 μM purified TbMORN1(2-15) in gel filtration buffer (20 mM Tris-HCl pH 8.5, 200 mM NaCl, 2% glycerol, 1 mM DTT) at a 1:1 ratio (30 min, RT)." The liposomes were at physiological pH and close to physiological ionic strength.

      Despite the use of an impressive array of techniques, this first part of the manuscript remains somewhat immature and incoherent. Due to the use of constructs that have not the full ability to oligomerize (point 1) and due to the inconsistent use of experimental conditions, it is hard to draw firm conclusions from this first part.

      Any biochemical study is conducted within the constraints of the choice of construct and the choice of buffer conditions, and the data are valid within those parameters. This applies as much to positive data as to negative data, so we are not clear why the reviewer is placing such emphasis on this point. In the case of the LiMA data, which are the most unbiased and comprehensive dataset in the manuscript, these experiments were well-controlled and there were also domains present that were recruited to membranes under the buffer conditions, allowing us to rule out that the assay conditions were completely unsuitable. Validating negative results should be done as carefully and with as many orthogonal approaches as the validation of positive results. The reviewer acknowledges below that "the data point in the direction that MORN proteins (or at least TbMORN1) does not directly bind to membranes". This is the conclusion that we wanted to communicate.

      For example: In Figure 2E TbMORN(2-15) does show some concentration-dependent binding, which -however- is interpreted as background binding. What are the results using this assay (or better: a liposome floatation assay) when using full-length TbMORN(1-15) in a more physiological buffer?

      As noted already, it is not possible to use the TbMORN1(1-15) construct for in vitro assays owing to the extremely low yields and polydisperse nature of the protein. The excess fulllength protein was associated with the cytosolic fraction and not the membrane fraction in vivo (OV Fig. 6B [NV Fig. 4B]).

      The statement that MORN proteins bind to lipids, but not to liposomes/membranes is -in my view- not sufficiently addressed to make a strong case.

      At no point do we suggest that MORN repeat proteins in general bind to lipids and not to liposomes/membranes. On the contrary, and as detailed in the manuscript, we set out to assay the lipid binding activity of TbMORN1, found that it appears to bind to lipids but not to liposomes/membranes, and have therefore cautioned that lipid or liposome/membrane binding of other MORN repeat proteins must be tested experimentally before claims of function are made.

      3) The physiological relevance of lipid binding to MORN proteins remains obscure (as also acknowledged by the authors). Does the binding of PE lipids to the MORN protein have a physiological role? Does the binding of fluorescent PI(4,5)P2 point to a physiological role of MORN proteins?

      These are interesting questions that we would like to address in future work.

      4) In light of recent data from the Chris Stefan lab (PMID: 31402097) a co-incidence detection of PI(4,5)P2, PS, and cholesterol seems possible. Can the authors address this possibility?

      Again, the involvement of cholesterol, PS, and PI(4,5)P2 would be interesting questions for subsequent work but are beyond the scope of the present study. We did partially address this issue in our use of PI(4,5)P2, POPC and cholesterol containing liposomes in liposome cosedimentation assays, which showed no binding (OV Fig. S3A [NV Fig. S4A]).

      Furthermore, the role of Ca2+ signaling / Ca2+ ions has not been addressed. In light of the important role of Ca2+ for the recognition of PI(4,5)P2 (PMID: 28177616), this point should be addressed.

      We carried out liposome pelleting assays in the presence of Ca2+ and Mg2+, and saw no binding by TbMORN1(2-15) in either condition (see data below). These data were not included in the MS because of the insufficient number of technical replicates available.

      5) For characterizing the binding of lipids to MORN proteins, the authors use nonphysiological fluorescent and short-chain lipid analogues at concentrations, which are unlikely to occur for endogenous PIPs in the cytosol of cells. Why choosing such an artificial system? Why introducing this system at length, if other -less artifact-prone- assays are available? I would recommend to not feature this assay as prominently as it was in the current study.

      Our aim was to stick to using the same fluorophore throughout all the experiments. The choice of short-chain lipids was constrained by what was commercially available with the BODIPY TMR fluorophore. We have implemented the reviewer's suggestion in the manuscript, and the text associated with the fluorescence anisotropy assays has been considerably shortened. We are aware that the chosen concentration of the fluorescent lipids was out of physiological range, but the requirements of the fluorescence anisotropy itself necessitated a compromise. The possible shortcomings of the fluorescence anisotropy assays are, we believe, more than amply compensated by the LiMA data.

      6) How would PE find its way to the lipid binding region in MORN? Would it diffuse to the MORN protein via the aqueous phase or would the MORN protein pickup PE form membranes up collision? The authors should address this point, by separating the lipiddepleted MORN protein from donor-vesicles containing PE by a dialysis membrane. If PE would not find its way to the lipid binding site of MORN, this would imply that MORN protein can extract lipids only upon colliding with the membrane. What is the stoichiometry of PE to MORN?

      These are all interesting questions that we would like to pursue in subsequent work, but we feel that they are beyond the scope of the present study. Until we have conditions suitable for obtaining high yields and monodisperse populations of the full-length protein, which probably also necessitates developing conditions for controlled oligomerisation, it would be premature to start this. As to how it picks up PE: it is well known that specific lipid binding/chaperoning proteins can deliver their lipid cargo to other proteins. Additionally, proteins that bind lipids use hydrophobic domains to both interact with and sequester fatty acids and/or lipids from membranes. The literature is populated with lots of such examples. https://www.sciencedirect.com/science/article/pii/S0092867416310765.

      Despite my critique raised above, I agree with the authors that the data point in the direction that MORN proteins (or at least TbMORN1) does not directly bind to membranes. Their data, however, would still be consistent with a role as lipid transfer protein and a recruitment of MORN proteins to the membrane by other proteins. Have the authors performed any additional experiments in this direction? Also, the potential role of palmitoylation is only mentioned in the discussion (page 22), while palmitoylation would provide a simple means for membrane recruitment.

      We are glad that the reviewer concurs with our main conclusion. We agree, as noted in the discussion, that a role as a lipid transfer protein might still be possible, and this is something that we would like to pursue in follow-up work. We have not yet performed any additional experiments in this direction. Concerning palmitoylation, the predictions using the CSS-Palm software were always weak and ambiguous, and in addition the best candidate cysteine residue was Cys351, which is in our structure engaged in the disulphide bond observed in the C2 crystal form. We feel that this is something to keep in mind, but is not yet a strong enough hypothesis to pursue intensively.

      Minor Points:

      Figure 1B: The authors should provide information on the void volume of the column.

      Implemented in the figure legend (7.2 ml).

      Page 17, line 696-701: The authors point out that the C2 crystal form is stabilized by two disulfide bridges. The authors should comment on the physiological relevance of these disulfide bridges.

      Given the reducing environment of the cytosol, it is an open question as to whether these disulphide bridges exist in vivo. We would prefer not to speculate on this point, as we do not feel it would be productive.

      Page 18, line 734-740: The authors should provide data on the potential role of Zn2+ on MORN function in a physiological context. The section describing that the dimer is stabilized by Zn2+ ions (pages 18 and 19) lacks a discussion if Zn2+ are functionally relevant. There is only a beautiful sequence analysis and a discussion of the conservation of the Zn2+ coordinating residues. Can the authors perform Zn2+ titrations and SEC-MALS experiments (or alternatives such as SAXS) to show that Zn2+ indeed affects the oligomeric state of only the PfMORN, but not the other MORN proteins that form alternative dimers?

      The known requirement for zinc ions in Plasmodium growth was already noted (OV lines 992- 993, Marvin et al., 2012), and is, we believe, sufficient to address the issue of physiological relevance at this stage. The zinc ions are predicted to affect the architecture of the apicomplexan (Plasmodium, Toxoplasma) MORN1 protein dimers, not their oligomeric state. For PfMORN1, SEC-MALS and SAXS were carried out in 20 mM Tris-HCl pH 7.5, 100 mM NaCl with no zinc present. When EDTA was added, no change in behaviour of the protein was seen by SEC-MALS. When “TPEN”, a strong zinc chelator, was added, the protein precipitated in SEC-MALS experiments.

      Reviewer #1 (Significance):

      A putative role of MORN proteins in membrane and lipid binding is addressed. The view the MORN proteins bind directly to membranes is challenged. Structures of dimeric MORN proteins provide important insight into the modes of dimerization.

      There is a recent structure of MORN proteins (which is referenced by the authors), but I feel that additional structural work is important and justified. The work on membrane vs. lipid binding is important, but not sufficiently addressed in the current manuscript.

      We are glad that the reviewer finds the structural work important and justified, although we disagree with the reviewer’s assessment of the lipid binding. As noted in the previous paragraph, our data challenge the assumption that MORN repeat proteins directly bind membranes, and we feel that this alone is a significant conceptual advance.

      I would recommend to separate the study in two parts. The audience is likely to confused (or bored) by the lengthy discussion on whether or not MORN proteins bind lipids and or membrane or not.

      We would prefer to implement the reviewer's other suggestion, namely that the manuscript is considerably shortened and less focus given to the negative data on lipid binding.

      I am not an expert in structural biology, but have a fair understanding of structural biology. I have worked on lipid binding proteins and have a very good understanding of lipid/membrane-binding assays.


      Reviewer #2 (Evidence, reproducibility and clarity):

      Summary

      The manuscript describes an extensive and detailed investigation into the structure and function(s) of MORN domains. It has to be acknowledged that, despite the considerable amount of work reported, the conclusions are rather limited. From a technical viewpoint, the experiments have been appropriately executed and, generally, I concur with the conclusions drawn. However, the manuscript is over-long: in general, I would recommend concentrating on positive conclusions which can be drawn from the data and avoid excessive speculation or inference (some examples given below).

      We are glad that the reviewer is satisfied with the technical quality of the work and (in general) the validity of the conclusions. We acknowledge that the original submission was fairly long, and have considerably shortened the revised manuscript and focused more on the positive conclusions in order to implement this suggestion.

      Major Comments

      There are three general- perhaps rather obvious- points to make. First, there is no particular reason to think that conservation of structure necessarily indicates conservation of a particular function. There seems to be an implicit assumption that MORN domains are associated with a specific, well-defined biological function. Given their diversity, are there particular reasons to think that this is the case?

      The reviewer is exactly right that there is an implicit assumption that MORN domains are associated with a specific, well-defined function: specifically, lipid binding. It is this assumption, which has been widely circulated in the almost complete absence of experimental evidence, that we are challenging. We agree that MORN repeats are likely to be capable of multiple functions, and protein-protein interactions are now better supported than protein-lipid interactions.

      Second, a strategy which examines the properties of just the recombinant MORN domains in vitro, removed from the context of the whole protein (eg junctophilin) or- importantly- its interacting partners in vivo, has obvious limitations. Frequently a reductionist approach is successful; however, in this case, MORN domains appear to be less tractable to that kind of approach. For all the in vitro binding and structural experiments presented, there is always a concern that the absence of other parts of the relevant MORN-containing protein or its partners could explain failure or inconsistency of in vitro biological activity measurements.

      Again, the reviewer is right that there is an inherent contextual limitation to any in vitro work that utilises a single protein, but this is a concern that - by definition - could be raised about any in vitro study utilising a single protein. It should be noted that we have also carried out in vivo experiments using TbMORN1 (OV Figs. 5, 6 [NV Figs. 3, 4]).

      Third, the possibility that MORN domains might mediate interactions with other proteins seems to be given little consideration, in spite of the Li et al (2019) paper. An experimental strategy which looked for binding partners (eg by pulldown assay) might have provided more insight.

      These data are already in the literature. A previous study by the same team (Morriswood et al., 2013) used proximity-dependent biotin identification to identify candidate binding partners and near neighbours of TbMORN1.

      In order to stress this point we added the following sentence in the discussion section, NV pages 18-19, lines 774-778.

      “The concluding data presented here suggest that TbMORN1 utilises this oligomerisation capacity to build mesh-like assemblies, which can reach considerable size in vitro (Fig. 7G). These mesh-like assemblies may reflect the endogenous organisation of the protein in vivo, where a number of binding partners have already been identified (Morriswood et al., 2013)”.

      Minor Comments

      1. In the abstract and elsewhere the authors refer to a possible function of MORN domains as 'dimerisation and oligomerisation devices' (line 53). What is the evidence that dimer formation is important for function in vivo?

      This is an interesting and important question and one that we would like to address in future work. We did attempt to generate trypanosome cell lines that inducibly expressed monomeric TbMORN1 (the double mutant, where the point mutations were simultaneously introduced in the dimerisation interface in repeats 13 and 14), but no expression of the ectopic protein was ever observed (9 separate clones obtained in 3 independent transfections). This might indicate the importance of the dimeric state in vivo, perhaps hinting that dimerisation is important for protection from degradation. In general, proteins assuming higher oligomeric states in homo- or heteromeric assemblies benefit from increased robustness in the cellular environment and optimised activity by the following means:

      • Increased stability by decreasing the surface area/volume ratio
      • Simple construction of larger complexes
      • Allosteric regulation
      • Co-localisation of distinct biological functions
      • Substrate channelling
      • Protection from aggregation or degradation

      Which or which combination of the factors is relevant for TbMORN1 being a functional dimer in vivo is difficult to say at this point.

      1. Did the authors attempt to co-crystallize TbMORN1(7-15) with PI(4,5)P2?

      No. For crystallisation, we used lysine methylated samples, and by doing this we neutralised positively-charged potential binding sites which would have interacted with the negatively charged lipid headgroup. We did not observe any bound lipids in the electron density maps obtained from the crystals.

      1. Fig 2C: did the authors also estimate binding stoichiometry as well as the equilibrium binding constants for these data? This should be determined by fitting a single binding site model to the data. Other methods (eg ITC) can probably determine this with more accuracy. The value of stoichiometry is sometimes forgotten in such binding measurements- is one ligand bound per monomer or dimer, for example?

      We discussed estimation of the binding stoichiometry in the fluorescence anisotropy assays at some length, but the conclusion was that the required experiments would contain too many approximations to provide high-confidence data. We did use ITC and also MST, but did not observe any binding with these assays.

      1. Lines 674-678 I found it hard to work out whether these constructs harbour the natural C-terminal sequence without truncation or addition of an affinity tag. I think the answer is 'yes' but it was difficult working this out from the details in M&M.

      TbMORN1(7-15) crystallisation was with a C-terminal Strep tag; TgMORN1(7-15) and PfMORN1(7-15) had their affinity tags removed by protease treatment prior to crystallisation. We have clarified this point in the M&M, page 29, lines 1189-1192: “Crystallisation of TbMORN1(7-15) (with a C-terminal Strep tag), TgMORN1(7-15) and PfMORN1(7-15) (both with affinity tags removed) was performed at 22 °C using a sitting-drop vapour diffusion technique and micro-dispensing liquid handling robots (Phoenix RE (Art Robbins Instruments) and Mosquito (TTP labtech).”

      1. Lines 688-694 The PISA interface analysis is useful here in distinguishing crystal contacts from those which persist in solution. The discussion of the results is unclear, however, on this critical point: were the dimer interfaces the only contacts which were significant in the various crystal forms?

      Yes, correct. PISA showed that the described dimerisation contacts were the only significant ones in the various crystal forms. Other crystals contacts had typically low P-values and poor ΔG and small “radar” surface in the complexive PISA analysis.

      In the case of both TbMORN1 crystal forms and in the case of the TgMORN1 P43212 crystal form we have a dimer in the asymmetric unit, while in the case of the PfMORN1 and TgMORN1 P6222 form we have one molecule in the asymmetric unit, and the dimer is created by the crystallographic twofold axis. In the latter cases the quaternary structure resulting from the symmetry operations was the top-scoring one considering either P-values and/or the number of stabilising interactions buried surface area.

      1. Lines 754-763 This paragraph seems rather speculative and is a good example where the text could be cut down.

      If the line citation is correct, then we disagree with this assessment and would prefer not to implement it. The paragraph in question concerns a detailed and very precise discussion of the side chain interactions that stabilise the V-shaped forms of TgMORN1 and PfMORN1.

      1. Line 765-788 This section is also rather overdone: such observations are only useful if they are subsequently tested by recording dimer conformation for a representative selection of MORN dimers from different species.

      Again, we disagree with the reviewer's assessment of this analysis. The analysis has considerable predictive power and already has some experimental validation via the SAXS observation that PfMORN1 is capable of forming extended dimers in solution (OV Fig. 10C [NV Fig. 7C]).

      1. Lines 800-801 I don't think this statement is strictly correct. The SAXS data show that PfMORN1(7-15) adopts an extended conformation, with no evidence of the 'V' shaped structure. Related to that point, from what I could glean from the SAXS Methods section, all solution conditions for these experiments were conducted without Zn2+? If some dimer interfaces require Zn2+, should it not be included?

      We have clarified this statement. The SAXS experiments were conducted without zinc, and, as we have stressed, the V-shaped form of TgMORN1 and PfMORN1 was only ever observed in the crystals. For PfMORN1, SEC-MALS and SAXS were carried out in 20 mM Tris-HCl pH 7.5, 100 mM NaCl with no zinc present. When EDTA was added, no change in behaviour of the protein was seen by SEC-MALS. When “TPEN”, a strong zinc chelator, was added, the protein precipitated in SEC-MALS experiments.

      Reviewer #2 (Significance):

      There is certainly value in establishing that MORN domains do not, in vitro, appear to bind to lipid vesicles, and to define their lipid binding capability (although it is rather complex). The crystal structures and SAXS data extend the rather limited structural data on MORN domains. Despite the effort involved, conclusions about likely functions of MORN domains in vivo are rather limited.

      We are glad that the reviewer acknowledges the value in challenging the assumption that MORN repeats are lipid binding devices, and that the structural data are important for expanding the knowledge base on this class of repeat motif proteins. In vivo functional work is being actively pursued at present.

      My expertise lies in X-ray crystallography and protein biochemistry.

    1. 10). In other words, we, as human beings, learn through and with stories earlyin our lives, and we constantly build ourselves through the various narratives that are present around us. And interestingly, our vicarious experiences of the world can be mediated through stories of all kinds, whether they are real or fict

      On the surface the authors' assertion might appear pedestrian but I think not! Throughout this article they allude to but never succinctly confront that which professional historians may claim as a well-argued factual presentation is when examined as literature looks suspiciously similar to narrative used as propaganda. Event sequences are stories, are they not? Analysis is hopefully well-reasoned opinion. However, in the end "our vicarious experience of the world can be mediated through" a well-composed narrative.

    1. But for learners of a second language, the classroom can be a cause of anxiety, greatly affecting the way they receive and process comprehensible input. By contrast, a house party with lots of international guests is a great place to practise languages, as everybody is relaxed and having a good time. Such an environment offers the language learner plenty of comprehensible input, but (hopefully) none of the anxiety.

      100% yes on this idea, but using the house party may not be the best example of how to create a less-anxiety-filled situation for students, especially social anxiety-prone ones. The affective filter is one of Krashen's ideas that is being borne out in lots of current research, though not described as such - in fact, I just attended a video conference on Mind, Brain, and Language Education, where two of the speakers (one that I saw) presented on this way that stress and emotions actually shut down and impair student learning by reducing cognitive resources like attention, retrieval, and working memory. Fascinating stuff! But one takeaway that resonated with me in a more practical sense is that not only is this internal to students' neurology, it is a shared, collective state - teacher's mindset and other students' mindsets and emotional states also can influence each other. So setting up a situation in class where there is a collective enhancement of the "good" emotions (rather than stress, anxiety, embarrassment, etc) is key to good language teaching. Some ways to do this? I think adapting to students' interests, giving plenty of time for spoken activities, encouraging meaningful interactions is a start. I am eager to learn more!

    2. Adult learners, both inside and outside the classroom, need this silent period, too. Teachers shouldn't be afraid when their students don't participate in debates in class – perhaps they are simply acquiring the language. Moreover, putting pressure on the learner to speak before they are ready will result in anxiety.

      While I generally agree that anxiety can be a huge detriment to the language learning process, I think it is much more nuanced than this. While Krashen's theory is based on the mainstays of L1 acquisition, I think it is important to recognize that there are differences between L1 acquisition and L2 or L3 learning. As we have seen in previous weeks, there is a pretty good amount of evidence to support the Critical Period Hypothesis. Additionally, students learning an L2 or L3 can have very different sources and levels of motivation, not to mention cultural backgrounds, personality types, and other affects. Of course, we should never push our students to the point that they feel anxious in the classroom. However, I do think fostering a comfortable atmosphere to gently challenge the students to try something new and step a bit outside of their comfort zone can be equally rewarding to many. And sometimes, it may be a matter of grading and participation- if we allow students to simply not participate until they are "ready", some will be chatting right from the beginning while others may take weeks. I suppose the biggest takeaway is to create an atmosphere to reduce the anxiety and build each student's knowledge of the language to help them feel more prepared to participate!

    3. Learners need to be exposed to what Krashen calls 'comprehensible input' – that is, exposure to interesting and understandable listening and reading material.

      Of course, this is just Krashen's hypothesis, but I could see a few points where this gets tricky when translating it to real-life classrooms. I think back to the beginner level classes I've taught- what input exactly is "comprehensible" to them? Especially because a lot of times, the lowest level classes may just be a catch-all for the students that couldn't score high enough into the next levels. From my experience, their individual understandings varied quite greatly. It's not always the simplest task to find material that is both interesting and understandable to each student. This is even truer as the students get older. If you are teaching adults at a very basic level of English who don't have much prior exposure to it, what they can understand will be quite different from what is interesting to them. There will always be a few categories that widely engage students - sports, music, movies, travel, food, etc. I suppose it is important to find many sources of materials to try and balance the interesting and understandable aspects of them.

    4. the language acquisition itself is not their objective. Rather, it is a by-product of the achievement of some other purpose,

      This part reminds me of some of our discussions regarding intrinsic and extrinsic motivation. I think language acquisition is probably most successful if students see a larger purpose to what they are doing and are motivated to engage in the learning as a result. For example, they need the language in the near future to enroll in college or succeed in a career. Or, on a more immediate level, they need to know how to order something at a restaurant or how to ask where the bathroom is. In communities where you can get by with your native language and never learn English to go about your daily life, people may not speak English well because they don't have to. When you are immersed in a community where you must communicate in the target language to get what you want, acquisition is probably more likely.

    1. As such, the microbiota profile may be a good representation of the environmental history of the individual

      That is interesting. Before this research, we always know that genetic,age, sex and diet are representation of individual profile. Now we also know that the microbiota profile is a good representation of history of the individual. I think it is a breakthrough in health care field contributing an important role to treat some certain diseases of human being.

    1. I think sometimes teachers feel like teaching such a diverse population is not what they signed up for. When I look at the demographics, I try to explain to people what the future will be.

      This is a powerful section of the article for me because even if you knew you would be (and wanted to be) working with a diverse population, there are still so many changes as your career progresses that you could not have anticipated. The source of your school's diversity may change over time. For example, we used to have a robust Bosnian bilingual program that no longer exists. Now, our Arabic speaking population is growing, as is our contingent of Somali refugees. ESL (and general education) methods have changed and are more focused on oral communication and collaborative group work. Desks being in rows used to be the norm, but now that room organization is less popular. Cell phones were virtually non-existent when I started teaching, and now technology in education is both a boon and a bane that teachers have to navigate. I don't think I ever could have anticipated teaching remotely for months through a pandemic, struggling to meet the needs of my EL and DL students through a computer screen. In the midst of all this change, teachers still need to provide all their students (and ESL newcomers in particular) with stability, access to what they need to thrive, and preparation for what the future may bring in an ever-changing country and world. That's a tall order.

    2. Being bilingual is not the same as being biliterate.

      I really like this quote. I think it's important for teachers especially to keep this in mind in the classroom. We so often have so much material to cover and we just run through it expecting our ELL students to understand as much as the English speakers simply because they can communicate with us. I think it's important to not only be sure the material in English is at the level of those ELL students but also to be sure we are checking in with them on a regular basis. It may be embarrassing or difficult for them to say in class or even privately to us that they are not understanding the content, so it's incumbent upon us to check-in with them in a non-threatening way.

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      Reply to the reviewers

      Response to reviewer comment for manuscript RC-2020-00207

      Reviewer #1 (Evidence, reproducibility and clarity (Required)):

      **Major Comments:**

      The authors of the paper start the paper with just one protein narrowed down ie. HRG. The rest of the paper uses affinity based proteomics, antibody validation, GWAS and survival analysis to validate this target and support their claim that HRG is an age associate protein linked to mortality and certain clinical outcomes. How did the authors conclude that HRG was the only target to explore further in this paper? What methods or analysis was done for this? What were the other proteins if any that showed up in these studies?

      We appreciate this comment which reveals unclear explanation how the protein was chosen for further analysis. The protein profile obtained using HPA045005 was the top and single hit out of 7258 protein profiles using a threshold of adjusted P-value below 0.01. In other words, only the profile of HRG was statistically significantly associated with age in the screening sample set (N = 156). The results of all protein profiles were attached as Supporting Table 1. Phrases about the alpha level were added to the text to make the threshold clear. Because antibody validation of these exploratory studies requires enormous efforts and time, we could not choose a more liberal and inclusive threshold.

      For mortality outcome, it is not clear which class of disease is most strongly associated with increased risk of mortality from elevated HRG levels. If cause-specific mortality exists among the cohorts, could authors provide a more exact breakdown of the type of associated mortality by a disease class?

      We thank the reviewer for the question and have now added cause-specific data in the manuscript. Using cause of death data, mortality risk by diseases in circulatory system were compared with the risk by neoplasm and others. ElevatedHPA045005-HRG profiles were found to associate with mortality risk by diseases of the circulatory system (HR = 1.46 per SD, P = 2.80 × 10‑4, ICD-10 code I00-I99). It was larger than the risk by malignant neoplasms (HR = 1.28 per SD, P = 1.73 × 10‑2, ICD-10 code C00-C97). We chose big categories as ICD-10 codes "I" and "C" because the number of events was too small to get enough power in the survival analysis.

      Page 4 Section 3 (Results)-

      The authors say "We found consistent age-associated trends with HPA045005 across all eight replication sets (Supporting Figure 3)". On examining the supporting figure we noticed that the slope for the set with the largest number of subjects (Set 3 with ~3000 people) is visually negligibly positive (showing weakest age associated trends with HPA045005). Some comments from the authors on why they think the largest data set showed the weakest association.

      The plot for each cohort (in Supporting Figure 3) had different ranges in the y-axes. To make those plots comparable, the ranges in the y-axes of the different panels in the figure were modified to be the same for all cohorts. In the new version of the plot, it is easier to notice that there in fact is an increasing trend of the profiles in set 3. As we briefly discussed in Discussion, weaker age-association of the sample set may be due to the set was near to a random sample of population in the age range. Set 1, however, had over-representation of older people by selecting equal number of people in every age-intervals.

      From Figure 2 C in the main manuscript one concludes that for HPA045005, binding for CC individuals is ~ 2 times higher than TT individuals. Is it possible the age association showing up for HPA045005 is primarily a function of changing/increase in allele frequency as a function of age?

      The authors could consider adding a clarifying plot of Age vs Allele frequency or adding an interaction term of Age and Allele Frequency in the regression and survival analysis to address this question.

      As suggested, we now added a test of age association, and average age was compared by genotype. The result was added in Supporting Table 3. The heterozygote (CT) group has slightly higher average age without statistical significance (ANOVA P = 0.096).

      It is interesting that the signals were significant with the HPA045005 antibody but not with the BSI037 antibody. This is in spite of the fact that the GWAS for BSI0137 signals had an even stronger hit to the same locus. Can the authors please comment on why the signals from HPA045005 and BSI0137 were not highly correlated with one another and why the better antibody could not replicate the survival analysis results?

      We thank the reviewer for the comments. We believe that our text about our findings were not clear enough, though it is a primary finding. We modified the main text to easily distinguish the HPA045005-derived profiles that were influenced by the 204th amino-acid of HRG protein, from the BSI0137-derived profiles influenced by the 493the amino-acid. The signals from those two antibodies were likely obtained by capturing different parts of HRG, which are schematically illustrated in Figure 2D. What we found is that only one binder's profiles, not the other's, had predictive power for mortality risk within about 8.5 years. That suggests some age-dependent changes around the 204th residue of HRG reflected biological aging rather than whole protein level. To make our finding clearer, the two binders were compared in Table 2.

      **Minor Comments:**

      Figure 1: The authors description of the figure could use more clarification. "For each sample set, the estimated effect from the linear regression model.." estimated effect of what on what? On reading the main text one concludes it is the effect of age on HPA045005. This needs to be clarified in the label.

      We agree with the reviewer and have added these words.

      Figure 3: The X axis for the Kaplan Meir survival curve is labelled as Age. Survival is usually time to event and time is usually the follow up time. Further clarification for the choice of this label might be helpful.

      We clarified the choice of the time scale in the figure legend with a reference, where it was further discussed (Thiébaut & Bénichou, 2004). We chose age as the time scale, seeing age is the strongest risk factor for all-cause mortality, as the suggestion in the reference. We attempted to use follow-up time as the time scale with age adjustment before, which gave us almost the same results but violated the proportionality assumption of COX models.

      Figure 3: it would be good to include a table with the number of individuals at risk at the bottom of the plot at defined time intervals. The figure currently compares the bottom and top quartiles of HRP for visual assessment of mortality risk, it would also be informative to include middle quantiles.

      The figure was updated accordingly. The risk table was included and the results of the middle group were presented.

      Supporting Table 5: The note at the bottom of this table states "standardized HRG values by linear regression and scaling." What does standardization by linear regression mean?

      A sentence that explains the standardization was added in the footnote of the table.

      Supporting Table 5: It would be useful to understand that HRG carries additional risk beyond known Age and known clinical biomarkers listed in Table 2 (APOA1, APOB, TC, TG, Glucose, LDL). Could authors include a multivariate CoxPH regression with just Age? and with Age + clinical covariates?

      The impact of those clinical variables on survival models was examined and the results were added to Supporting Table 6 (which was Table S5). It turned out that the addition of those variables barely changed the results of the model for the HRG profile affected by 202th amino-acid.

      Reviewer #2 (Evidence, reproducibility and clarity (Required)):

      **Summary**

      The manuscript by Hong et al. describes the identification and validation of histidine-rich glycoprotein (HRG) as a marker of chronological age and all-cause mortality. HRG was determined using proteomics of serum and plasma samples in 9 different cohorts (total sample size ~4,100). The association with mortality was tested in the largest available cohort (TwinGene), comprising ~3,000 samples. The association with mortality seems to be stronger in women in comparison to men and could not be explained by CRP or diabetes-related traits. The HRG levels determined using an alternative antibody, BSI0137, did not show any association with mortality, indicating that the effect on mortality is likely isoform-dependent. The performed analyses seem to be statistically solid. However, the association with mortality still needs to be replicated in independent studies and the HRG measurement does not yet seem to be ready for standardized high-throughput measurement, which is necessary to make it usable as biomarker.

      **Major comments**

      • Although the authors have convincingly identified HRG to be associated with chronological age and mortality, it will require quite some additional work (including replication of the observed association with mortality in independent cohorts, testing the predictive ability, and making the measurement standardized and high-throughput) to prove its use as potential biomarker. At the moment, this is not at all discussed in the manuscript. Moreover, there have been some recent large-scale studies that identified biomarkers at the metabolic level that are not at all mentioned by the authors. The authors only refer once to the recent proteomic study by Lehallier in the Introduction, but do not at all discuss their findings in relation to this paper. Last but not least, HRG has already been associated with mortality in a previous study (https://www.ncbi.nlm.nih.gov/pubmed/29303798), but there is no mention of this anywhere in the manuscript. Hence, I think it would be good if the authors perform a thorough literature search to place their findings into context and rewrite their Discussion accordingly.

      We appreciate the reviewer's comments on the limitation of our paper. We are aware of the requirement of further investigation on HPA045005-HRG profiles as a biomarker to confirm it with independent cohorts. Instead, we supported our findings with a set of confirmatory analyses; we validated and annotated age-associated profile applying GWAS, sandwich assays, peptide arrays and mass spectrometry. Comparing two antibody profiles, we narrowed down to age-associated region within the protein HRG. The approach and finding, we believe, is novel.

      We added some discussion about recent large-scale proteomic studies such as Tanaka et al, 2018 and Lehallier et al, 2019. Unexpectedly, HRG was found not measured in those studies despite of the protein is one of the abundant proteins in blood (Poon et al, 2011). It may reflect challenges in assay development and missing piece in those large studies. The papers lack further investigation for molecular targets, which is common in proteomic papers, and makes it difficult to compare between studies and technologies. In that sense, our approach is different from other proteomic studies, because we invested time and efforts to investigate the molecular target.

      We are though thankful for the introduction of the suggested HRG publication, which we did not know about. We concluded that there are substantial differences in the subjects and suggested functions for the protein. Kuroda et al. found HRG as a biomarker for sepsis of ICU patients, while our study was done on the general population. They were measuring HRG protein level, whereas we found one particular region in HRG as a biomarker for all-cause mortality. Hence, we briefly discussed the reference in the paragraph about general information about HRG.

      • The authors need to add a Supplementary Table showing the association of all their 7,258 HPA antibodies with chronological age. Although I trust the authors, I can currently not tell if it is indeed correct that only one antibody was significantly associated with age in set 1.

      We agree with the reviewer. The table of association test results of all 7258 antibody profiles was attached to the paper as Supporting Table 1. We were also surprised that only one passed a conventional P-value threshold 0.01 after Bonferroni correction. It might be due to the low number of samples in the sample set 1 (N=156), compared to the number of antibodies or tests.

      • According to description in the Supporting Information, several samples in set 3-5 were overlapping with set 1 (45 in total). These samples should be removed from datasets 3-5 to make sure that there are no overlapping samples in the meta-analysis. However, I am not sure if the authors have actually done this. For the GWAS the overlapping samples from set 3 could still be included, given that set 1 is not involved in that. The authors could actually use these 45 overlapping samples to provide additional details about the reproducibility of HPA045005 between different measurements, for example by showing a correlation plot.

      We agree with the reviewer. Those 45 overlapping samples were excluded in the meta-analysis. As the reviewer's comment, only the data of sample set 3 was used for the GWAS.

      We also appreciate the comment regarding reproducibility and acknowledge that there are limitations to the technical performance of our exploratory SBA method. The procedure is tailored to handle large number of antibodies and profile 384 sample in the analysis plates. This setup allowed us to process relatively large number of samples per batch but it might be affected by batch effects. In our study set 3, there were 2999 samples randomized and analyzed in 8 different 384-well plates. The 44 overlapping samples between sets 1 and 3 were added to one of these 8 plates. This resulted in 1-11 samples to be analyzed on the same plate, hence, comparing these 44 with previous assays might be influenced if not dominated by plate effects. We went back to the initial data set generated during 2011/2012 and compared the first data with replicated assays using the same freeze-thawed samples. For HPA045005 we found the data to correlate by r=0.45. The next analyses of these 44 samples were conducted during 2015 using different sample aliquots and preparations as well as different SBAs. The correlation to previous assays was r

      • When looking at the effect of the rs9898-stratified analysis (Table S2) it seems that there only is an effect in the presence of the C-allele. Have the authors considered the presence of a potential recessive effect of this variant when looking at mortality?

      Average age of the individuals of each genotype of the SNP was compared and added into Supporting Table 3 (which was Table S2). No significant difference between the genotypes was found. As the reviewer noted, the mortality association of the HRG profiles affected by 204th amino-acid in the TT genotype group of rs9898 was milder and did not reach statistical significance. We believe that it is due to substantially smaller sample size and number of deaths in the genetic group. To clarify the difference in numbers, those numbers were added into the Supporting Table 3 (which was Table S2).

      • The authors need to discuss in more detail the implications of the difference between the two HRG antibodies in their association with mortality, for example in light of the use of HRG levels as a potential biomarker (i.e. how should one deal with the fact the way the levels are measured influences the outcome).

      We appreciated this valuable comment, which clearly reveals that our claim was not explained sufficiently. We modified the main text to distinguish those two antibody profiles more clearly. We also added Figure 2D and changed the structure of Table 2 to highlight the difference between the two antibody profiles.

      • Why did the authors put part of their Discussion in the Supplement? This is not common practice. They should either move it to the manuscript or remove it completely.

      We moved the discussion in the supplement to main text as the reviewer's suggestion.

      Reviewer #2 (Significance (Required)):

      The manuscript is clearly written and the analyses seem to be solid. However, although the findings described in the manuscript are interesting for the ageing field, they only provide a small step in the process of the usability of HRG as biomarker, i.e. many validation and follow-up studies will be necessary to prove its value. There have been some recent biomarker studies that have been much more advanced in this respect, which limits the novelty of this manuscript. I therefore feel that this manuscript may be best suitable for a medium-impact ageing-specific journal. My fields of expertise are ageing, genetics, and molecular epidemiology. Given my limited expertise when it comes to proteomics, I was not able to provide detailed comments on the methodology concerning this part.

      We thank the reviewer for the honest and constructive assessment of our work and agree with the suggestion to transfer this work to a medium-impact journal covering aspects of ageing research.

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      Referee #1

      Evidence, reproducibility and clarity

      Summary:

      The paper applied affinity based proteomics and antibody validation to choose and validate histidine-rich glycoprotein (HRG) as a protein/target of interest. Survival analysis techniques were used to show associations between this protein and certain biomarkers, age and all cause mortality.<br> These results and findings were used to conclude that HRG may serve as a molecular indicator of age and mortality risk.

      Major Comments:

      The authors of the paper start the paper with just one protein narrowed down ie. HRG. The rest of the paper uses affinity based proteomics, antibody validation, GWAS and survival analysis to validate this target and support their claim that HRG is an age associate protein linked to mortality and certain clinical outcomes. How did the authors conclude that HRG was the only target to explore further in this paper? What methods or analysis was done for this? What were the other proteins if any that showed up in these studies?

      For mortality outcome, it is not clear which class of disease is most strongly associated with increased risk of mortality from elevated HRG levels. If cause-specific mortality exists among the cohorts, could authors provide a more exact breakdown of the type of associated mortality by a disease class?

      Page 4 Section 3 (Results)-

      The authors say "We found consistent age-associated trends with HPA045005 across all eight replication sets (Supporting Figure 3)". On examining the supporting figure we noticed that the slope for the set with the largest number of subjects (Set 3 with ~3000 people) is visually negligibly positive (showing weakest age associated trends with HPA045005). Some comments from the authors on why they think the largest data set showed the weakest association.

      From Figure 2 C in the main manuscript one concludes that for HPA045005, binding for CC individuals is ~ 2 times higher than TT individuals. Is it possible the age association showing up for HPA045005 is primarily a function of changing/increase in allele frequency as a function of age? The authors could consider adding a clarifying plot of Age vs Allele frequency or adding an interaction term of Age and Allele Frequency in the regression and survival analysis to address this question.

      It is interesting that the signals were significant with the HPA045005 antibody but not with the BSI037 antibody. This is in spite of the fact that the GWAS for BSI0137 signals had an even stronger hit to the same locus. Can the authors please comment on why the signals from HPA045005 and BSI0137 were not highly correlated with one another and why the better antibody could not replicate the survival analysis results?

      Minor Comments:

      Figure 1: The authors description of the figure could use more clarification. "For each sample set, the estimated effect from the linear regression model.." estimated effect of what on what? On reading the main text one concludes it is the effect of age on HPA045005. This needs to be clarified in the label.

      Figure 3: The X axis for the Kaplan Meir survival curve is labelled as Age. Survival is usually time to event and time is usually the follow up time. Further clarification for the choice of this label might be helpful.

      Figure 3: it would be good to include a table with the number of individuals at risk at the bottom of the plot at defined time intervals. The figure currently compares the bottom and top quartiles of HRP for visual assessment of mortality risk, it would also be informative to include middle quantiles.

      Supporting Table 5: The note at the bottom of this table states "standardized HRG values by linear regression and scaling." What does standardization by linear regression mean?

      Supporting Table 5: It would be useful to understand that HRG carries additional risk beyond known Age and known clinical biomarkers listed in Table 2 (APOA1, APOB, TC, TG, Glucose, LDL). Could authors include a multivariate CoxPH regression with just Age? and with Age + clinical covariates?

      Significance

      The authors have identified a new biomarker for aging and mortality. Understanding the mechanism and pathways involved in HRG homeostasis and how aging causes dysregulation of this HRG could be a topic for further research. Overall, this pathway provides an opportunity of a new molecular target for aging-based drugs and research.

      This article should be of interest to researchers interested in the biology of aging and for researchers developing drugs to slow down the process of aging. In addition, it should be of interest to researchers studying the HRG as a biomarker (for example, in sepsis (https://ccforum.biomedcentral.com/articles/10.1186/s13054-018-2127-5, https://papers.ssrn.com/sol3/papers.cfm?abstract_id=3437790).

      This paper was reviewed by 3 co-reviewers, a senior principal investigator with extensive bioinformatics, metabolomics/proteomics, epidemiological experience, a highly experienced computational biologist with a record of developing and applying methods in bioinformatics and computational biophysics and lastly an computational biologist with a background in applied mathematics and statistical analysis. All three scientists are interested in aging research and understanding how human physiology and biomarkers in specific, change as a function of age.

    1. 303

      M - you will see numbers like this throughout the text. I preserved the page numbers as they appear in the PDF that I downloaded. We may decide these are helpful or not. I do think it is good to have them so that readers may refer to them.

  7. clas3209.wordpress.com clas3209.wordpress.com
    1. into their clothing

      They did not only sew the skin of their enemies on the cloths (specifically cloaks). Onyshkevych clearly indicates that they were stitched onto horse bridles and quivers.

      I also think that what we wrote for the db titled "Do you Believe?" can also be written about here. Some of us wrote about how the quotes about the Scythians drinking blood and using their enemies' flesh and skulls could not be proven with primary evidence. So, the readers should be made aware of the fact that the Scythians may not have been as barbaric as the Greek and Roman writers described. After all, they weren't big on writing about their own history and culture, so there is no way to prove these facts.

    1. Human Resource is a part that has changed step by step. Everything from an individual’s examination, the recruiting procedure to programming frameworks, has developed. Since HR patterns continue transforming, it is fundamental for you to keep awake to-date on the present patterns. Throughout the previous 10 years, the consideration of HR has been on worker fulfillment and representative commitment. Today, there are a few instruments just as must-have assets that help you to fabricate an illuminate HR culture. The following are seven developing HR trends. Audit Culture & Ceaseless Input One of the most anticipated HR patterns is the way of life of audits and ceaseless input in the working environment. The techniques that individuals use to give criticism have changed throughout the years. As an HR chief or an administrator, you should concentrate on improving your specializations by giving continuous and productive input. As indicated by a few examinations, most workers would incline toward constant audits or input rather than the yearly execution surveys. This is on the grounds that the training encourages them to know where they may be turning out badly, and offer them an opportunity to address themselves before the yearly assessment. They don’t need to trust that a year will realize where they turned out badly. Ceaseless criticism will likewise enable the association to develop and stay away from huge losses. Review Culture & Continuous Feedback We have come to discover that activity fulfillment and execution go together. Both appear to create whenever representatives have a chance to investigate their inventiveness. Most driving endeavors apply this information to offer the development of their workers. By creating frameworks that gauge and furthermore oversee execution, you can have the option to accelerate development and notice the noteworthy change. It is indispensable to elevate the solid execution of the executive hiring to both individual-level and hierarchical level on the off chance that you need your organization to succeed. Digitized Prizes & Acknowledgment Probably the best spark for most workers has been getting applause and acknowledgment from their chiefs. Each individual anticipates that the pattern should progress. We as a whole plan to see digitized prizes and acknowledgment. Consequently, to make your representatives beneficial, advanced acknowledgment is a pattern you have to execute. Another HR pattern expected is shared acknowledgment. Start by utilizing the social stages and offer a reasonable stage for your representatives to perceive and remunerate their commitment. HR Bots (AI Driven HR) Another intriguing HR pattern to the center is AI-driven HR. Despite the fact that it isn’t required to dispense with the standard human HR, it will change and help to investigate the information. It will likewise aid the essential redundant HR assignments. At the point when you utilize Artificial knowledge in selecting the workforce, you will have the option to kill inclination. It will assist with getting to the competitors dependent on their requirements without partiality. Learning the Executives Frameworks The requirement for aptitudes advancement is significant for every one of your representatives regardless of which phase of the profession they are right now. Steady learning is required to be among the most well known and developing HR inclines. It is basic for any association that needs to develop to give learning chances to all representatives. Through learning, your representatives can have the option to improve their abilities and have the option to give the best while working. Your association can have to learn the board frameworks that will assist you with checking and track your workers learning process precisely, and furthermore help to advance cooperation between different offices in your organization. Increment in Low Maintenance Business & Unexpected Workforce The Board In most progressive nations, there is a standard pattern where in excess of 40 percent of representatives are being employed on an unforeseen premise. This implies you have to become familiar with your workforce structure and whether you can reevaluate organizing it. You have to learn and comprehend the sort of frameworks and advances that can be actualized in your association. Despite the fact that it isn’t down to earth for any association to receive another workforce structure in a flash, it is fundamental to know about the rising HR patterns. Remember that your workforce is the most basic resource in your association. Utilization of Online Expertise Evaluations Online evaluations have likewise gotten progressively famous as a successful ability to the executive’s device. Representative evaluation is not, at this point pretty much agreeing to an association’s rules. It is presently an indispensable piece of the executive system. In the present profoundly serious worldwide economy, an association must have a grasp of what precisely the workforce knows, or doesn’t have the foggiest idea. Online appraisals including tests, overviews, tests, and tests have been utilized to alleviate the dangers of depending on negligible self-evaluation. Wellness & Health Applications to Create Representative Commitment Another essential HR functions pattern that most associations are required to concentrate on is wellness and wellbeing applications. Most organizations nowadays are concentrating on making life and work balance. In the event that you need your representatives to be beneficial, and to have the option to assemble a practical workforce, you have to adjust work and life. To accomplish this target, you can present health and wellness application for your workers. The applications will assist with making harmony between their expertise and their own lives. For instance, your representatives can approach dietary directing, yoga, and work and life advising among numerous others. From People Analytics to Analytics For The Individuals An absence of trust can impact numerous workforce investigation endeavors. On the off chance that the emphasis is fundamentally on productivity and control, workers will question if there are any advantages for them. In general there is a move to more worker driven associations, albeit now and again you can question how real the endeavors are to improve the representative experience. Posing the inquiry: “By what method will the representatives profit by this exertion?” is a decent beginning stage for a great many people examination ventures. It additionally assists with making purchase in, which turns out to be progressively significant. Simply estimating the “disposition” of workers, and other key individuals markers (efficiency, residency) doesn’t really carry advantages to representatives. It may really reverse discharge: workers feel that they are controlled, and their voice isn’t heard. Learning In the Progression of Work It has any kind of effect if a representative must look effectively for a learning module that the individual needs, or if that the small scale learning module is offered at a suitable second in the work process, in light of on-going perceptions of the conduct the worker. In the event that there is a gathering with organization X in your journal, your own learning help may ask: “Would you like to study organization X?”. In the event that you are stuck in structuring a troublesome Excel large scale, the Excel chatbot asks you: “Would I be able to assist you with designing the full scale?”. On the off chance that you have a gathering planned with a representative with a low presentation rating (the PC gets this data in the HRIS), you are offered a short module “how to manage to fail to meet expectations workers”. During your online deals call, you get recommendations in your screen on the most proficient method to improve the discussion (“Ask a few inquiries”, “Attempt to close”), and thereafter your discussion is contrasted and top tier models, bringing about some learning focuses. Comprehensive authority The desires workers and different partners have of authority, are frequently excessively high. Frequently you hear: “Change needs to begin at the top”, and “Pioneers need to show others how its done”. These kinds of articulations can be deadening. On the off chance that workers are sitting tight for directions from the top and get debilitated if their pioneers are not immaculate people, associations will be in an awful shape. Changing initiative into progressively comprehensive administration can be gainful to associations. Comprehensive initiative has been centredaround the attributes of the comprehensive head. It is additionally about the attributes of the association and the way to deal with initiative turn of events. I despite everything see numerous administration improvement educational plans that are develop customarily: a restrictive program for the best, a program for centre administrators and the leader program for high possibilities. Set-ups like this don’t strengthen comprehensive authority. Time for HR to start new methodologies. Efficiency In the most recent years, there has not been a great deal of spotlight on efficiency. We see a moderate change at the skyline. Generally, limit issues have been fathomed by enlisting new individuals. This has prompted a few issues. I have seen this multiple times in quickly developing scale-ups. As the development is constrained by the capacity the find new individuals, the choice rules are (regularly unknowingly) brought down, the same number of individuals are required quick. These new individuals are not as gainful as the current group. Since you have more individuals, you need more directors. Lower quality individuals and more administrators brings down profitability. Another methodology is, to concentrate more on expanding the profitability of the current representatives, rather than recruiting extra staff, and on improving the choice measures. Utilizing individuals examination, you can attempt to discover the qualities of top performing individuals and groups, and the conditions that encourage top execution. These discoveries can be utilized to build efficiency and to choose competitors that have the attributes of top entertainers. At the point when profitability expands, you need less individuals to convey similar outcomes. Corporate & Representative Activism Numerous associations are still deep down centered. The key inquiry is more “How might we take care of our issues?” than “how might we take care of issues in our general public?”. Taking responsibility for corporate social duty can be more than offering representatives the chance to do great on one day out of every year. Research by Povaddo indicated that the greater part of those working in America’s biggest organizations feel that corporate America needs to assume a progressively dynamic job in tending to significant cultural issues. There are sufficient issues to handle. Representatives are eager to contribute. HR can assume a significant job in encouraging and invigorating corporate/representative activism. Main concern While no individual can determine what HR patterns 2018 will offer, specialists and various examinations focuses towards representative fulfillment and advanced devices. In any case, one thing to be certain is that innovation will affect the HR office, and how they work. To remain on top of things, you should begin setting up your association at the earliest opportunity. Be that as it may, however the above HR patterns guarantee astounding occasions ahead, you ought to be mindful.You ought to think about computerized instruments as a help for your association procedures, and not for them to drive your association completely. It is urgent to be open and adaptable before grasping new innovations to help your HR office. Likewise, don’t dismiss the master plan in view of the accessibility of apparatuses and rising patterns. You have to learn, comprehend, break down and counsel generally before responding to any of the rising patterns. For instance, AI is a phenomenal device that is the eventual fate of HR and different offices. Notwithstanding, you have to become familiar with the advantages and the issues that can’t be unravelled by the Bots. Today, the HR office is not, at this point a help work. Henceforth, it is important to have appropriate framework this basic office

      HR Practices are very essential for your business to organize your business. In this article, we bring you top 10 Trending HR Practices In Organization.

    1. Why would people share news they think might not be accurate? We identify a factor that, alongside accuracy, drives the sharing of true and fake news: the ‘interestingness-if-true’ of a piece of news. In two pre-registered experiments (N = 604), participants were presented with a series of true and fake news, and asked to rate the accuracy of the news, how interesting the news would be if it were true, and how likely they would be to share it. Both interestingness-if-true and accuracy played an important role in explaining the sharing of true and fake news, with participants more willing to share news they thought interesting-if-true, and accurate. Participants also found fake news less accurate but more in-teresting-if-true than true news, and were more likely to share true news than fake news. Higher trust in mass media was associated with a greater ability to discern between true and fake news, and partic-ipants rated as more accurate news that they had already been exposed to (especially among true news). These results suggest that people may not share news of questionable accuracy by mistake, but instead because the news has qualities that make up for its potential inaccuracy, such as being interesting-if-true.
  8. learn-us-east-1-prod-fleet01-xythos.s3.amazonaws.com learn-us-east-1-prod-fleet01-xythos.s3.amazonaws.com
    1. In ~l~bber, when Jill is simply following the crowd and is bullying Linda because It IS what everyone else is dOing, she occasionalIy ponders her actions, but continues to follow Wendy, the popular ringleader.

      This is a common thing that occurs especially amongst newly socialized children. They tent to follow the group regardless of wright or wrong and must deal with conflicting thoughts and emotions about this practice. I think bringing this to light as many children's literature authors have done gives an important lesson that many may be uncomfortable with but still learn regardless. This shows the duality in the lessons we must teach our children. Not only must we teach them the good things, but we also much teach them the bad as well.

    1. We may have good intentions with our students' best interests at heart, but that does not change the fact that we are using grades as a form of control.

      Such good intentions are themselves a violation of personal integrity. Teachers are forever deciding what 'students need' - often times without any clear evidence. I once asked a math teacher why students needed to know quadratic equations and all reasoning was circular. Asked what he used this for, he could not think of any time in his life when he actually needed this.

  9. May 2020
    1. Reasons You Might Want to change your job! Leave a Comment / Blog Contact If you’re planning on leaving or changing your current job, this blog is going to help you in a certain way. Given what’s going on right now, some of you might be saying that the last thing you’re thinking about is changing jobs. I totally understand. But there are companies hiring today. And they’re looking for the best talent. So, deciding whether to change or leave your job remains a very personal decision. There are three things that I would suggest to someone who’s trying to make the decision about changing jobs. I can’t answer these questions, but I do think the answers will help someone figure it out for themselves. Your job is impacting your health. First and foremost, if your job is making you physically or emotionally sick, you need to step back and think. There are jobs where risks do exist, and individuals take those roles knowing that. Jobs in health care, construction, etc. come to mind. Individuals in these industries are taking as many preventive measures as they can. Your work doesn’t make you happy anymore. This could be one of two things: 1) You love what you do but you don’t love the company (or your boss) anymore. OR 2) You’ve fallen out of love with the work. Maybe you used to love traveling as part of your job and now, not so much. It’s important to understand which one you’re dealing with. (NOTE: It’s also possible that the answer is both #1 and #2.) Your career doesn’t make financial sense. I don’t want to simply say that the job doesn’t pay enough. Because maybe the pay is fine. It’s possible that the benefits package doesn’t suit your current situation. Or the cost of maintaining your professional license is getting expensive and the company isn’t reimbursing. The question is “Does your current position adequately cover your living situation?” Once you honestly and seriously answer the above questions, it might help you decide if you want to make a change AND more importantly, what you might want to make a change to. There is some truth to the saying that the best time to look for a new job is when you have a job. I realize not everyone gets that opportunity which is why it can make some sense to always be thinking about your job wants and needs. If you’re thinking about a new opportunity, I want to give you something else to consider. Now is the time to start planning. Don’t wait until you have to make a move to start planning for it. Here are three action steps that will help you find your next job. Also, there are some other important questions that you might want to ask yourself before taking the big step : What specifically about my current situation is frustrating to me? Pinpointing the issue is the first step towards solving it. Kimberly Bishop, recruiter and chief of her eponymous career management firm based in New York, advises employees to identify how their job is failing them. Is the problem the people, the environment or the work itself? After you’ve defined the frustration, consider the scope. If you decide you’re creatively stifled, for example, you may not need to quit to fill the void. Seek an outlet outside of work or raise your hand for another department or project. Have I taken every action possible to make my current job workable? If you realize your situation is not abusive and could be manageable, consider the steps you might take to improve it. Try taking a positive attitude, altering your time management or work habits, and communicating more clearly with your manager. Perhaps a schedule change or clearing an item off your workload will make a big difference. Ultimately, what do I want for my job, career, and life? “A big mistake: When people decide to quit they think they’ll just update their resume and start networking,” says Bishop, who advises being more thoughtful about what you really want and how you’ll get there. Define your priorities. Going to law school may be intellectually stimulating but will not help you achieve the flexible schedule you’ve been craving. Similarly, if you’d like to make a career change, think about all the necessary steps. They may include more school, a pay cut, or working your way up from the bottom–again. Once you know exactly what you want, you may want to ask: How much do I want it? Have I saved enough to cover nine to 12 months of expenses? Susan Hirshman, financial planner and author of Does This Make My Assets Look Fat?, says a few years ago she told people to save enough for six months of expenses. Now she tells people they need nine to 12 months. “If you’re quitting, you won’t get unemployment,” she cautions. Hirshman suggests mapping out fixed expenses like mortgage, credit card, and loan payments, transportation, and food, as well as factoring in the “what if” costs. You may need a little extra to cushion against the unexpected, like car or appliance repairs. How might I cut expenses or earn income while between jobs? After completing a detailed budget, you may realize you’re coming up short and need to create some cash. Often, income is easily supplanted with a part-time service job. However, Hirshman warns that even waiter jobs are difficult to come by in the current economy. You only have two options: Cut expenses or bring in more money. Figure out what will work for you and be honest with yourself, Hirshman says. Have I timed this appropriately? Agryie suggests that employees who’ve decided to quit consider their timing. Firstly, are you in the midst of the busiest season or working on a big project? You may want to honor your commitments so that your team isn’t left in a bind and you’re able to leave on good terms. Secondly, “maximize the money,” he says. If you’d like to get your quarterly bonus or the holiday vacation, it might be smart to wait a few months. So after answering all these questions for yourself, you’d be able to decide if you’d want to continue with your old job or career or switch to a new one.Put together a job search plan. Grab a notebook and start plotting your strategy. Think about your skills. Make note of the knowledge and skills you want to work on before starting to interview. List your must-haves and nice-to-haves for your next company and job. Start thinking about your professional network, both online and the one on one type. Identify the resources you need. It’s possible that you would benefit from taking a class, joining a professional group, or reading some books. Make a list of everything you need and roughly how much it will cost. Start budgeting for these items. Also, think about if you will be out of work for a while and if you will need to cover health insurance in-between jobs. That needs to be budgeted as well. Ask for support. Once you have a plan, reach out to your network. Start reconnecting with them. If you’ve been doing that all along – fantastic! If you haven’t, it will take some time before you can ask for favors. Also, be sure to speak with your family and make sure they’re prepared to support you through this transition. Changing jobs will impact them too. Regardless of where you are in your career and what’s going on in the economy, the job search process is hard. It takes time. The best suggestions I can give someone is to think about why you’re considering a change and create a plan to get from where you are to where you see yourself. The worst thing someone can do is react too quickly and find themselves in another toxic workplace. I know that the current work situation is tough but remember it’s tough you know. Have a plan and work the plan out. You’ll definitely succeed. All the best!

      A job change is a big decision and requires a good consideration. We are here to give reasons why you might want to change the job.

    1. Contact Big and successful businesses are built on a talented workforce this is not just a saying but a solid fact. Hiring an employee is one of the most important tasks for a business and it becomes even more critical when it comes to hiring the right employee.  From Mark Zuckerberg to Richard Brandson every billionaire has similar thoughts when it comes to building the team and every one of them has one thing in common, they always seek quality, punctuality, and skills in an employee. Hiring an employee could be challenging also it may seem like a play of fate and this is a very common feeling but, having a guide or tips on how to hire the right employee could help you in tons. So, here is the list of 7 best tips on hiring the right employee. Make a Plan No matter what you do, planning is the very first step when it comes to achieving a goal. A written plan which contains essential components of the hiring like job descriptions, a skill required, Pay grade, timing, etc. is needed to hire a person. The plan that you make for hiring must contain various details regarding the requirement of the job. The details that would want to add in the plan is as follows-  Title  The post or title of the job that you’re looking for should also be given clearly. Someone who’s educated enough to be a CEO wouldn’t want to be a normal worker. Hence, the specifications of the position while looking for a new candidate should be clear.  Pay Grade  For hiring the new employees, you should first and foremost be true about the pay grade. Pay grades facilitate the employment process by providing a fixed framework of salary ranges, as opposed to a free negotiation. So, you should just align with the grading system to let your company work smoothly.  Reporting Person Reporting Person means the person who prepares reports on updates, progresses, accomplishments, etc. of the employees as well as the company in total. While hiring new employees, you should first submit their resumes to this person so that he can well-prepare a report and it gets easier for you to decide.  Timings You should also clearly mention the shift timings while looking out or hiring new employees. The working hours, the holidays, the relaxations should be clearly told to them and see if they can work with it or not.  Department Name You should also clearly defined the department for which you’re trying to hire the new employees. Human resource departments are often organized along with functions and may be hierarchical in nature. Common units in human resource departments include recruiting, training and development, compensation and benefits administration, health and safety, and employee and labor relations.  Job Summary, Purpose/Value A job summary is a brief, general statement regarding the important functions and responsibilities that comes with a job. Job summaries hold a huge role in enticing a qualified candidate to apply for the position and an employee’s performance of his or her responsibilities.  Challenges With a job, comes its challenges. So you should explain the kind of challenges that your newly appointed employee is going to face and mentally prepare. If your plan contains details like these then it becomes easy for you to post job requirements on job portals as well so, having a written plan on the hiring process is very important. Internal Job Rotation When we talk about hiring, we always think about finding a new person and use his services but, internal job rotation is something that no one thinks of and in certain conditions the best choice. If someone from your Department “A” has skills and talent to handle Department “B” then why not promote him/her to department “B”. There are many reasons why internal job rotation is better than hiring a new person, but to name a few- employee retention encourages development, eliminates boredom, and much more. Culture Fit If you want to hire the right employee, you need to make sure that they’ll fit in the company culture, your team, as well as the job. The best way to do that is by setting up a good interview panel. The interview panel is the one who asks questions and reviews the profile of the person to be hired. So asking a few cultural questions, liking, interest, hobbies, etc. helps the interview panel in deciding whether they’ll be a good match for our business or not. Go Beyond the Resumes Resumes are the best-foot-forward of a person. You need to go beyond that. A resume contains a person’s experiences, education, the last job is done, and much more but a person is much more than a piece of paper so, analyzing a person on the bases of personality, attitude, and style of speaking should also be analyzed thoroughly. Background reference Background reference is a very critical part of the hiring process. It is done to ensure that whoever you are hiring has a clean record. You can use the information provided by the employee like aadhaar card, social security number, etc. we can identify whether the person seeking a job is criminal or not. You can get police verification done along with enquiring with the previous employer. Competency Mapping Competency mapping is a process of identifying the competencies of an employee based on education and skills. When a person does a job basically he is using his skills and experiences in the job to get a favorable outcome. As a business manager or CEO, your job is to help your employee in bringing the best out of them and that can be easily done by working on their skills, education, and experience. Many business hire special corporate training specialists to train their employees and motivate them to achieve a better result in the job they do. When you are hiring a person it becomes hard for you to identify the competency of a person, at such situations you need to look at their previous work, question them on technical grounds, and watch their skill in action by conducting a test. Choose a Good Employment Agency Many small businesses often hire an employment agency to make their recruitment easy but they often miss out on key information on identifying a good employment agency. Basically, there are three types of recruitment agencies which are as follows- General Recruitment AgencyStaff Recruitment AgencyExecutive Recruitment AgencyAll in one agency Genera Recruiting Agency helps you find industry-based staff meaning this agency focuses on a niche industry like healthcare, engineering, etc. A staff recruitment agency is focused on temporary staffing solutions, this type of recruiting is done for seasonal businesses or project-based hiring. Executive Recruitment Agency is the agency that helps businesses in recruiting in executive positions like CEO, CFO, and Managers, etc. You can categorize the top-level position to C-level positions in this type of recruiting. As the name suggests, All in one recruitment agency is the agency that provides all these services like Yunic Solutions, We provide multiple recruitment services in every industry. After selecting the type of agency, you can decide your budget and go about researching the best recruitment agencies. Hiring the right employee in a nutshell Hiring the right employee is all about the experience. For some people, the first employee could be the right employee and for some people no employee is the right employee.But in general, Hiring is a core HR process that is essential for business and requires dedicated time and effort.

      Hiring the right employee is very important for any business. You can use these 7 tips on hiring the right employee to ensure you get the best quality team.

    1. College courses emphasize expository writing, writing that explainsor informs

      Creative writing is less common in college than in high school, which may create a problem for some of us who were taught to be very descriptive, as now we will need to be more concise in order to inform in a meaningful way. We will need to learn new writing techniques, possibly with help from this textbook, as that's why I think they brought it up.

    1. nce. We recognize this as a unique set of circumstances and realize that others may not be in a similar position

      I think you bring up an important point here about privilege. Do you have any plans for expanding or extending this work to institutions that are different than your own?

    2. nce. We recognize this as a unique set of circumstances and realize that others may not be in a similar position

      I think you bring up an important point here about privilege. Do you have any plans for expanding or extending this work to institutions that are different than your own?

    1. Note: This rebuttal was posted by the corresponding author to Review Commons. Content has not been altered except for formatting.

      Learn more at Review Commons


      Reply to the reviewers

      Reviewer #1:

      In this manuscript the authors explore the requirements for centromere transcription using single-molecule FISH. Previous studies have found that centromeres are transcriptionally active in a wide variety of organisms. Centromere transcription has been proposed to facilitate Cenp-A deposition through chromatin remodeling and to directly contribute to centromere/kinetochore function by producing a functional ncRNA. However, we currently know almost nothing about how transcription is initiated at the centromere or how levels of centromere transcripts are controlled. This manuscript makes several major findings that are potentially of importance to groups studying centromere transcription. 1.) Centromere RNAs are produced by RNA Polymerase II and are localized in the nucleus of a wide-range of cell types. 2.) Centromere RNAs do not localize to the centromere, which is in contrast to several recent studies. 3.) Centromere proteins are not required for transcription of alpha-satellite sequences. 4.) Localization of centromeres to the nucleolus represses centromere transcription. Overall, this is a solid manuscript and has the potential to make a significant impact in the field. Below I suggest a couple of experiments and modification to the data presentation that could improve the manuscript.

      We thank this reviewer for their interest in this paper and agree with their clear articulation of the key points.

        • All of the experiments in this manuscript rely on detection of centromere RNAs using single molecule FISH probes. These probes are validated by showing the RNase treatment removes the FISH signal. A strength of this approach is that the authors use multiple different probe sets and achieve comparable results. However, there is no orthogonal validation that the probes detect alpha satellite RNA. All of the experiments in this manuscript would be significantly improved by showing that the results presented here can be confirmed by a different approach. I suggest that the authors use Q-RT-PCR to validate the smFISH results. * The smFISH probes provide a powerful and unique strategy to detect alpha-satellite transcripts. To ensure that these experiments are carefully controlled, we analyzed multiple distinct probe sequences that recognize alpha-satellite transcripts derived from different chromosomes, as this reviewer highlights. We also conducted an in-depth computational analysis to ensure that these probes do not match genomic sequences outside of alpha-satellite regions. However, we recognize and agree that a complementary method to detect these transcripts would be a useful addition to this paper. We are currently highly constrained in our ability to conduct these experiments due to COVID-19-related laboratory closures, but if feasible our goal for a revised manuscript would be to conduct qPCR experiments for a subset of the conditions that are the most central to the key results in this paper (focusing particularly on HeLa and Rpe1 control cell lines, CENP-C iKO, Ki67 KO, and RNA Polymerase I and RNA Pol II inhibitors).
      • Several results in this manuscript directly contradict results in published studies, but these discrepancies are not discussed. I believe the authors need to discuss the following discrepancies between their results and those in the literature: *
      • McNulty et al. Dev. Cell. 2017. Show that alpha-satellite RNA is transcribed from all centromeres and remains localized to the site of transcription. The different results and possible explanations for the differences should be discussed. *
        • Additionally, Rosic et al. JCB 2014, Blower Cell Reports 2016 and Bobkov et al. JCB 2018 all show that centromere RNAs localize to centromere regions. The differences between these studies and the authors results should be discussed. *
      • The authors show that satellite RNA cannot be detected on mitotic chromosomes. However, Johnson et al. Elife 2017, Bobkov et al. JCB. 2018, and Perea-Resa et al. Mol. Cell. 2020 show that EU-labeled RNA can be detected at the centromere during mitosis. The authors should discuss the discrepancy between their results and these studies. Is it possible that their smFISH probes do not detect nascent, chromatin-bound transcripts? *

      We believe that a strength of our paper is that it assesses alpha-satellite transcripts in individual intact cells using fixation conditions that preserve the native behaviors without disruptive and harsh extraction. As our results differ from those of other laboratories in some cases, we agree that it would be helpful to comment more directly on these differences with prior work. Points a, b, and c above all relate to the presence of alpha-satellite transcripts at centromeres. For the revised paper, we will include a discussion of these prior observations and some possible reasons for the differing results. In particular, we think that these discrepancies reflect two key differences:

      1. Other strategies with harsh extraction conditions likely eliminate soluble alpha-satellite transcripts that are not tightly associated with centromeres, whereas our work preserves these.
      2. It is possible that we are unable to detect nascent transcripts by smFISH as these are embedded within the RNA polymerase. Extraction conditions: An advantage of the smFISH probes used in our paper is that these require mild fixation conditions without prior extraction to better preserve cellular structures allowing us to analyzed intact cells, rather than chromosome spreads. Thus, our approach maintains the diverse alpha-satellite transcripts that are not bound to centromeres, and which may have been washed away in other studies. In contrast, some prior studies used stringent extraction conditions and primarily conducted experiments in chromosome spreads (not intact cells). Although it is not feasible to precisely determine the basis for differences without repeating this work the precise approaches and conditions from each paper and working closely with each group, we believe that these substantial technical differences explain our differing observations that reveal that the majority of alpha-satellite transcripts do not remain at centromeres..

      Nascent transcripts: As suggested by this reviewer, we agree that our differing conditions may mean that we are unable to detect nascent transcripts that are closely associated with the RNA polymerase, inaccessible due to their chromatin proximity, or that are not sufficiently elongated such that they are present to hybridize to multiple copies of the smFISH probes to be detectable. The alpha-satellite transcripts must be derived from centromeric and pericentromeric regions and so must exist there at some point (as also attested to the EU signals that this reviewer mentions in the work from our collaborative the Blower lab; we have also detected EU signal at centromeres). However, our work suggests that alpha-satellite transcripts do not persist at centromeres indefinitely once generated, with mature transcripts in the nucleoplasm and liberated from chromosomes during mitosis. We believe that the combination of the relative inability of our smFISH probes to detect nascent transcripts, but stringent conditions disrupting non-centromere bound transcripts for prior work likely explain these distinctions.

      • The authors show nicely that deletion of Ki-67 reduces centromere localization to the nucleolus and increases centromere transcription. However, this has no effect on centromere function. Studies from the Earnshaw lab (e.g. Nakano et al. Dev Cell 2008 and Bergmann et al. EMBO J. 2011) show that increasing or decreasing centromere transcription results in loss of kinetochore function on a human artificial chromosome. The authors should discuss the differences between their results and these studies. Is it possible that the small size of the HAC exaggerates the importance of the correct levels of centromere transcription? *

      We are big fans of the Earnshaw lab work. In this case, there are a couple of possibilities to explain the strong effect that the Earnshaw lab observed on kinetochore function by perturbing centromere transcription. First, the degree of the change in centromere transcription may make a big difference. The Ki-67 results in an approximately 2-fold increase in alpha-satellite smFISH foci, which may still be within a permissive range for normal kinetochore function. Second, the experiments from the Earnshaw lab rely on targeting activating or silencing proteins to the centromere region, and it is possible that changes in centromere chromatin downstream of these factors contribute to the observed phenotypes in addition to altering the amount of centromere transcription. We will include a brief discussion of the Earnshaw work in a revised paper.

      • The authors treat cells with transcriptional inhibitors for 24 hours. I am concerned that this may result in massive cell death. It would be helpful to include cell viability data from these experiments. *

      We appreciate this point and agree that cell lethality is an important consideration given the essential role of the RNA polymerases. For the inhibitors, we first treated the cells for a variety of different time points to evaluate these behaviors. For example, we found that we could treat cells with RNA Polymerase II inhibitors for as much 48-72 hours without detecting noticeable cell death. Thus, at the 24 hour time point, the cells remain viable and intact, as is also visible in the images showing DNA staining for these treatments in Figure 3. We also note that this timing is consistent with prior studies that block transcription or translation. However, we did additionally conduct these experiments at earlier time points (5 hours and 12 hours post-drug addition) and obtained similar results. For example, for the Cdk7 inhibitor using the ASAT probe, we observed the following smFISH foci/cell: Control (3.4 foci/cell), 5 h (1.5 foci/cell), 12 h (1.2 foci/cell), 24 h (0.9 foci/cell). There is a clear effect even at 5 hours of treatment and a continued downward trend. Both for simplicity and because the replicates and number of cells that were quantified were lower for these conditions, we chose not to include these in the paper. We will include a statement regarding these earlier time points in the revised version.

      • In Figure 3C the authors examine the effects of centromere protein knock outs on centromere transcription. To me this is the most important experiment in the manuscript and is a major step forward for the field. The authors use inducible CRISPR knock out cell lines that are not 100% penetrant. It would be helpful if the authors could describe how they ensured that cells included in the image quantification were knock out cells. *

      Based on this comment and the other questions from the other reviewers, we recognize that we need to provide a much better description of the CRISPR knockout strategy, the prior validation of these cell lines, and the strategies that allow us to use these cell lines in a robust manner to ensure that we are effectively eliminating the target genes. We have systematically tested this strategy in multiple cases and find that this strategy is superior to RNAi for its efficacy and the potency of the phenotype, particularly for this type of cell biological assay.

      The Cas9-based strategy is a highly effective way to conditionally eliminate essential genes. In this case, the efficiency of the Cas9 nuclease ensures that the genomic locus is cleaved in essentially 100% of cases. As this is repaired in an error prone manner and typically using non-homologous end joining, 66% of individual events result in frame shifts mutations that disrupt the coding sequence of a target gene, with ~50% of cells resulting in frame shifts in both copies of a gene. In addition, if a sgRNA targets a region of a gene that cannot tolerate mis-sense mutations, this will result in an even greater fraction of mutant cells. Thus, these inducible knockout cell lines result in robust and irreversible gene knockout, with a large fraction of cells (50% or more) displaying a clear phenotype. However, it is also true that there are a subset of cells within the population that will repair the DNA damage following Cas9 cleavage in a way that preserves protein function such that they behave similarly to control cells. Importantly, this means that there will be two classes of cells within a population – those that are unaffected, and those that are strongly affected. As we are analyzing each cell individually instead of creating a population average, this will capture this phenotypic diversity to reveal two populations of behaviors in cases where eliminating a gene results in a substantial change in smFISH foci. For example, the smFISH foci/cell data for the CENP-C inducible knockout (Fig. 3C and 3E) indicates that many cells have smFISH foci numbers that are comparable to control cells, but others that display substantial differences and highly increased numbers. An ideal control in these experiments would be to additionally analyze the levels of the target protein together with the smFISH analysis. Unfortunately, many of the antibodies are not compatible with the conditions needed for the smFISH. For CENP-C, the antibody that we have is not compatible with the conditions that we are using for the smFISH, so it is not feasible to co-stain these cells as suggested. Instead, for our analysis of the centromere-nucleoli localization (for example), we used the presence of a clear CENP-C interphase phenotype (“bag of grapes” resulting from chromosome mis-segregation) as an indication that the cells had been knocked out for CENP-C.

      The majority of the Cas9-based inducible knockouts that we used for this paper were generated previously in the lab (McKinley et al. 2015; McKinley et al. 2017). For the centromere protein knockouts (McKinley et al. 2015), these were analyzed previously with respect to phenotype and monitored for the depletion of each gene target over time. For the larger collection of cell cycle and cell division inducible knockouts, for our prior work we systematically validated each of these with respect to their phenotype (see http://cellcycleknockouts.wi.mit.edu). Thus, we are confident that each of these cell lines is functional and effective for eliminating the target gene.

      For conducting the experiments using the inducible Cas9 cell lines in this paper, we used the presence of these previously-defined phenotypes within the population as a validation that the strategy is working. Again, in general we find these knockouts are both penetrant and severe in their phenotypes. Importantly, for this diverse set of genes, we note that our goal was to broadly survey diverse factors to identify changes in alpha-satellite transcript levels. We intended this analysis as a “screen” where we would identify factors that resulted in a substantial change in the number of smFISH foci. As with any larger analysis, it is possible that there are false negatives where we did not detect a strong effect on transcript levels (such that they may contribute to centromere transcription). We have tried to use caution not to indicate that this data excludes any possible role for these factors in transcript levels, although in general the majority of the tested factors did not show a substantial change in smFISH foci. For the revised paper, we will make an explicit statement to this effect.

      • On p8. The authors cite Quenet and Dalal. eLife 2014 for the idea that transcription during G1 is important for new Cenp-A loading. They should also cite Chen et al. Dev. Cell 2015 and Bobkov et al. JCB. 2018. *

      Thank you for these helpful suggestions. We will update the text to incorporate these references.

      Reviewer #2:

      The study by Bury et al. investigates the formation of two different types of alpha-satellite transcripts (ASAT, SF1 and 3) in different human cell lines. Using smFISH they find that during the cell cycle these centromeric transcripts don’t stay at the centromere and are found in the cytoplasm after mitosis. Using specific inhibitors, they find that transcription is dependent on RNAPII, but not on various centromere and kinetochore proteins taking advantage of an inducible CRISPR-depletion system that the lab had previously developed. Interestingly, they find that CENP-C, a major component of the centromere and previously characterised as an RNA-binding protein, negatively regulates alpha-satellite transcript levels. Another regulator for transcript levels appears to be centromere-nucleolus interactions (as also indicated in the title) acting to suppress expression of these non-coding RNAs.

      This is overall a really interesting study and indeed, transcription at the centromere is little understood at this point. Given the importance of the centromere the findings in this manuscript will be of high interest to both researchers in the field and a general audience. There are novel and interesting insights into centromeric transcripts but the study still requires some controls.

      We appreciate this reviewer’s kind words and their clear description of our work.

      1) The authors state that the majority of smFISH foci do not colocalise with centromeres in a combined IF/FISH experiment (some quantification and a % of that subpopulation should be given somewhere). This is a bit concerning but of course could also be true. It either means that alpha-satellite transcripts leave the centromere as suggested by the authors (although some should be visible at the centromeres during the act of transcription). Alternatively, a trivial explanation would be that there is a lot of unspecific staining, which can occur in FISH-experiments to varying degrees. The RNase treatment to control for the absence of potential DNA hybridization is convincing, but the FISH probe could also interact with non-centromeric cellular RNA. With the centromere localisation as a reference point gone, some control is needed to validate that the RNA-FISH signals are indeed recognising alpha-satellite RNA that emerged from centromeres. The authors could try competition experiments titrating unlabelled specific or unspecific DNA probes alongside their labelled specific FISH probe into their FISH experiment to see if they lose or maintain the signal and the number of foci. The specific RNA FISH probes could also be used in DNA FISH, to demonstrate they are working and recognising specific centromeres.

      For understanding this behavior, we believe that an important feature of alpha-satellite transcripts is that they are relatively stable (protected from nucleases within the nucleus), but that their overall number is low, consistent with transcription of other non-coding regions across the genome. Thus, if a transcript were produced at centromeres, but subsequently diffuses away, only a small subset would be detectable at centromeres. In addition to our validation these probes using RNAse, we would like to highlight that we have analyzed multiple distinct sequences that recognize different subsets of alpha-satellite repeats. In each case, the observed behaviors are very similar. In addition, the nature of the oligo FISH method requires multiple individual probes to anneal to the same transcript such that a signal is only detected if a sufficient number of oligos bind to the same transcript. This makes nonspecific binding unlikely to contribute to a false signal. Finally, a subset of the perturbations that we tested that are relevant to centromere function (including the CENP-C inducible knockout) clearly affect the levels of these transcripts, supporting a centromere origin. The additional control experiments suggested by the reviewer could be useful, but are technically complex with their own caveats in interpretation and we do not feel that they would add substantially to the existing paper. Instead, as discussed in response to Reviewer #1, point #1, we plan to validate key results described in the paper using qRT-PCR (if possible based on current experimental constraints in the lab associated with COVD-19).

      As described above in response to Reviewer #1, point #2, we also believe that some differences with prior work suggesting that alpha-satellite transcripts localize to centromeres may be due to stringent extraction conditions that eliminated non-centromere bound transcripts, while at the same time reflecting our inability to detect nascent transcripts. Quantifying “colocalization” within the nucleus is limited by the resolution in light microscopy, and we would prefer to use caution in defining which transcripts in our smFISH analysis overlap with centromeres. However, we believe that our work clearly highlights the fact that a general feature of mature alpha-satellite transcripts is that they localize throughout the nucleoplasm and are not strongly associated with mitotic chromosomes.

      2) Apart from Figure 4, there is no analysis shown for statistical significance. This should be done for most if not all quantifications. Are indeed ASAT and antisense RNA Foci number not significantly different? The authors say that the levels of alpha-sat RNA in Rpe1 cells are not substantially different from other cell lines, but is it also not significant (Fig 1F)? In Figure 2D it is concluded that transcripts foci number are increased in S/G2 (from G1) and remain stable in mitosis, but it looks like there is an increase in mitosis. Again, it looks like the higher number of smFISH foci/Cell is significantly higher for both ASAT and SF1, so some statistical analysis would be required here.

      For this paper, we quantified hundreds of cells for each condition, measuring the number of foci/cell in each case. Because of these large n’s, even relatively small differences between samples become statistically significant when tested using standard statistical comparisons (unpaired T test and one-way ANOVA test amongst others). For our experiments, every sample condition included an analysis of control cells, allowing us to compare the control condition to any perturbations on the same day. However, there is some variability between these different replicates, with the average number of ASAT smFISH foci/cell in HeLa cells ranging from 3.4 to 5.6. When compared relative to each other, a subset of these control samples will appear to be statistically different from each other despite the fact that this is not a substantial difference between replicates. Similarly, the majority of the tested inducible knockout cell lines are statistically different from control cells, even when the differences are relatively minor. Therefore, we have tried to use caution when applying the double-edged sword of statistics to these analyses. Instead, we have tried to consider differences with a “substantial magnitude” instead of “statistically significant” differences that may make modest, but statistically significant differences seem artificially more important. We believe that the graphs in which every data point is represented, together with listing the average number of foci/cell in each condition allow the reader to evaluate this data for themselves. Many of the trends that this reviewer highlights are indeed interesting comparisons to consider for future work.

      3) Starting with the description of Figure 1E in the main text the paper equates foci count of smFISH per cell with RNA transcript levels. I'm not convinced that these are necessarily the same. You could have many weak foci or few very bright with the same amount of overall transcripts in both. The authors start out introducing smFISH as highly sensitive "for accurate characterisation of number ...of RNA transcripts". This suggests that foci intensity could be used as a read-out for transcript levels. It should be possible to measure individual intensity of the foci for a subset of images. Do foci intensity correlate or anti-correlate with foci numbers? Is the sum of the intensities of all the foci less variable than the foci number for an individual cell type?

      Due to the repetitive nature of alpha-satellite sequences, an increased intensity of a smFISH foci could reflect either the close proximity of multiple separable transcripts, or a longer transcript with multiple binding sites for the smFISH probes. Because of this, throughout the paper, we have referred to these as “foci” instead of stating a specific transcript number. As part of the automated computational analysis of the smFISH images, we additional analyzed foci intensity. In general, these values were similar across a cell population and between various perturbations with the key results and findings consistent whether we measured foci number or overall foci intensity per cell. However, foci intensity can vary slightly across a coverslip (technical constraints, not biological differences), and thus we have focused on foci number as a more consistent metric that correlates with the production of alpha-satellite transcripts.

      4) I really like the use of the inducible CRISPR system to remove various centromere factors. However, some validation would be required to show that the system is effective in removing the proteins of interest in these experiments. For instance it would be helpful to show in Figure 3D an additional panel with CENP-C staining. Also for a subset of factors, some antibody staining co-staining with the smFISH could be provided in the supplemental material.

      We appreciate this point. However, we feel that the existing experiments appropriately consider the nature of the knockout. First, we primarily used Cas9-based inducible knockouts that were generated previously in the lab (McKinley et al. 2015 and McKinley et al. 2017). As these knockouts have been described previously and extensively validated with respect to phenotype (in every case; see http://cellcycleknockouts.wi.mit.edu for example) and antibody staining (in selected cases), we have not repeated this here for the diverse cell cycle knockouts used. In general, we find these knockouts are both penetrant and severe in their phenotypes. Given the broad number of knockouts that we tested, this is not feasible in every case. We also intended this analysis as a type of “screen” where we could validate any “hits” that were observed, and will use caution in our wording not to imply that a negative result is decisive.

      The important exceptions to this are CENP-C (which we analyzed more closely) and Ki67 (for which both the inducible and stable knockouts were generated for this paper). For Ki67, the antibody staining is shown and we believe that this is clear. For CENP-C, the antibody that we have is unfortunately not compatible with the conditions that we are using for the smFISH, so it is not feasible to co-stain these cells as suggested. For the smFISH analysis in the inducible CENP-C knockout, we analyzed every single cell, including some cells that are likely to have intact CENP-C levels. Thus, if anything, the potent increase in smFISH foci underrepresents the dramatic effect of CENPC depletion. Based on our prior work (McKinley et al. 2015) we found that the CENP-C knockout results in a pervasive “bag of grapes” phenotype in which chromosomes mis-segregate during mitosis and are packaged into separable interphase nuclei. For the analysis of the nucleoli, we selected cells that displayed this clear phenotype (as shown in the figures).

      5) Since none of the CRISPR iKO has a particular inhibiting phenotype it would be useful to include some positive control in the CRISPR experiment. Would it be possible to use a CRISPR iKO target that affect some factor of the transcription machinery (RNA Pol II or similar) to reduce transcript levels?

      Generating additional Cas9 iKO cell lines is feasible, but would be time consuming. In this case, we are not convinced of the value of generating and validating these additional cell lines (particularly with the additional current constraints due to COVID-19). For evaluating the role of the RNA polymerases, we believe that the effect of the drug treatment is clear. For creating a positive control to assess whether the CRISPR iKO strategy is a feasible way to conduct these experiments, we would like to highlight the CENP-C iKO cell line, which has a potent effect in this assay.

      6) The authors find a negative correlation between the nucleolus-centromere association and the number of alpha sat foci. This is really interesting and they suggest that the nucleolus association could negatively regulate centromere transcription. However, this correlation is rather indirect in the sense that cells with a higher-degree of nucleolus-centromere localisation have fewer smFISH foci and the inverse, disruption of the nucleolus increases smFISH foci number as a whole. A model based on physical association would suggest that a nucleolus associated centromere produces less or no transcripts. Given that this is not a population-based assay, it should be possible to address this directly by analysing the location of individual centromeres and corresponding transcripts to strengthen the hypothesis. This could be done by either analysing the smaller subset of centromere-associated foci that colocalise with the smFISH signal and test whether the majority of these signals are proximal or distal to the nucleolus (this would not work or be less meaningful if the subpopulation is very small). Or doing a combined DNA/RNA FISH experiment. The expectation would be that DNA FISH signals of centromeres close to the nucleolus would not produce an RNA FISH signal somewhere else, and vice versa.

      We predict that centromere-nucleolar associations are dynamic. Thus, we anticipate that centromeres would be associated transiently with the nucleolus (perhaps for a few hours), and that a given centromere would not be associated with the nucleolus in every cell at a specific time point. Thus, we believe that analyzing these behaviors across a diverse range of cells, as we did for this paper, is appropriate. In addition, technical considerations make these suggested experiments prohibitive. Defining the relationship between a centromere RNA and its originating centromere would require combined DNA and RNA FISH. The repetitive nature of alpha-satellite repeats and the strong similarity of these sequences between chromosomes makes it highly complex to visualize an individual centromere. Even if we were able to do this, the conditions required to simultaneously detect nucleoli (immunofluorescence), RNA (smFISH), and DNA (requires denaturation and hybridization) make this such that it would be complex to correlate the localization of an individual centromere with the levels of the corresponding alpha-satellite transcripts. In addition, these RNAs are likely to persist for an extended duration (possibly throughout the course of an entire cell cycle), such that they would not necessarily correlate with the current localization behavior of the centromere from which they are derived. For future work (beyond the scope of this paper), we plan to create cell lines expressing both centromere (CENP-A) and nucleolar markers (for example, Ki67) to conduct time lapse imaging to assess the dynamic associations between these structures.

      7) At the end of the abstract, the authors conclude that the control of centromere transcription might be regulated by the centromere-nucleolar contacts to modulate chromatin dynamics. What does that really mean? One possibility they give in the discussion is rejuvenating centromeric chromatin. It would be nice if they could show some effect along those lines at the centromere in one of the manipulations they did (either through inhibiting or increase transcription). At least as discussed in the paper (Supp. Fig 3 D) it appears that overall levels of CENP-A are not affected. Is this different for newly loaded CENP-A? Or some other aspect of chromatin dynamics that is modulated? I realise that this might have been difficult to detect and therefore missing in the current study.

      In a separate study from our lab as part of our recent work (Swartz et al. 2018), we found that CENP-A is gradually incorporated at centromeres in non-dividing quiescent cells, including non-transformed human Rpe1 cells and starfish oocytes. In the case of oocytes, which contain a substantial pool of mRNAs such that they do not require ongoing transcription for viability, we found that inhibiting RNA Polymerase II and preventing ongoing transcription blocked the incorporation of newly synthesized histones, including both canonical histone H3 and CENP-A. We realize that our description of this prior work was not sufficient to understand our integrated model, which relies on information from both papers. For the revised paper, we will update our discussion to better describe this data and present our model.

      • Page 8: The authors state that as cells entered mitosis, dissociation of smFISH foci from chromatin was observed. While the absence of co-localisation of DAPI and smFISH signals is obvious in mitotic cells, what evidence is there that smFISH foci are chromatin associated in interphase nuclei? Rephrasing this bit might avoid confusion here. *

      We appreciate this point. We did not mean to imply that the smFISH foci are bound to (or associate with) chromatin in the interphase nucleus. We will reword this as suggested.

      Reviewer #3:

      The manuscript of Bury et al. addresses how alpha-satellite transcription around centromeres is regulated. Using smFISH to detect alpha-satellite RNA transcripts, the authors find that alpha satellites are transcribed by RNA pol II, but their transcription is independent of centromeric proteins. In addition, they present evidence that nucleolar association represses alpha-satellite transcription. The data is convincing, solid and generally supports the conclusions. The manuscript includes appropriate control experiments, such as test for the validity of the RNA FISH probes. The manuscript is well-written and easy to follow, also for someone who is not directly an expert in the field.

      The authors use a single-cell technique (smFISH) to look at the localization and transcription of alpha-satellite transcription from centromeres. The technical advance of this paper is limited, as smFISH is a well-established technique by now. Nevertheless, applying this single-cell approach to these repetitive regions has resulted in new insights regarding the regulation of alpha-satellite transcription, especially their localization of centromeres to nucleoli. Regarding the significance of these insights in the context of centromere biology/regulation and its literature is hard to evaluate for me, because this is not my field of expertise (my background is in single-cell transcription regulation). As a researcher from a related research field, I think the findings of this manuscript are mostly relevant for the direct research community of centromere and alpha-satellite biology, but not for researchers outside the field.

      We appreciate these comments regarding the carefully controlled nature of our paper and the value of the advances for understanding alpha-satellite transcription. We also agree that smFISH is an established technique, although it has not been applied to these repetitive alpha-satellite sequences in prior work, allowing us to make important new observations usng the studies in this paper.

        • The description of the inducible knock out cell lines is very limited. My main concern is how is checked that the gene is actually knocked out. I went back to the referenced paper, but it is still is not clear to me whether the new knockouts are sufficiently checked. It would be more convincing if the authors could show western blots or other evidence that their knockouts are working. In any case, the description of the knockout generation should be more elaborate. * This important point was also noted by the other reviewers. Please see our responses to Reviewer #1 point 4 and Reviewer #2 point 4. As described above, for a revised paper, we will provide an improved description of these knockout cell lines, our validation of these tools, and how we conducted the experiments in this paper.
      • The authors nicely show that there is an inverse correlation between nucleolar association of the centromere and alpha-satellite transcription. The data supports this claim, but given the many knockouts and cell lines that were tested, with many intermediate phenotypes (such as CENP-B), I find the correlation based on 4 points a bit sparse. I would recommend filling up figure 4C with a few more mutants, to show that the inverse correlation holds for all mutants. These experiments would be straightforward for the authors, as the knockout/cell lines and techniques are already available. *

      We see a compelling general correlation between the fraction of nucleolar-localized centromeres and alpha-satellite transcript levels. Our goal for Figure 4C was to highlight this correlation for a selected subset of conditions. However, we do not believe that there will be a precise linear correlation between transcript levels and nucleolar centromeres under every condition. Indeed, it is quite possible that some perturbations would affect transcript levels without altering nucleolar associations. This is particularly true for perturbations that cause subtle phenotypes. Systematically analyzing centromere-nucleolar co-localization for each of the knockouts represents a substantial undertaking that we do not feel would contribute substantially to this existing paper.

      • The nucleolar repression is also supported by the Fibrillarin and Ki67 knockout. These are nice experiments which support their findings. What I am missing is whether these data quantitatively agree with the inverse correlation. Are these mutants completely lacking nucleoli, and if so, would you not expect both mutants to show the same upregulation? Similar to my point above, where do these mutants fall in the graph of figure 4C? *

      For the perturbations described in this paper, we believe that inhibiting RNA Polymerase I most closely approximates the condition where nucleolar function is eliminated. Although Ki67 is a nucleolar protein in interphase, loss of Ki67 does not cause lethality indicating that nucleolar function is largely intact. We agree that it would be a good experiment to assess nucleolar-centromere associations in the Ki67 knockout. In fact, we have tried these experiments several times. However, due to the absence of Ki67 (for which we have the best localization tools), we instead needed to use Fibrillarin to monitor nucleoli. We have found this antibody to be much more finicky and not as readily compatible with the fixation conditions needed to detect centromeres. Thus far, we have not been able to generate clear data for this behavior.

      • Related to this, since their imaging techniques have single-cell resolution, I wonder if cells that contain many centromeres in the nucleolus have less alpha satellite transcripts than cell with few centromeres. *

      The correlation between centromere-nucleolar associations and alpha-satellite transcript numbers is strongly supported by our data across a population. However, analyzing this in individual cells is additionally complicated by the fact that we found that transcript levels vary over the cell cycle (low in G1, higher in S/G2). In addition, monitoring each of these markers in individual cells is technically complicated. Thus, while we appreciate this suggestion, we believe that our data stands on its own.

      • One claim that is a bit speculative is the suggestion that transcription itself and not the RNA may be required for the function of the alpha-satellites. This is indeed supported by the fact that most transcripts are not localized at the centromeres. However, this contrasts to the findings of the papers that increasing alpha-satellite transcription in different mutants does not appear to result in any phenotype on centromere function. For a non-expert, the function of these transcripts/transcription itself is not clear from the current manuscript, so I would recommend discussing the nuances of its functions in more detail in the discussion. *

      We agree that our model is speculative, but have chosen to include this to provide our perspective on the possible roles for centromere transcription based on this paper and our other recent work (Swartz et al. 2018). We believe that our data provide a context and set of constraints for potential roles of centromere transcription, but also agree that future work is needed to resolve these. Based on this comment and those from the other reviewers, we will also provide a better description of the data in the Swartz et al. paper, which analyzed different features of centromere transcription.

      • To quantify the smFISH data, the authors count the number of foci. From the images, it looks like the different foci have very different intensities. This may occur if the transcripts are different length when transcribed from different genomic regions. However, this may also occur if several RNA co-localize to the same spot, i.e. if one spot contains several RNAs. Can the authors verify that the distribution of spot intensities matches the expected intensities based on the different transcribed alpha-satellite regions? *

      Please see our response to Reviewer #2, point #3.

  10. learn-us-east-1-prod-fleet01-xythos.s3.amazonaws.com learn-us-east-1-prod-fleet01-xythos.s3.amazonaws.com
    1. But, some of you may be saying, I am not really all that interested in the history of youth literature; I plan to teach contemporary youth literature to contemporary children, tweens, and teens. Or: I want to write for young people, so my goal is to

      I think it is interesting to consider the idea of historic negligence, when we as educators are in the profession of teaching the truth and not censoring it. To that end at what point does this teaching of this historical literature become selective based on our own biases and interest?

    2. "Ever since there were children, there has been children's literature:' While this is very likely true, it requires a feat of informed imagination to reflect on the forms that literature took before print, as well as to extrapolate from the bits and scraps that have been preserved over time in order to make some generalizations about what young readers might have read and enjoyed prior to our present age; well-loved books, like well-loved toys, may not have survived the ravages of time and rough handling as well as those items that were not subjected to everyday use. Yet this kind of imaginative reconstruction is precisely what we must do to fully account for the stories, educative texts, and poetic forms shared with young people throughout the course of human history.

      I think it is interesting to form the idea about what constitutes children's literature and how it has changed throughout the times. Is it natural for the progression of these literatures to change in tone and purpose as our taste for literature changes?

    1. We may learn the wish (will) of nature from the things in which we do not differ from one another: for instance, when your neighbor's slave has broken his cup, or anything else, we are ready to say forthwith, that it is one of the things which happen. You must know then that when your cup also is broken, you ought to think as you did when your neighbor's cup was broken. Transfer this reflection to greater things also. Is another man's child or wife dead? There is no one who would not say, This is an event incident to man. But when a man's own child or wife is dead, forthwith he calls out, Woe to me, how wretched I am! But we ought to remember how we feel when we hear that it has happened to others.

      I think this is his way of saying "those is glass houses shouldn't throw stones". Also to maintain the idea of "karma" or what comes around, goes around.

    1. With the backing of Green Alliance and some philanthropic funders, I set up a training programme. We offered parliamentary candidates and new MPs the chance to learn about the science, policy and politics of climate in a series of tailor-made workshops. We worked with small groups of around 10 politicians, all from the same party, to allow them to question and debate freely.

      I wonder how much this cost, to design these kinda of workshops?

    1. NCTE / IRA Standards for the English Language Arts

      Are there reasons to accept or reject these standards? How do NCTE's standards reflect our personal values as English teachers? Do they reflect the values of Greenhills?

      Have we created any documents that distill our mission as an English department? I think there may be one from a few years ago? []

    1. Over the weekend, The Washington Post published a chilling description of the first day of reopened business at a mall in an upscale suburb of Atlanta, Georgia. Bored rich people wandered the aisles of Anthropologie and Crate & Barrel, pawing at the wares, some with no masks or hand sanitizer in sight. One woman getting a manicure said, “I went to the antique mall yesterday on Highway 9 and it was just like—it was like freedom. We have to get out.” Everyone who is lucky enough not to have to work is chafing at being stuck at home, but one has to laugh at the idea that American Freedom is visiting the antique mall on Highway 9. Then again, maybe she’s right: Maybe that is American Freedom. The right to visit whatever business you please is not one enshrined in the Constitution. There is no constitutional right to go to Arby’s. But from the start, lockdown protesters claimed their constitutional rights were being trampled by the stay-at-home orders. Protest signs in Southern California read: “Pandemics does [sic] NOT cancel our Constitutional rights!! Freedom over fear,” and “No Liberty, No Life, Reopen California.” My colleague Matt Ford is not the only one suggesting tweaks to America’s foundational documents, it seems: The angry bourgeoisie of the “reopen America” movement has invented an entirely new category of civil rights. On Monday, in New Jersey, a protester at a gym that opened despite the shutdown order held a sign that said, “The constitution is essential,” and another held a sign that said, “Right to work/Right to worship/Right to free speech/Right to be free.” A protester in Washington said: “I lost my job as a bartender and now I live on way less income, and I’m upset that my constitutional rights are being trampled all over.” Advocates of a federal job guarantee will be thrilled to hear that there is a constitutional right to a job. This line of criticism might make a little more sense if the issue in question was the federal government enforcing a broad order to physically keep residents inside their homes. But there are no patrols of soldiers forcing citizens to remain indoors, certainly not among the white and wealthy. Take Newport Beach, California, for example: The city said on April 3 that it would not enforce Governor Gavin Newsom’s stay-at-home order, saying the governor “expects Californians to do the right thing” and “self-regulate.” Californians can still go to the grocery store, the pharmacy, the laundromat. They can still shop online. They can still go for walks. They can even go to Arby’s—drive-thru, at least. But the people who took their AR-15s to Subway, citing their “God-given freedoms as Americans,” are not talking about their actual constitutional rights. They are talking about their unique understanding of the mythical promise of this nation, which is a place where a man can drive a big-ass truck, drink a 64 oz Slurpee, and go to the air-conditioned mall to purchase Stuff. Because this is America, and if that’s what America is, that’s what the Constitution must necessarily be protecting. Children are educated in our schools that the Constitution defends the American “way of life.” It’s not that surprising if the end result is that people think this is actually true. In this case, they rage at being prevented from living their normal lives—going to Bed Bath & Beyond, drinking margaritas at Chili’s with friends, golfing—because they consider the conduct of their normal lives to be inseparable from the purpose of America. The right of essential workers not to die of the coronavirus does not seem to enter the equation. White dudes who own big pickup trucks and live in Newport Beach have rarely been at risk of overaggressive policing. The fact remains that even if a mandatory quarantine were harshly enforced by the government, all the available evidence suggests that the white people who feel threatened enough by government overreach to parade around state capitols with guns are not the ones who would be at risk of violence from the police. New York City data revealed earlier this month that 35 of the 40 people arrested for social distancing violations were black, four were Hispanic, and only one was white. White dudes who own big pickup trucks and live in Newport Beach have rarely been at risk of overaggressive policing. This is simply the conservative mentality: a constant state of perceived victimhood. The notion that oppressed groups enjoy being persecuted, because it lets them complain about victimization, becomes a weird sort of envy. This leads the would-be wretches to invent forms of oppression, like having to buy two iced teas instead of getting a free refill, so they get to have a turn at complaining. They may genuinely not realize that actually oppressed groups are not having fun when they object to their situation. The conservative-fear-industrial complex runs on conjuring ever-changing enemies of liberty, each browner and more Marxist than the last, because it makes them feel alive. Hence the outrage directed at Representative Alexandria Ocasio-Cortez for noting that we will need to consume less beef if we want to prevent apocalyptic climate change, which was in turn reminiscent of the anger at Michelle Obama for proposing that children should be given healthy food at school instead of pizza and white bread. How dare you, government bureaucrat, suggest that I shouldn’t kill myself with a diet of only red meat and Mr. Pibb? Never mind the millions for whom a diet of processed food and soda is all that’s affordable or available; the real oppression is you criticizing the choices I use to define myself, which, for some reason, now include eating burgers. For anyone, particularly nonwhite women, to suggest otherwise is essentially the new Gestapo. There is no logical connection between your constitutional rights and shopping at businesses you like. But it makes the desire for normality, which we all crave, much more defensible if you frame it as being a martyr for American liberty in the vein of Thomas Jefferson, and not a sad admission that the America of sterile malls, connected by vast freeways and staffed by the underpaid masses, is what you would actually die for.

      Obviously written by someone who looks down on people that either have different values or beliefs from her. She has never been to Newport Beach because there are very few big pickup trucks in Newport Beach - more electric cars).

    1. Recovering heroin addicts in Edinburgh who need to self-isolate are now receiving home deliveries of methadone, as experts call for further creative thinking to save lives. With Scotland’s drug deaths rate expected to have risen even higher when annual figures are published this summer, there have been warnings of another public health emergency on top of the coronavirus pandemic, as addicts abandon their substitute treatment prescriptions because of lengthy pharmacy queues, become estranged from support services, and risk overdose by turning to more dangerous alternatives when street supplies of heroin disrupted. The Guardian spent time on the Edinburgh delivery run with two managers from the recovery charity Change Grow Live. After picking up chemist prescriptions, Rab Dylan and Lukas Waclawski then drop off the bags of opiate substitute – usually methadone – clean needles, or kits of the life-saving overdose reversal drug naloxone at the doorstep. Some visits are daily, in particular where an individual is deemed an overdose risk or in need of regular supervision, while others are every few days in an attempt to ease pressure on services. One homeless couple have been housed four miles away from their regular pharmacy, to the north of the city. Dylan and Waclawski meet them in the car park of their bed and breakfast for a contactless delivery, wearing gloves and masks. As a salty breeze blows in from the Firth of Forth, Waclawski demonstrates the use of the pre-filled naloxone syringe while maintaining strict physical distancing. “You can inject it through your clothes,” he advises, “into the largest muscle, which is usually the thigh.” Karen (not her real name), who has been in treatment since January, says the deliveries have been “so helpful” since she and her partner were instructed to self-isolate after developing coronavirus symptoms. “Before we had to go to the chemist every day and I was paranoid, because people in the queue were coughing and sneezing, not covering their mouths.” “The guys are just happy to have someone to check in with,” adds Dylan. “Given that a lot of services are moving away from face to face, if we can get out there and get some interaction, it has got to be positive.” Lauren Gibson, the lead pharmacist for substance misuse and prisons at NHS Lothian, who is coordinating the delivery scheme, says: “Relationships with this patient group are so important and we had to find ways to keep that going. It’s not just about getting access to methadone and medications; it’s about the full recovery package, such as food parcels, naloxone and overdose support, welfare checks, mental health – it’s about keeping those vital relationships going with patients, even if that’s virtually or on doorsteps.” Last week the Scottish government’s drug deaths taskforce called for home delivery outreach networks to be rolled out across the country, and many local authorities have followed suit. The taskforce also urged ministers to treat drug users as a priority group for coronavirus testing, and floated the use of depot injections, like a once-a-month injection of slow-release heroin substitute, which the Welsh government has now made routinely available for recovering addicts. There is a wide consensus about the need for immediate access to substitute treatment, particularly with the expectation that more addicts will be seeking help as street supplies dwindle or become more dangerous or prohibitively expensive. But street workers report that in some areas desperate people are still waiting for more than a week for a prescription. They take the pragmatic view that some take-home methadone will inevitably end up back on the street, but that this must be balanced with the urgent need to make treatment as simple as possible. Tracey Clusker, a nurse manager for substance misuse at Midlothian health and social care partnership, is blunt: “We have to think creatively to save lives. If there’s any time people need rapid access to a prescription, it is now.” Clusker has been delivering methadone, along with food parcels, condoms and sanitary towels, to at-risk addicts who normally attend her innovative cafe clinic, and holding weekly Zoom groups where they can discuss how they are coping with isolation. Gibson and Clusker are hopeful that the pandemic may foster a shift in priorities both for those struggling with addiction and the public at large. Gibson says: “One positive in a negative time is that we are managing to engage people into treatment who wouldn’t consider it previously. We are working hard to keep treatment open and accessible for those vulnerable and struggling and we will work hard to ensure this continues post-Covid.” Clusker is cautiously optimistic. “Once this passes, will people understand this group better, will it reduce the stigma of addiction if the public are now viewing the world through a different lens?”

    1. Judgment, decision making, and risk researchers have learned a great deal over the years about how people prepare for and react to global risks. In recent years, risk scholars have increasingly focused their energies on climate change, and as pandemic coronavirus has swept the globe many of these scholars are comparing the coronavirus pandemic with climate change to inform risk management. Risk communication research and the best practices developed from it are predicated on findings from the 1970’s to the present showing that there are structural similarities in how people think about widely divergent risks. Consequently, these lessons from risk communication of climate change (and from the canon of best practices) apply to the coronavirus pandemic. In the empirical comparison of student perceptions reported here, we replicate these structural similarity findings, but also find that moral concerns in particular deserve attention as a potentially distinct dimension of risk perception, on which different risks may also differ, as pandemic risks appear to evoke less moral concern than climate change. The need for communications to be timely, honest, credible, empathetic, and informative for useful individual actions is fundamental and essential for communicating effectively about the coronavirus epidemic. Some countries have heeded risk sciences, and are coping more successfully with pandemic coronavirus. Others have failed to implement these old lessons, which our data suggest still apply. While these failures may reinforce cynicism about political and public enthusiasm for accepting science, comparisons between the coronavirus pandemic and climate change may also foster greater aspirations for collective action.
    1. Father, there is a man, unlike any other, who comes down from the hills. He is the strongest in the world, he is like an immortal from heaven. He ranges over the hills with wild beasts and eats grass; the ranges through your land andcomes down to the wells. I am afraid and dare not go near him. He fills in the pits which I dig and tears up-my traps set for the game; he helps the beasts to escape and now they slip through my fingers.'His father opened his mouth and said to the trapper, ‘My son, in Uruk lives Gilgamesh; no one has ever pre-vailed against him, he is strong as a star from heaven. Go to Uruk, find Gilgamesh, extol the strength of this wild man. Ask him to give you a harlot, a wanton from the temple of love; return with her, and let her woman's power overpower this man. When next he comes down to drink at the wells she will be there, stripped naked; and when he sees her beckoning he will embrace her, and then the wild beasts will reject him.

      If we look at this altogether, we can begin to see an interesting form of "self" of the identity of this people. They see themselves as civilized. They see Enkidu as "other". They see this wild man and think he should be tamed by a "harlot" who will strip naked and lay with Enkidu so that he may too be civilized. It's interesting as well the language used her to describe this woman. This implies that without women, men would still be uncivilized, that they would be wild and roam the lands as wild beasts do. However, the author still translates to harlot. So, it is not merely the act of a woman, but the physical act that somehow tames the wildness within men, yet she does not gain any respect for keeping men civil.

      Zach Long CC BY-NC-ND

    1. Note: This rebuttal was posted by the corresponding author to Review Commons. Content has not been altered except for formatting.

      Learn more at Review Commons


      Reply to the reviewers

      We are grateful for the reviewers’ appreciation and comments. We have tried to address all concerns, and believe that those changes have greatly ameliorated the precision and presentation of our findings. All of our responses are in green in this document, and so are the changes in the manuscript and figure legends.

      Reviewer #1 (Evidence, reproducibility and clarity (Required)):

      In "An asymmetry in the frequency and position of mitosis in the epiblast precedes gastrulation and suggests a role for mitotic rounding in cell delamination during primitive streak epithelial-mesenchymal transition", Mathiah, Despin-Guitard and colleagues study divisions during mouse gastrulation. They perform ex vivo culture, live imaging and immunostaining to observe the frequency and position of mitosis within the embryo as well as the destiny of daughter cells after their divisions. The find that divisions on the posterior side of the embryo tend to be more basally located and could contribute to cell delamination into the mesodermal layer. Authors also affect antero-posterior signaling by genetically preventing the migration of the anterior visceral endoderm, which leads to mitosis away from the apical side of the epithelium on all lateral parts of the embryo.

      This study tackles a key developmental process which is poorly understood in mammals due to its concomitance with the implantation phase. Therefore, any carefully-made description of this process has the capacity to be eye-opening. This is potentially the case for this report, which provides nice images that most likely required skills and important efforts to obtain. The authors have written a clear manuscript with an interesting narrative. However, the quantifications are very poorly described, which makes it impossible for anyone to reproduce these results. I describe below a number of suggestions to clarify the quantifications, which is in my opinion a prerequisite to consider the conclusions from the authors.

      Fig1: Authors describe differences in the formation of rosettes between the anterior and posterior sides of the embryo. The microscopy images and movies provided are overlaid with drawings from the authors but without this visual help, I, and I assume other readers, see more rosettes than highlighted and fail to see some of the rosettes that are marked. To avoid this subjectivity, a clear methodology is required. In the methods, the authors state: "For quantification, rosettes were manually annotated and counted on Z sections located 5 μm from the basal side of the epiblast." And that is all. What defines a rosette? How many cells need to share a vertex to be considered as part of a rosette? How long do they need to persist not to be considered as occurring by chance? What about cells part of multiple rosettes? Does the rosette organization need to be apical, basal or all the way? Having those clearly defined criteria would be essential for anyone else to reproduce this quantification and would also offer a much more comprehensive description of the phenomenon and allow for more powerful conclusions.

      A rosette is defined as a multicellular transient structure composed of at least 5 cells converging to a central vertex. Practically, a region of interest where cell contours are in focus is determined on the Z-section located 5 mm from the epiblast basal side, which is easily identified as the epithelial architecture changes radically when one enters the visceral endoderm. Only rosettes that are visible throughout the epiblast layer, from the basal to the apical side, are counted. To ensure this, manual segmentation of all cells (for all Z plan acquired, from the basal plane to the apical side) contributing to a rosette was performed for lightsheet imaging. This is illustrated in Video 2. For confocal imaging, segmentation was annotated only at the basal plane, but visual verification that the rosette structure is persistent throughout the layer was performed. One cell could be part of several rosettes, and rosette events were counted even when visible only on one timeframe, but this was consistent for all embryo sides. Due to the time resolution of confocal imaging, rosettes could not be followed overtime. However, the time resolution of lightsheet imaging allowed observing rosettes lifespan and resolution. The protocol for image analysis has been better detailed in the results (lines 162-163) and the methods section of the revised version of the manuscript (lines 452-467, copied hereunder).

      "Rosettes: For lightsheet imaging, embryos were dissected at E5.75. Images were acquired for 10 to 12 hours. Quantification focused on the first 20 to 30 frames (around 3 hours) to capture pregastrulation events and reduce the risk of bias from imaging. The rest of the frames showed that the embryo continued growing for several hours. Z-stacks from 4 sides were fused using Zeiss plugin for lightsheet Imaging. Images were then processed using Arivis Vision4D v2.12.3 (Arivis, Germany). Embryo contours were segmented manually on each Z-slice and time point, in order to adjust for embryo rotation manually if necessary. For each side of the embryo, Z stack was cropped to an average of 30 Z slices, from the basal side (5 microns from VE layer, which can be morphologically distinguished due to cell shape and membrane Tomato distribution) to the cavity, marking the apical side. Rosettes were identified and counted on Z sections located 5 µm from the basal side of the epiblast. Practically, vertices were systematically scanned to find those in which 5 cells or more met. Cells contributing to a rosette were then manually segmented on each Z-slice and time point by highlighting cellular membranes using Wacom’s Cintiq 13HD, to create a 3D reconstruction. For confocal imaging, rosettes were identified using the same method, and counted on Z sections located 5 µm from the basal side of the epiblast after visual verification that it was present throughout the Z-stack. For both techniques, presence of associated apical rounding was assessed for each vertex. Cells could contribute to several rosettes."

      In addition, the data are given as "rosette/frame" and as "rosette/mm2". What is the point of giving both data, which are essentially the same? The frame is irrelevant. It would be more interesting to know how many cells there are in this area, as cell packing could be a determinant of rosette formation. "Rosette/mm2/min" is very confusing. It should state "rosette.mm-2.min-1" or "rosette/mm2.min".

      Following this comment, we indeed chose to get rid of the data expressed as “rosette/frame”. Cells were counted in the area of the epiblast in focus to present data as number of rosettes normalized by the number of cells in the region of interest for both lightsheet and confocal microscopy data (described in results section lines 140 and 164). These measurements led to a similar conclusion, confirming that rosettes are more frequent in posterior epiblast. Difference in cell packing was indeed essential to rule out. We estimated cell packing as the ratio of cell number to surface area, and found it to be similar in posterior, anterior, and lateral sides of embryos at a given stage, which indicates that cell packing is not a determinant for difference in rosette frequency in this context. We discussed packing in the Results section (lines 169-173, copied hereunder).

      "The cell number per surface area was similar on all sides, which indicates that the higher number of rosettes was not due to increased cell packing. Rosettes have also been identified in the chick PS (Wagstaff, Bellett, Mogensen, & Münsterberg, 2008), where they were proposed to facilitate ingression during gastrulation."

      We modified the legend to use "rosette/mm2.min”.

      On a conclusive note, I fail to understand how relevant the formation of rosettes would be. The authors should clarify this point.

      Epithelial rosettes have been observed as common intermediates in numerous morphogenesis events. In particular cases, such as Drosophila germ band extension, or zebrafish lateral line development, the mechanisms of formation (planar cell polarity (PCP) and apical constriction, respectively) and resolution have been very well described. In the mouse embryo, anterior visceral endoderm (AVE) migration has been linked to PCP signaling-dependent rosette formation (Trichas 2012). In primitive streak (PS) formation, rosettes with actin-rich centers were described in the chick PS and found to be Nodal dependent (Wagstaff 2008 and Yaganawa 2011). Their mode of formation or resolution is currently unknown. Our observations confirm the findings in chick and highlight the presence of rosettes at an earlier stage, before PS can be identified. Interestingly, rosettes are enriched on the posterior side at the same time when Nodal signaling becomes asymmetric, leading to posterior restriction of basal membrane perforations (Kyprianou 2020). To progress towards understanding rosettes’ significance in the mouse gastrulation context, it would be interesting to study whether the distribution of rosettes is homogenous before anterior-posterior axis specification. Additionally, it would be important to assess whether random epiblast cells delaminate before PS formation, as observed in chick (Voiculescu 2014). We could not attempt those experiments so far, as we perform most experiments by two-photon microscopy, by which only one embryo side can be recorded at a time, and have no way to distinguish embryo orientation before AVE migration. A better understanding of rosette mode of formation and resolution, including the role of Nodal, would also be necessary to assess the importance of our observations. The technical evolution in mouse embryo imaging will probably permit solving those questions in the near future, through prolonged imaging with tracking of every cell fate (McDole 2018). We have tried to improve the discussion (lines 314-326, copied hereunder), and acknowledge the limitations of our findings to a description of a phenomenon without proven significance at this stage.

      "However, since we observed a marked imbalance in rosette frequency as soon as the anterior-posterior axis was specified, it is possible that rosettes reflect increased epithelium fluidity in posterior epiblast, which is exposed to a distinct mechanical context, at the very beginning of PS morphogenesis. Indeed, a posterior shift in the distribution of basement membrane perforations was identified just after AVE migration, due to an asymmetry in Nodal signaling dependent metalloproteinase activity (Kyprianou et al., 2020). To progress towards understanding rosette formation significance in this context, it would be interesting to study whether the distribution of rosettes is homogenous before anterior-posterior axis specification, and to assess whether random epiblast cells delaminate before PS formation, as observed in chick (Voiculescu, Bodenstein, Lau, & Stern, 2014). As Nodal plays a major role in PS initiation, the presence and distribution of rosettes should be studied in models in which Nodal signaling can be tuned (Kumar, Lualdi, Lewandoski, & Kuehn, 2008)."

      Fig2: I have essentially the same issue for bottle cells and delamination counting as for rosettes. In this case, there is nothing in the methods section.

      We have added a paragraph to describe the mosaic analysis in the Methods section (lines 472-488):

      "Mosaic: Embryos were recorded in a lateral position. As the proportion of GFP positive cells varied between mosaic embryos, normalisation was performed by dividing by the number of green cells in a given embryo. Anterior and posterior halves were defined by drawing a line perpendicular to the embryonic/extraembryonic boundary and passing through the distal tip. Bottle-shaped cells were identified as having a thin attachment on the apical surface (less than a third of the larger section), and the majority of the cell body located in the basal side. Quantification was performed both on the 3D rendering, and through navigating through the Z-stack. The same criteria where used on all sides of the embryo, and quantification was verified by two independent investigators. Delamination was defined as retraction of the apical process, and displacement of the cell body in the mesoderm layer, which could be identified because of the ubiquitous membrane Tomato labelling. Cell division was characterized by cell rounding followed by the appearance of daughter cells. Cell dispersion after mitosis was defined as absence of basolateral contact between daughter cells, which implies presence of at least one epiblast cell (more often 2 or 3) between daughter cells. Mitosis was considered “non-apical” when happening at least 10 µm away from the apical pole, hence not in the first pseudo-layer of nuclei lining the apical pole."

      What defines a cell as bottle shape and not bottle shape (apical vs basal width for example)?

      Bottle-shaped cells were visually identified as having a thin attachment on the apical surface (less than a third of the larger section), and the majority of the cell body located in the basal side. Quantification was performed both on the 3D rendering, and by navigating through the Z-stack. Due to the large variation in shape, no systematic measurement was performed. However, the same criteria were used on all sides of the embryo, and quantification was verified by two independent investigators. As proposed by Reviewer 2, those criteria would include scutoids with smaller apical surface, which explains why we observe bottle-shaped cells both on the anterior and posterior sides. In addition to Methods, we included a better description of the methodology in the Results (lines 196-200).

      "The quantification of bottle-shaped cells was performed in 3D and through Z-stack navigation and included all cells with an apical section smaller than a third of the basal section. Some cells had a round basal cell body and a thin apical extension while others resembled the recently described scutoids performing apico-basal transitions (Gómez-Gálvez et al., 2018)."

      Where does a cell need to be to be counted as delaminated (a distance needs to be stated, absolute (better) or relative)?

      Delamination is defined as retraction of the apical process, and displacement of the cell body in the mesoderm layer. Using the ubiquitous membrane tomato marker we could easily distinguish the epiblast, mesoderm and visceral endoderm layers, notably through cell packing, morphology and arrangement. This was described in Results (lines 200-204).

      "Asymmetrical cells were present on both sides, but more frequent on the posterior side, and cell delamination (retraction of the apical process and cell body shift in the mesoderm layer) only took place on the posterior side. Cells maintained an apical attachment until their basally located cell body had begun crossing the PS/mesoderm border, and only fully detached after delamination."

      What defines sister cells as dispersing after division? How far apart do they have to be? After how much time? From the movies provided, the acquisition time seems to short to assess cell dispersal.

      Cell dispersion after mitosis was defined as absence of basolateral contact between daughter cells as they extend towards the basal side, which implies intercalation of at least one epiblast cell (more often 2 or 3) between daughter cells. After cytokinesis was completed, extension and separation of daughter cells was visible in the next time point (after 25 min). The time resolution was thus sufficient to note that daughter cells were not adjacent, which is consistent with other studies (Abe 2018).

      We have modified the Methods (copied above) and the Results section of the revised version of the manuscript (lines 213-217).

      "Upon elongation of daughter cells to reach the basal pole of the epiblast, the majority displayed no basolateral connection between each other and were instead separated by intercalating epiblast cells, which would be expected to result in daughter cells dispersion over time, as described in (Abe, Kutsuna, Kiyonari, Furuta, & Fujimori, 2018)."

      Fig3: Mitotic index calculation is described in the figure legend but not in the methods section. It should also be in the methods section and made explicit that the number of mitotic cells is normalized to green cells only, not the entire cell population. The mitotic index seems higher in this population than in the entire embryo as seen in Fig4.

      The mitotic index (MI) was indeed calculated differently so numbers cannot be directly compared. MI identified for anterior and posterior epiblast is not statistically different from the ones found in Figure 4 for E7 embryos. In mosaic embryos, we do not have a way to delimitate the PS. In Figure 4, measurements of MI in the PS (delimitated by the area where the basement membrane is degraded) include cells that are destined to delaminate as wells as those that won't. In the mosaic embryos, MI is measured in cells that delaminate only, and is indeed higher. This represents a small population, which likely explains why it does not reach statistical significance and manifests as a trend.

      We have fixed the Methods (see above) and Results (lines 222-228) sections.

      "For systematic quantification, epiblast regions were defined as anterior or posterior by tracing a line passing by the distal pole and perpendicular to the embryonic/extraembryonic border, and GFP positive cells undergoing rounding were followed overtime (Fig. 3a-c). Although the frequency of cell division (normalized to the total number of GFP positive cells) was similar in anterior and posterior epiblast, there was a trend towards a higher division rate specifically in cells undergoing delamination to become mesoderm (Fig. 3d)."

      What defines an exiting cell**?

      An exiting cell is characterized by morphological remodelling, apical retraction, as well as the position of the cell body across the mesoderm/epiblast border visualized by the precise membrane Tomato labelling. It is now described in Methods and in Results (lines 201-204: " cell delamination (retraction of the apical process and cell body shift in the mesoderm layer) only took place on the posterior side. Cells maintained an apical attachment until their basally located cell body had begun crossing the PS/mesoderm border, and only fully detached after delamination".

      Regarding the non-apical rounding, why not calling it basal rounding? How far from the apical side does a cell need to be counted as non-apical?

      The reason for that denomination is that these so-called “non-apical mitoses” are not strictly basal either. Indeed, mitosis is considered “non-apical” when happening at least 10µm away from the apical pole, meaning that these mitoses do not occur within the first pseudo-layer of nuclei lining the apical pole. This is described in Methods.

      In the panel h, with the posterior division outcome, is that for all divisions or only for non-apical divisions?

      The panel (Fig. 3g, there was an error in figure labelling in the previous version) has been modified to better precise cell outcomes. It represents all posterior divisions, and quantifies the outcome according to the position of mitosis along the apical-basal axis of the cell. See Results, line 230-232: "Non-apical mitosis in the posterior epiblast was preferentially associated with EMT, as it resulted in formation of one or two mesoderm cells (Fig. 3g)."

      Do basal divisions give rise to more epi?

      No, non-apical divisions mainly give rise to mesoderm cells. Indeed, approximatively 66% of basal divisions give rise to two mesoderm cells, and 33% to an epiblast and a mesenchymal cell (Figure 3g). We never observed a non-apical division resulting in two epiblast cells.

      Is epi or meso fate only determined by location in a different layer or are fate markers used?

      Epiblast or mesenchymal fate was determined by both morphological and localization criteria. Epiblast cells have an apical and a basal pole. Mesoderm cells have no apical process, and display initiation of front-rear polarity often defined by the presence of nascent migration appendix. As stated before, membrane Tomato labelling allows exact distinction of germ layers.

      What happens to the non-apical mitosis on the anterior side?

      On the anterior side, the very few anterior non-apical mitoses only give epiblast cells (not shown).

      Fig4: Methods state "For Phospho-histone H3 quantifications, sections were chosen at least 10 μm apart to ensure that each cell was only counted once, and counting was performed using the Icy software" and legend states "The PS region is defined by the area where the basal membrane (yellow) is degraded, and the posterior region quantification excludes counts from the PS region".

      What about cells at the boundary between PS and non-PS regions? This needs to be extended and brought together in the methods section. **Also, the tissue architecture in the PS is not as well defined as in the rest of the tissue.

      A cell was counted as being part of the PS region if at least 50% of its cell body (visual measuring) was within the area where the basal membrane is non-ambiguously degraded, and if the cell retained its attachment to the apical pole (cell contours were determined by F-actin detection using Phalloidin). The Methods section has been completed in the revised version of the manuscript (lines 490-501).

      "Phospho-histone H3: For Phh3 quantifications, sections were chosen at least 10 mm apart to ensure that each cell was only counted once, and counting was performed using the Icy software (http://icy.bioimageanalysis.org). For sagittal sections, anterior and posterior regions were defined by drawing a line perpendicular to the embryonic/extraembryonic boundary and passing through the distal tip. For transverse sections, anterior-posterior boundary was placed at mid-distance between the anterior and posterior poles. The PS region was defined by the area where the basement membrane was degraded, and the posterior region quantification excluded counts from the PS region. A cell was counted as being part of the PS region if at least 50% of its cell body was within the area where the basement membrane was non-ambiguously degraded, and if the cell retained its attachment to the apical pole (cell contours were defined by F-actin detection using Phalloidin)."

      Is the epithelial polarity clear enough to be determined without AB marker in the PS?

      We considered that a cell retained its AB polarity if the cell extended to both apical and basal pole. Even if the pseudostratified epithelium architecture is complex, most cell contours could be delimited when navigating through the Z-stack.

      Finally, the number of cells counted is missing. This has been fixed in the Figure legend.

      Supp Fig5: based on available images of the Rac1KO embryo, I am not sure that epithelial architecture is established well enough to assess the location of mitosis along the apico-basal axis.

      Indeed, the architecture of the Rac1 KO mutants is vastly altered. As a consequence, only a small number of Rac1 mutants in which we could delimitate the germ layers were analysed, and only the cells we could unambiguously locate were considered. The Rac1 VE-deleted phenotype, on the other hand, was not severe enough as there is only a partial AVE migration defect in most mutants (Migeotte et al., 2010). This is why we confirmed the data on AVE migration defective embryos by using the RhoA VE-deleted mutant, which has a strong AVE migration defect but retains good tissue architecture. We tried to increase figure clarity by annotating the embryo cavity as well as the embryonic/extraembryonic boundary. We also submit a less compressed version of the figures, which we hope will facilitate image analysis.

      Reviewer #1 (Significance (Required)):

      Although I am not as familiar with mouse gastrulation as I would like to be, I am familiar with gastrulation, live imaging and analysis. At this point, I find it difficult to discuss the conclusions of the study since the methodology is so unclear. Nevertheless, any carefully-made description of mammalian gastrulation has the capacity to be eye-opening. This is potentially the case for this report, which provides nice images that most likely required skills and important efforts to obtain.

      We hope the changes we made help better understanding the methodology, and thank Reviewer 1 for positive comments and the help in identifying the points we had failed to properly describe.

      Reviewer #2 (Evidence, reproducibility and clarity (Required)):

      This manuscript, from Mathiah and colleagues, describes an in-depth analysis of differences in cell organization and division within the epiblast of the very early mouse embryo, and in particular, with the onset of gastrulation. Their data indicate a difference in the organization of cells between the anterior/lateral and posterior regions of the epiblast even before gastrulation has commenced, as well as differences in the location of mitoses relative to the apical and basal ends of the cells. The data provide new insight into the early regionalization of the epiblast. However, the authors should include reference to, and discussion of, the paper by Michael Snow on growth and regionalization of the epiblast (Snow MHL (1977) Gastrulation in the mouse: Growth and regionalization of the epiblast. J. Emb. Exp. Morph. 42: 293-303), where he did a much more fine-grained analysis of mitotic index across the entire epiblast, defining a proliferative zone in the anterior part of the primitive streak where the mitotic index was higher between E6.5 and E7.5. He also describes non-apical mitoses specifically in the primitive streak region as compared to all other regions of the epiblast. The results of the present study dovetail nicely with the results presented by Snow.

      This was indeed a major oversight, and we apologize for it. The work of Snow identifies very nicely a proliferative zone in the anterior part of the PS. We did not comment on that as our study focuses on posterior PS. We included the reference in the revised version of the manuscript, and pointed the fact that he first described non-apical mitosis in the PS (lines 233-236).

      "Remarkably, this concurs with the observation by Snow (Snow, 1977) that in the PS of E6.5 and E7 embryos, mitosis could be found at all levels of the tissue, including adjacent to the endoderm, while it was located at the apical surface of the pseudostratified tissue everywhere else."

      Overall, this is a very nice study, but some revisions would help with clarity at certain points. The data on rosette formation are interesting, but it is not clear what an increase in rosettes in the posterior region means. The authors contend (lines 169-170) that this represents a dynamic epithelium primed for EMT, but it is not clear how rosettes facilitate or promote EMT, and especially why that would be seen at E5.75 before EMT has begun. An alternative interpretation might be that the shape of the epithelium may be changing and the packing of the epithelial cells has to change to accommodate this. We do know that the overall shape of the embryo changes from elongate medial-lateral to elongate anterior-posterior just as EMT is initiated (Perea-Gomez et al., (2004) Current Biology 14: 197-207) and it may be that changes in cell packing are required to accommodate this. The authors may want to consider whether the rosettes that they observed represent scutoids (Gomez-Galvez et al., (2018) Scutoids are a geometrical solution to three-dimensional packing of epithelia. Nat. Commun. 9:2960). An analysis of the 3-dimensional organization of the cells within the rosettes (i.e. at all Z levels) may shed some light on this.

      Following on the comments by Reviewer 1 and 2, we quantified cell packing, and found it to be identical on all sides at a given stage. We have added a better description of rosette quantification (lines 169-172 and lines 452-470), a video showing 3D reconstruction of cells in a rosette (Video 2), and an extended discussion (lines 314-326) in the revised version of the manuscript. Some cells within the epiblast are indeed likely to be shaped as scutoids, some with an apical-basal asymmetry (lines 196-202). The reference was added to the manuscript (line 200).

      Figure 2b,c and Figure 3a, a', b, c - Addition of dotted lines to indicate the apical and basal ends of the epiblast would be helpful in orienting the reader**.

      We have added lines to indicate apical and basal ends of the epiblast.

      Figure 2c' - what these graphs represent exactly is somewhat vague, and the figure legend is also very vague. In particular, the third graph on cell dispersion is not clear. Does this mean that the daughter cells are separated from one another following division? Or that they are in different compartments (epiblast/mesoderm) after division? A better description should be included in the figure legend.

      Following on the comments by Reviewer 1 and 2, we have added a better description of cell dispersion in the Results (lines 213-217), Methods (lines 483-486) and figure legend.

      Figure 3g would appear to show the proportion of the total number of posterior divisions that give rise to particular combinations of daughter cells (epi/epi, epi/meso, meso/meso). However, the discussion of this graph in the text (lines 215-219) suggests that it demonstrates that non-apical mitoses always result in meso/meso and epi/meso daughter cells, which it does not. That analysis would be very interesting to add to Figure 3, with the daughter cell types broken down into those coming from apical mitoses and those coming from basal mitoses.

      The analysis was broken down as suggested, and has been added to the revised version of the manuscript (line 352, Figure 3g).

      In Figure 4, it is not clear how anterior and posterior are defined, and what criteria were used to distinguish posterior from primitive streak. This is nicely demonstrated in Supplementary Figure 3 - maybe panels A and B could be included in Figure 4 to improve the clarity of the analysi**s.

      We have better described the quantification methodology in the Methods section (lines 490-501), moved panel a from Supplementary Figure 3 to Figure 4a as suggested, and added an explanatory drawing (Figure 4b) to the revised version of the manuscript.

      The data on mitotic index in Figures 2, 4, and 5 do not appear to be consistent. The mitotic index for E7.25 in Figure 2e is similar between anterior and posterior, even though the posterior includes the primitive streak, while the mitotic index presented for the three stages in Figure 4b would imply that the mitotic index for the entire posterior region should be higher than the anterior at all three stages. Similarly, in Figure 5a' and b', the mitotic index in anterior and posterior regions of E5.75 and E6.25 embryos are not significantly different despite the primitive streak being included in the posterior count, while the data presented in Figure 4 would imply that the entire posterior region including primitive streak should be much higher than the anterior. The authors should clarify this in the Results.

      In Figure 2 and 3, the mitotic index (MI) is calculated as number of cell division among GFP+ cells divided by the total number of GFP+ cells, while in Figure 4 and 5 it is quantified as Phospho-histone H3+ cells per total number of cells (DAPI). We have clarified this in the revised graphs and legends of the novel version of the manuscript. Those numbers cannot be directly compared. Nonetheless, we found no statistical difference between the MI shown in Figure 3d, and the MI shown in Figure 4c third row (E7). In sagittal view, the PS area cannot be delimited, so we compared anterior and posterior regions, with the PS included in the posterior region, and saw no difference in MI. In transverse section, there was no MI difference when comparing anterior and posterior embryo halves. However, when we refined the analysis by defining the PS as the area where the basal membrane was degraded, a higher MI emerged specifically in the PS compared to anterior and posterior (not including PS) regions. This difference was thus lost by dilution when the PS area was included in the posterior region. We have also stated this distinction more clearly in the revised version of the manuscript (lines 269-271).

      The data on non-apical mitoses in the RhoA-VE deleted (Figure 6) and Rac1ko embryos (Supplemental figure5) are not particularly compelling. It is hard to see the basal mitoses in the new AVE-opposed regions in the mutant embryos in the images presented. Perhaps the graphs in these two figures could have the AVE-opposed data broken down into two groups - the region that is posterior and the region that is anterior but not adjacent to AVE. Better images would improve the clarity of these data as well.

      As explained in response to Reviewer 1, we have attempted to clarify the anatomy through annotation, and provide less compressed images. We agree that the embryos are altered. Nonetheless, especially in RhoA-VE deleted, the germ layers could be distinguished and non-apical mitosis identified through combining 3D analysis and navigation through the Z-stack. We honestly admit those are the best images we could get, and we believe that they allow to make the point that non-apical mitosis are only found in the area further away from the AVE.

      Reviewer #2 (Significance (Required)):

      The data on differential proliferation and apical vs. basal mitoses are complementary to data already published, but the present study updates the existing data by the addition of live imaging and 3-dimensional reconstruction of cell shapes, providing a more complete insight into the process. The observation that rosettes are detectable at the basal ends of the epiblast, and more so in the posterior, is novel, but the significance for embryonic development is not well rationalized.

      These data are of interest to those investigating the mechanisms of early morphogenesis, as well as those interested in the cellular correlates of molecular regionalization that results from the well-described signaling pathways regulating axis specification.

      My background is in early mouse embryo morphogenesis, therefore I feel that I have sufficient expertise to evaluate the data presented.

      We thank Reviewer 2 for positive comments, and are grateful for the constructive criticism and important references.

      \*Referees Cross Commenting***

      I agree with Reviewer 1 on the lack of detail about the methods - my comments stemmed from the same confusion about how measurements were made, but Reviewer 1 more articulately addressed the key points. I agree with Reviewer 3 on the quality of the videos. It is very difficult to see how they could follow cells with a 20 minute interval.

      I would like to address the comment by Reviewer 3 on the use of agarose in the imaging experiments. The methods section states that agarose was used to make the culture "chambers" used for light-sheet imaging, which was not the major approach used for imaging in this study. Only the data in Figure 1A came from those experiments, and it was validated by confocal data in 1B,C where the embryos were cultured in Ibidi chambers with culture medium and no agarose present. So I don't think agarose effects on embryo development are a major worry. Also, this same approach was used by Ryan Udan in Mary Dickinson's lab to visualize yolk sac vasculogenesis, and it did not appear to have a deleterious effect on development in that case, although the embryos imaged here were much earlier and are definitely differentially sensitive to culture conditions from those cultured at E8.

      Reviewer #3 (Evidence, reproducibility and clarity (Required)):

      This is an essentially descriptive study, looking at primitive streak formation and cell ingression from the epiblast in the mouse from about E5.75 to E7.5 or so using time-lapse microscopy (light sheet and confocal) of cultured embryos. The study also takes advantage of some genetically encoded reporters, some of them inducible by tamoxifen, which allow following cells, closer examination of their shapes, and in some cases unambiguous orientation of the embryos based on expression of the reporter. Overall the study is well designed.

      I have two very major concerns about this paper - first, the culture system used in most experiments uses agarose, which has been found in several labs to affect normal cell movements and other cell behaviours. It is essential to determine that embryos cultured in these conditions develop normally for much longer than the period of imaging to ensure that the findings are relevant to normal development rather than an artefact. This is particularly important because mouse embryos develop rather poorly at peri-implantation stages with any culture method, and this one could make matters even worse.

      Embryos were mounted into an agarose cylinder in which a tunnel had been created with a 150 microns wide copper wire. Embryos were mounted vertically, with the cone oriented on the bottom, to avoid restriction of growth at distal tip of the embryo. As embryos had a smaller diameter than the tunnel, they could comfortably grow without being restricted (Methods, lines 413-414). Although embryos could not been recovered after the long imaging period (12h), embryos similarly mounted in the agarose cylinder but not imaged were kept in culture, and showed normal growth compared to a free-floating embryo (Methods, lines 431-433). In addition, we focused on the first hours of imaging to reduce the risk of phototoxicity-induced anomalies (Methods, lines 453-455). Moreover, although we identified the asymmetry in rosette abundance through lightsheet imaging, we confirmed the finding through confocal imaging of free-floating embryos, and found similar results (Results lines 153-167, Figure 1b and c, Video 3).

      While it has been reported that agarose can affect the development of chick embryos in culture, agarose has been a widely used culture matrix for live imaging particularly for lightsheet imaging in other organisms including drosophila, zebrafish, and mouse. We thank Reviewer 2 (in cross-comments) for highlighting that in Udan et al., (2014), a report from Mary Dickinson’s lab, embryos are cultured in agarose “chambers” for lightsheet. Although some of the experiments in Udan et al., (2014) are performed at E8, this paper also focuses on pre-gastrulation mouse embryos as they culture E6.5 embryos for 24 hours, image from 5 view angles, analyze 572 z-slices representing half of the embryo (Fig5 and Fig6 Udan et al., 2014) and show no adverse effects.

      The second concern is that for a paper that is almost entirely about time-lapse microscopy observations of live embryos, the movies are very poor. Although the images are generally good and the 3-d sequences/images from the light-sheet microscope sequences are quite impressive (and have good spatial resolution), the time resolution is extremely poor and the movies very short. It is largely impossible to follow cell behaviours or movements in these sequences.

      Indeed, the time lapse between time points as well as the total duration of the acquisition is limited, especially when embryos are imaged by confocal microscopy. These measures were taken mainly to preserve the integrity of the embryo and thus ensure that growth conditions were the closest to optimal in vivo conditions. For rosette analysis, the 20 minutes interval was too long to follow rosette resolution, as stated in the manuscript. For mosaically labelled embryos, we quantified only the cells for which the fate and/or progeny could be identified without ambiguity, which was made easier as we chose a 4OH-tamoxifen posology that resulted in a low proportion of labelling. As both cell delamination and mitosis are relatively slow processes, this time resolution proved sufficient. Time resolution for lightsheet was 7 minutes, which is similar compared to other works on mouse gastrulation (such as Williams et al., 2012), and actually higher than most two-photon or confocal studies, including that of our previous reports (Migeotte et al., 2010, Saykali et al., 2019, Trichas et al., 2012) in which cell tracking could be efficiently performed. This high time resolution allowed following individual rosettes overtime (Sup. Fig.2c).

      Reviewer #3 (Significance (Required))

      The study focuses on cell shape changes and various processes that accompany ingression and reports that ingression may occur through a variety of different mechanisms that occur at the same time, including rosette formation, individual ingression of bottle-shaped cells, and larger population ingression events. This is very similar to what has been described in chick embryos (eg. Voiculescu et al. eLife 2014 - surprisingly this is not cited), although in rodents primitive streak formation occurs in the absence of large-scale movements of cell sheets. Basically there are no surprises in the findings either for mouse or in comparison with other species, but the study is OK in terms of contributing useful information about streak formation and function in mouse (if the above problems are fixed).

      We thank Reviewer 3 for helpful comments and references. We respectfully disagree concerning the risk of bias due to agarose cylinder culture, as exposed above. Concerning the videos, we have provided less compressed videos to retain as much image quality as possible. Although it would evidently be better to have a higher time resolution and longer movies, we believe it is not a limitation for the events we study and describe as they can be reliably followed with the time resolution and observation length we provide. The reference to Voiculescu et al., 2014 is indeed important, we have added it to the revised version of the manuscript (line 324) and apologize for the oversight.

      \*Referees Cross Commenting***

      In response to reviewer 2: One issue with this is that one does not know whether there is a "deleterious" effect of the agarose on movements until one is sure that (a) one understands what the movements would look like without agarose and that there are no differences, and (b) (a serious shortcoming here) that embryos need to be shown to develop completely normally in those culture conditions WAY beyond the period of imaging. There are lots of observations by several labs (some unpublished of course, but some are published) suggesting that agar and agarose do interfere with cell movements. In chick for example the Chapman and Schoenwolf method where embryos are placed on agarose, there are always head defects due to impaired movements and the agarose interfering with tissue tensile forces.

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      Referee #2

      Evidence, reproducibility and clarity

      This manuscript, from Mathiah and colleagues, describes an in-depth analysis of differences in cell organization and division within the epiblast of the very early mouse embryo, and in particular, with the onset of gastrulation. Their data indicate a difference in the organization of cells between the anterior/lateral and posterior regions of the epiblast even before gastrulation has commenced, as well as differences in the location of mitoses relative to the apical and basal ends of the cells. The data provide new insight into the early regionalization of the epiblast. However, the authors should include reference to, and discussion of, the paper by Michael Snow on growth and regionalization of the epiblast (Snow MHL (1977) Gastrulation in the mouse: Growth and regionalization of the epiblast. J. Emb. Exp. Morph. 42: 293-303), where he did a much more fine-grained analysis of mitotic index across the entire epiblast, defining a proliferative zone in the anterior part of the primitive streak where the mitotic index was higher between E6.5 and E7.5. He also describes non-apical mitoses specifically in the primitive streak region as compared to all other regions of the epiblast. The results of the present study dovetail nicely with the results presented by Snow.

      Overall, this is a very nice study, but some revisions would help with clarity at certain points. The data on rosette formation are interesting, but it is not clear what an increase in rosettes in the posterior region means. The authors contend (lines 169-170) that this represents a dynamic epithelium primed for EMT, but it is not clear how rosettes facilitate or promote EMT, and especially why that would be seen at E5.75 before EMT has begun. An alternative interpretation might be that the shape of the epithelium may be changing and the packing of the epithelial cells has to change to accommodate this. We do know that the overall shape of the embryo changes from elongate medial-lateral to elongate anterior-posterior just as EMT is initiated (Perea-Gomez et al., (2004) Current Biology 14: 197-207) and it may be that changes in cell packing are required to accommodate this. The authors may want to consider whether the rosettes that they observed represent scutoids (Gomez-Galvez et al., (2018) Scutoids are a geometrical solution to three-dimensional packing of epithelia. Nat. Commun. 9:2960). An analysis of the 3-dimensional organization of the cells within the rosettes (i.e. at all Z levels) may shed some light on this.

      Figure 2b,c and Figure 3a, a', b,c - Addition of dotted lines to indicate the apical and basal ends of the epiblast would be helpful in orienting the reader.

      Figure 2c' - what these graphs represent exactly is somewhat vague, and the figure legend is also very vague. In particular, the third graph on cell dispersion is not clear. Does this mean that the daughter cells are separated from one another following division? Or that they are in different compartments (epiblast/mesoderm) after division? A better description should be included in the figure legend.

      Figure 3g would appear to show the proportion of the total number of posterior divisions that give rise to particular combinations of daughter cells (epi/epi, epi/meso, meso/meso). However, the discussion of this graph in the text (lines 215-219) suggests that it demonstrates that non-apical mitoses always result in meso/meso and epi/meso daughter cells, which it does not. That analysis would be very interesting to add to Figure 3, with the daughter cell types broken down into those coming from apical mitoses and those coming from basal mitoses.

      In Figure 4, it is not clear how anterior and posterior are defined, and what criteria were used to distinguish posterior from primitive streak. This is nicely demonstrated in Supplementary Figure 3 - maybe panels A and B could be included in Figure 4 to improve the clarity of the analysis.

      The data on mitotic index in Figures 2, 4, and 5 do not appear to be consistent. The mitotic index for E7.25 in Figure 2e is similar between anterior and posterior, even though the posterior includes the primitive streak, while the mitotic index presented for the three stages in Figure 4b would imply that the mitotic index for the entire posterior region should be higher than the anterior at all three stages. Similarly, in Figure 5a' and b', the mitotic index in anterior and posterior regions of E5.75 and E6.25 embryos are not significantly different despite the primitive streak being included in the posterior count, while the data presented in Figure 4 would imply that the entire posterior region including primitive streak should be much higher than the anterior. The authors should clarify this in the Results.

      The data on non-apical mitoses in the RhoA-VEdeleted (Figure 6) and Rac1ko embryos (Supplemental figure5) are not particularly compelling. It is hard to see the basal mitoses in the new AVE-opposed regions in the mutant embryos in the images presented. Perhaps the graphs in these two figures could have the AVE-opposed data broken down into two groups - the region that is posterior and the region that is anterior but not adjacent to AVE. Better images would improve the clarity of these data as well.

      Significance

      The data on differential proliferation and apical vs. basal mitoses are complementary to data already published, but the present study updates the existing data by the addition of live imaging and 3-dimensional reconstruction of cell shapes, providing a more complete insight into the process. The observation that rosettes are detectable at the basal ends of the epiblast, and moreso in the posterior, is novel, but the significance for embryonic development is not well rationalized.

      These data are of interest to those investigating the mechanisms of early morphogenesis, as well as those interested in the cellular correlates of molecular regionalization that results from the well-described signaling pathways regulating axis specification.

      My background is in early mouse embryo morphogenesis, therefore I feel that I have sufficient expertise to evaluate the data presented.

      Referees Cross Commenting

      I agree with Reviewer 1 on the lack of detail about the methods - my comments stemmed from the same confusion about how measurements were made, but Reviewer 1 more articulately addressed the key points.

      I agree with Reviewer 3 on the quality of the videos. It is very difficult to see how they could follow cells with a 20 minute interval.

      I would like to address the comment by Reviewer 3 on the use of agarose in the imaging experiments. The methods section states that agarose was used to make the culture "chambers" used for light-sheet imaging, which was not the major approach used for imaging in this study. Only the data in Figure 1A came from those experiments, and it was validated by confocal data in 1B,C where the embryos were cultured in Ibidi chambers with culture medium and no agarose present. So I don't think agarose effects on embryo development are a major worry. Also, this same approach was used by Ryan Udan in Mary Dickinson's lab to visualize yolk sac vasculogenesis, and it did not appear to have a deleterious effect on development in that case, although the embryos imaged here were much earlier and are definitely differentially sensitive to culture conditions from those cultured at E8.

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      Reply to the reviewers

      Reviewer #1 (Evidence, reproducibility and clarity (Required)): \*Summary:** Reproducibility of genetic interactions across studies is low. The authors identify reproducible genetic interactions and ask the question of what are properties of robust genetic interactions. They find that 1. oncogene addiction tends to be more robust than synthetic lethality and 2. genetic interactions among physically interacting proteins tend to be more robust. They then use protein-protein interactions (PPIs) to guide the detection of genetic interactions involving passenger gene alterations. **Major comments:** The claims of the manuscript are clear and well supported by computational analyses. My only concern is the influence of (study) bias on the observed enrichment of physical protein interactions among genetic interactions. 1. Due to higher statistical power the here described approach favors genetic interactions involving frequently altered cancer genes (as acknowledged by the authors). 2. Also some of the libraries in the genetic screens might be biased towards better characterized screens. 3. PPI networks are highly biased towards well studied proteins (in which well studied proteins - in particular cancer-related proteins - are more likely to interact). The following tests would help to clarify if and to which extend these biases contribute to the described observations:*

      Our response: We thank the reviewer for the positive assessment of our manuscript and have addressed the issue of study bias in response to the specific queries below.

      * 1 . The authors should demonstrate that the PPI enrichment in reproducible vs non-reproducible genetic interactions is not solely due to the biased nature of PPI networks. One simple way of doing so would be to do the same analysis with a PPI network derived from a single screen (eg PMID: 25416956). I assume that due to the much lower coverage the effect will be largely reduced but it would be reconfirming to see a similar trend in addition to the networks on which the authors are already testing. Another way would be to use a randomized network (with the same degree distribution as the networks the authors are using and then picking degree matched random nodes) in which the observed effect should vanish.

      *

      Our response: We appreciate the reviewer’s point and have now assessed both of the suggested approaches.

      The overlap with unbiased yeast two-hybrid (y2h) screens, even the recent HuRI dataset (Luck et al, Nature 2020), was too small in scale to draw any conclusions. Among the ~140,000 interactions tested for genetic interactions, only 51 overlap with y2h interactions. Two of the discovered genetic interactions were supported by a y2h interaction, while one of the robust genetic interactions was supported by a y2h interaction. While this is actually more than would be expected based on the overlap of interactions in the test space the numbers are not especially convincing.

      We therefore focused on two alternative assessments. We first compared our results with the network derived from the systematic AP-MS mapping of protein interactions in HEK293 cells (BioPlex 3.0, Huttlin et al, Biorxiv 2020). We restricted our analysis of genetic interactions to gene pairs that could conceivably be observed in the BioPlex dataset (i.e. between baits screened and preys expressed in HEK293T). We found that although the numbers were small, the same pattern of enrichment was observed:

      This analysis has now been added to the revised manuscript as Supplementary Table S4 and Figure S3E (shown below):

      We next compared the results we observed with the real STRING protein-protein interaction network to 100 degree-matched randomisations of this network. We observed that the number of discovered and validated genetic interactions observed using the real STRING interaction network was greater than that observed using the randomised networks. With this in mind, we have now revised the manuscript to state:

      ‘Previous work has demonstrated that the protein-protein interaction networks aggregated in databases are subject to significant ascertainment bias – some genes are more widely studied than others and this can result in them having more reported protein-protein interaction partners than other genes(Rolland et al., 2014). As cancer driver genes are studied more widely than most genes, they may be especially subject to this bias. To ensure the observed enrichment of protein-protein interactions among genetically interacting pairs was not simply due to this ascertainment bias, we compared the results observed for the real STRING protein-protein interaction network with 100 degree-matched randomised networks and again found that there was a higher than expected overlap between protein-protein interactions and both discovered and validated genetic interactions (Supplemental Fig. S4).’

      Supplemental Figure S4. Genetic interactions are more enriched in real protein-protein interaction networks than randomised networks. Histograms showing the overlap between 100 degree matched randomisations of the STRING medium confidence protein-protein interaction and discovered (a and b) and validated (c and d) genetic interactions. The observed overlap with the real STRING protein interaction are highlighted with the orange lines.

      * 2 . What's the expected number of robust genetic interactions involving passenger gene alterations? Is it surprising to identify 11 interactions? This question could be addressed with some sort of randomization test: When selecting (multiple times) 47,781 non-interacting random pairs between the 2,972 passenger genes and 2,149 selectively lethal genes, how many of those pairs form robust genetic interactions?

      *

      Our response: We have now addressed this as follows:

      “At an FDR of 20% we found 11 robust genetic interactions involving passenger gene alterations (Supplemental Table S6). To assess whether this is more than would be expected by chance we randomly sampled 47,781 gene pairs from the same search space 100 times. The median number of robust genetic interactions identified amongst these randomly sampled gene pairs was 1 (mean 1.27, min 0, max 6) suggesting that the 11 robust genetic interactions observed among protein-protein interacting pairs was more than would be expected by chance.”

      \*Minor comments:**

      Two additional analyses would add in my opinion value to the manuscript:

      -The authors state that reasons for irreproducibility of genetic interactions are of technical or biological nature. Is it possible to disentangle the contribution of the two factors given the available data? Eg how many genetic interactions are reproducible in two different screening platforms using the same cell line vs how similar are results of screens from two different cell lines in the same study?

      *

      Our response: We are also very interested in this question, but with the available data, we are not confident that we could draw solid conclusions.

      -The authors state that "some of the robust genetic dependencies could be readily interpreted using known pathway structures" and argue that they recover for example MAPK or Rb pathway relationships. Is this a general trend? Do genes forming a robust genetic interactions have a higher tendency to be in the same pathway as opposed to different pathways?

      Our response: We have now systematically tested the robust genetic interactions for each driver gene for enrichment in specific pathways. Relevant text is as follows:

      ‘To test if this enrichment of pathway members among the robust dependencies associated with specific driver genes was a common phenomenon, for each driver gene with at least three dependencies we asked if these dependencies were enriched in specific signalling pathways (see Methods). Of the twelve driver genes tested, we found that five of these were enriched in specific pathways and in all five cases found that the driver gene itself was also annotated as a member of the most enriched pathway (Table SX). As expected RB1 (most enriched pathway ‘G1 Phase’) and BRAF (most enriched pathway ‘Negative feedback regulation of MAPK pathway’) were among the five driver genes, alongside PTEN (‘PI3K/AKT activation’), CDKN2A (‘Cell cycle’), and NRAS (‘Ras signaling pathway’).’

      Details in the methods are as follows:

      ‘Pathway enrichment was assessed using gProfiler (Raudvere et al., 2019) with KEGG (Kanehisa et al., 2017) and Reactome (Jassal et al., 2020) as annotation databases and the selectively lethal genes as the background list.’

      *I think the pathway topic could be in general better exploited: eg does pathway (relative) position play a role?**

      *

      Our response: We agree that pathway position, especially distance from driver gene in an ordered pathway, would be very interesting to tease out but we don’t think that current pathway annotations are reliable enough nor the set of robust genetic interactions large enough to analyse this properly.

      *Reviewer #1 (Significance (Required)):**

      Personalized cancer medicine aims at the identification of patient-specific vulnerabilites which allow to target cancer cells in the context of a specific genotype. Many oncogenic mutations cannot be targeted with drugs directly. The identification of genetic interactions is therefore of crucial importance. Unfortunately, genetic interactions show little reproducibility accross studies. The authors make an important contribution to understanding which factors contribute to this reproducibility and thereby providing means to also identify more reliable genetic interactions with high potential for clinical exploitation or involving passenger gene alterations (which are otherwise harder to detect for statistical reasons).

      REFEREES CROSS COMMENTING

      Reviewer 2 raises a few valid points, which if addressed would certainly increase the clarity of the paper. In particular addressing the first point (the self interactions of tumor suppressors) seems important to me. From what I can see all of reviewer 2's comments can be addressed easily.

      *

      End of Reviewer 1 comments

      Reviewer #2 (Evidence, reproducibility and clarity (Required)):

      *In this manuscript, Lord et al. describe the analysis of loss-of-function (LOF) screens in cancer cell lines to identify robust (i.e., technically reproducible and shared across cell lines) genetic dependencies. The authors integrate data from 4 large-scale LOF studies (DRIVE, AVANA, DEPMAP and SCORE) to estimate the reproducibility of their individual findings and examine their agreement with other types of functional information, such as physical binding. The main conclusions from the analyses are that: a) oncogene-driven cancer cell lines are more sensitive to the inhibition of the oncogene itself than any other gene in the genome; b) robust genetic interactions (i.e., those observed in multiple datasets and cell lines driven by the same oncogene/tumour suppressor) are enriched for gene pairs encoding physically interacting proteins.

      **Main comments:**

      I think this study is well designed, rigorously conducted and clearly explained. The conclusions are consistent with the results and I don't have any major suggestions for improving their support. I do, however, have a few suggestions for clarifying the message.

      *

      Our response: We thank the reviewer for this positive assessment of our manuscript and have addressed the requests for clarity below.

      -Could the authors provide some intuitive explanation (or speculation) about the 2 observed cases of tumour suppressor "addiction" (TP53 and CDKN2A)? While the oncogene addiction cases are relatively easy to interpret, the same effects on tumour suppressors are less clear. Is it basically an epistatic effect, which looks like a relative disadvantage? For example, if we measure fitness: TP53-wt = 1, TP53-wt + CRISPR-TP53 = 1.5, TP53-mut = 1.5, TP53-mut + CRISPR-TP53 = 1.5. That is, inhibiting TP53 in TP53 mutant cells appears to be disadvantageous (relative to WT) only because inhibiting TP53 in wild-type cells is advantageous?

      Our response: The reviewer is correct – the TP53 / TP53 dependency is similar to an epistatic effect. In a TP53 mutant background targeting TP53 with shRNA or CRISPR has a neutral effect, while in a TP53 wild type background targeting TP53 with shRNA or CRISPR often causes an increase in cell growth. We have clarified this in the text below (new text in bold)

      ‘We also identified two (2/9) examples of ‘self vs. self’ dependencies involving tumour suppressors -TP53 (aka p53) and CDKN2A (aka p16/p14arf) (Supplemental Fig. S2c). This type of relationship has previously been reported for TP53: TP53 inhibition appears to offer a growth advantage to TP53 wild type cells but not to TP53 mutant cells(Giacomelli et al., 2018). Inhibiting TP53 in TP53 mutant cells has a largely neutral effect, while on average inhibiting TP53 in TP53 wild type cells actually increases fitness growth. Consequently, we observed an association between TP53 status and sensitivity to TP53 inhibition. A similar effect was observed for CDKN2A, although the growth increase resulting from inhibiting CDKN2A in wild-type cells is much lower than that seen for TP53 (Supplemental Fig. S2c).;

      *-In the analysis of overlap between genetic and physical interactions, the result should be presented more precisely. Currently, the text reads "when considering the set of all gene pairs tested, gene pairs whose protein products physically interact were more likely to be identified as significant genetic interactors". However, the referenced figure (Fig. 5a) shows an orthogonal perspective: relative to all gene pairs tested, those that have a significant genetic interaction are more likely to have a physical interaction as well. In other words, in the text, we are comparing the relative abundance of genetic interactions in 2 sets: tested and physically interacting. However, in the figure, we are comparing the relative abundance of protein interactions in 2 sets -- tested and genetically interacting. The odds ratio and the p-values stay the same but the result would be more clear if the figure matched the description in the text.

      *

      Our response: Due to the fact that genetic interactions are rare (~1% of all gene pairs tested have a discovered genetic interaction, ~0.1% have a validated genetic interaction) it’s hard to convey the enrichment effectively. This is demonstrated in the below figure – it’s clear that there are more discovered / validated genetic interaction pairs among the protein-protein interaction pairs but the scale is hard to appreciate:

      Focusing only on the discovered/validated genetic interactions makes the picture a little clearer but does not effectively show that the discovered pairs themselves are enriched among protein-protein interaction pairs

      As we feel the original figures convey the main message most effectively, we have altered the text rather than the images as follows:

      “We found that, when considering the set of all gene pairs tested, gene pairs identified as significant genetic interactors in at least one dataset are more likely to encode proteins that physically interact (Fig. 5a)”

      \*Minor comments:**

      There're a few places where the more explicit explanation would improve the readability of the manuscript.

      -Page 5: The multiple regression model used to identify genetic interactions is briefly mentioned in the text (and described more extensively in the methods). I think it would be better to explicitly describe the dependent and independent variables of the model in the text, so that the reader can intuitively understand what is being estimated*.

      Our response: We have added additional information to the main text as follows:

      ‘This model included tissue type, microsatellite instability and driver gene status as independent variables and gene sensitivity score as the dependent variable (Methods). Microsatellite instability was included as a covariate as it has previously been shown to be associated with non-driver gene specific dependencies (Behan et al., 2019), while tissue type was included to avoid confounding by tissue type.’*

      -Page 5: "Using this approach, we tested 142,477 potential genetic dependencies..." -- could the authors provide a better explanation of where that number is coming from? E.g., 142,477 = ... driver genes x 2470 selectively lethal genes?*

      Our response: Because not every selectively lethal gene is tested in every dataset (e.g. DRIVE only screened ~8,000 genes instead of the whole genome) the 142,477 number does not correspond to a simple multiplication of number of driver genes times number of selectively lethal gene. However, we have added additional information in bold as follows:

      ‘Using this approach, we tested 142,477 potential genetic dependencies between 61 driver genes and 2,421 selectively lethal genes. We identified 1,530 dependencies that were significant in at least one discovery screen (Fig. 2a, Supplemental Fig. S1). All 61 driver genes had at least one dependency that was significant in at least one discovery screen while less than half of the selectively lethal genes (1,141 / 2,421) had a significant association with a driver gene. Of the 1,530 dependencies that were significant in at least one discovery screen, only 229 could be validated in a second screen (Supplemental Table S3, Fig. 2a). For example, in the AVANA dataset TP53 mutation was associated with resistance to inhibition of both MDM4 and CENPF, but only the association with MDM4 could be validated in a second dataset (Fig. 2b, 2c). Similarly, in the DEPMAP dataset NRAS mutation was associated with increased sensitivity to the inhibition of both NRAS itself and ERP44, but only the sensitivity to inhibition of NRAS could be validated in a second dataset (Fig. 2b, 2c).

      The 229 reproducible dependencies involved 31 driver genes and 204 selectively lethal genes.’

      -Page 5: Repeating the number of findings of each type would help understanding the landscape of the genetic dependencies (suggested numbers in brackets): "Of the (229?) reproducible genetic dependencies nine were 'self vs self' associations". "The majority (7/9?) of these ... were oncogene addiction effects". "We also identified 2 (2/9?) examples of 'self vs self' dependencies involving tumour suppressors".

      Our response: We have taken the reviewer’s advice and added these figures to the main text for clarity

      * -Page 12: "Three of these interactions involve genes frequently deleted with the tumour suppressor CDKN2A (CDKN2B and MTAP) and mirror known associations with CDKN2A". It is not clear what "mirror" means -- do they recapitulate known interactions?

      *

      Our response: Yes, we meant to indicate that they recapitulate known CDKN2A interactions and have now replaced ‘mirror’ with ‘recapitulate’.

      -Page 15: "Although we have not tested them here, other features predictive of between-species conservation may also be predictive of robustness to genetic heterogeneity" -- could the authors explicitly list the features?

      Our response: We have now explicitly listed these features as follows:

      “Previous work has also shown that genetic interactions between gene pairs involved in the same biological process, as indicated by annotation to the same gene ontology term, are more highly conserved across species (Ryan et al., 2012; Srivas et al., 2016). Similarly, genetic interactions that are stable across experimental conditions (e.g. that can be observed in the presence and absence of different DNA damaging agents) are more likely to be conserved across species (Srivas et al., 2016). Although we have not tested them here, these additional features predictive of between-species conservation may also be predictive of robustness to genetic heterogeneity.”

      *Reviewer #2 (Significance (Required)):

      The identification of a significant overlap between genetic and physical interactions in cancer cell lines is an interesting and promising observation that will help understanding known genetic dependencies and predicting new ones. However, similar observations have been made in other organisms and biological systems. These past studies should be referenced to provide a historical perspective and help define further analyses in the cancer context. In particular, studies in yeast S. cerevisiae have shown that, not only there is a general overlap between genetic interactions (both positive and negative) and physical interactions, but at least 2 additional features are informative about the relationship: a) the relative strength of genetic interactions and b) the relative density of physical interactions (i.e., isolated interaction vs protein complexes). Here's a sample of relevant studies: 1) von Mering et al., Nature, 2002; 2) Kelley & Ideker, Nat Biotechnol, 2005; 3) Bandyopadhyay et al., PLOS Comput Biol, 2008; 4) Ulitsky et al., Mol Syst Biol, 2008; 5) Baryshnikova et al., Nat Methods, 2010; 6) Costanzo et al., Science, 2010; 7) Costanzo et al., Science, 2016.

      Similar observations have also been made in mammalian systems: e.g., in mouse fibroblasts (Roguev et al., Nat Methods, 2013) and K562 leukemia cells (Han et al., Nat Biotech, 2017). I don't think that past observations negate the novelty of this manuscript. The analysis presented here is more focused and more comprehensive as it is based on a large integrated dataset and is driven by a series of specific hypotheses. However, a reference to previous publications should be made.

      As a frame of reference: my expertise is in high-throughput genetics of model organisms, including mapping and analyzing genetic interactions.

      *

      Our response: We thank the reviewer for highlighting this point.

      We have attempted to provide better context for our work in the discussion as follows:

      ‘In budding and fission yeast, multiple studies have shown that genetic interactions are enriched among protein-protein interaction pairs and vice-versa (Costanzo et al., 2010; Kelley and Ideker, 2005; Michaut et al., 2011; Roguev et al., 2008). Pairwise genetic interaction screens in individual mammalian cell lines have also revealed an enrichment of genetic interactions among protein-protein interaction pairs (Han et al., 2017; Roguev et al., 2013). Our observation that discovered genetic interactions are enriched in protein-protein interaction pairs is consistent with these studies. However, these studies have not revealed what factors influence the conservation of genetic interactions across distinct genetic backgrounds, i.e. what predicts the robustness of a genetic interaction. In yeast, the genetic interaction mapping approach relies on mating gene deletion mutants and consequently the vast majority of reported genetic interactions are observed in a single genetic background (Tong et al., 2001). In mammalian cells, pairwise genetic interaction screens across multiple cell lines have revealed differences across cell lines but not identified what factors influence the conservation of genetic interactions across cell lines(Shen et al., 2017). While variation of genetic interactions across different strains or different genetic backgrounds has been poorly studied, previous work has analysed the conservation of genetic interactions across species and shown that genetic interactions between gene pairs whose protein products physically interact are more highly conserved (Roguev et al., 2008; Ryan et al., 2012; Srivas et al., 2016). Our analysis here suggests that the same principles may be used to identify genetic interactions conserved across genetically heterogeneous tumour cell lines.’

    2. Note: This preprint has been reviewed by subject experts for Review Commons. Content has not been altered except for formatting.

      Learn more at Review Commons


      Referee #2

      Evidence, reproducibility and clarity

      In this manuscript, Lord et al. describe the analysis of loss-of-function (LOF) screens in cancer cell lines to identify robust (i.e., technically reproducible and shared across cell lines) genetic dependencies. The authors integrate data from 4 large-scale LOF studies (DRIVE, AVANA, DEPMAP and SCORE) to estimate the reproducibility of their individual findings and examine their agreement with other types of functional information, such as physical binding. The main conclusions from the analyses are that: a) oncogene-driven cancer cell lines are more sensitive to the inhibition of the oncogene itself than any other gene in the genome; b) robust genetic interactions (i.e., those observed in multiple datasets and cell lines driven by the same oncogene/tumour suppressor) are enriched for gene pairs encoding physically interacting proteins.

      Main comments:

      I think this study is well designed, rigorously conducted and clearly explained. The conclusions are consistent with the results and I don't have any major suggestions for improving their support. I do, however, have a few suggestions for clarifying the message.

      -Could the authors provide some intuitive explanation (or speculation) about the 2 observed cases of tumour suppressor "addiction" (TP53 and CDKN2A)? While the oncogene addiction cases are relatively easy to interpret, the same effects on tumour suppressors are less clear. Is it basically an epistatic effect, which looks like a relative disadvantage? For example, if we measure fitness: TP53-wt = 1, TP53-wt + CRISPR-TP53 = 1.5, TP53-mut = 1.5, TP53-mut + CRISPR-TP53 = 1.5. That is, inhibiting TP53 in TP53 mutant cells appears to be disadvantageous (relative to WT) only because inhibiting TP53 in wild-type cells is advantageous?

      -In the analysis of overlap between genetic and physical interactions, the result should be presented more precisely. Currently, the text reads "when considering the set of all gene pairs tested, gene pairs whose protein products physically interact were more likely to be identified as significant genetic interactors". However, the referenced figure (Fig. 5a) shows an orthogonal perspective: relative to all gene pairs tested, those that have a significant genetic interaction are more likely to have a physical interaction as well. In other words, in the text, we are comparing the relative abundance of genetic interactions in 2 sets: tested and physically interacting. However, in the figure, we are comparing the relative abundance of protein interactions in 2 sets -- tested and genetically interacting. The odds ratio and the p-values stay the same but the result would be more clear if the figure matched the description in the text.

      Minor comments:

      There're a few places where the more explicit explanation would improve the readability of the manuscript.

      -Page 5: The multiple regression model used to identify genetic interactions is briefly mentioned in the text (and described more extensively in the methods). I think it would be better to explicitly describe the dependent and independent variables of the model in the text, so that the reader can intuitively understand what is being estimated.

      -Page 5: "Using this approach, we tested 142,477 potential genetic dependencies..." -- could the authors provide a better explanation of where that number is coming from? E.g., 142,477 = ... driver genes x 2470 selectively lethal genes?

      -Page 5: Repeating the number of findings of each type would help understanding the landscape of the genetic dependencies (suggested numbers in brackets): "Of the (229?) reproducible genetic dependencies nine were 'self vs self' associations". "The majority (7/9?) of these ... were oncogene addiction effects". "We also identified 2 (2/9?) examples of 'self vs self' dependencies involving tumour suppressors".

      -Page 12: "Three of these interactions involve genes frequently deleted with the tumour suppressor CDKN2A (CDKN2B and MTAP) and mirror known associations with CDKN2A". It is not clear what "mirror" means -- do they recapitulate known interactions?

      -Page 15: "Although we have not tested them here, other features predictive of between-species conservation may also be predictive of robustness to genetic heterogeneity" -- could the authors explicitly list the features?

      Significance

      The identification of a significant overlap between genetic and physical interactions in cancer cell lines is an interesting and promising observation that will help understanding known genetic dependencies and predicting new ones. However, similar observations have been made in other organisms and biological systems. These past studies should be referenced to provide a historical perspective and help define further analyses in the cancer context. In particular, studies in yeast S. cerevisiae have shown that, not only there is a general overlap between genetic interactions (both positive and negative) and physical interactions, but at least 2 additional features are informative about the relationship: a) the relative strength of genetic interactions and b) the relative density of physical interactions (i.e., isolated interaction vs protein complexes). Here's a sample of relevant studies: 1) von Mering et al., Nature, 2002; 2) Kelley & Ideker, Nat Biotechnol, 2005; 3) Bandyopadhyay et al., PLOS Comput Biol, 2008; 4) Ulitsky et al., Mol Syst Biol, 2008; 5) Baryshnikova et al., Nat Methods, 2010; 6) Costanzo et al., Science, 2010; 7) Costanzo et al., Science, 2016.

      Similar observations have also been made in mammalian systems: e.g., in mouse fibroblasts (Roguev et al., Nat Methods, 2013) and K562 leukemia cells (Han et al., Nat Biotech, 2017). I don't think that past observations negate the novelty of this manuscript. The analysis presented here is more focused and more comprehensive as it is based on a large integrated dataset and is driven by a series of specific hypotheses. However, a reference to previous publications should be made.

      As a frame of reference: my expertise is in high-throughput genetics of model organisms, including mapping and analyzing genetic interactions.

      REFEREES CROSS COMMENTING

      I agree with the questions raised by reviewer #1. And I think the authors should be able to address them (either through analyses or reasoning) within 1-3 months.

    1. In short, scale-free networks are resilient when it comes to random deletionof nodes but highly vulnerable if hubs go down or can be targeted for attack

      This gets me thinking about what constitutes the network and if there is a type of network that wouldn't be as highly vulnerable if a large hub went down. For example, if we consider the journalistic/broadcasting network where you have major hubs (NYT, Washington Post, NBC News, Fox, etc.), would it really wreak havoc? It may just be taken over by another one of the hubs. Can we think about it in this way?

      But, if we talk about the individual New York Times as a network itself, then you may see more obvious havoc within that network if a large hub (VP or President or CEO) "goes down".

    1. Abstract This report presents a rapid review of the evidence on reflective practice in crisis situations. In the light of the current Covid-19 pandemic we asked the question “What lessons can we learn about the value of reflective practice that may be of service to our health professionals on the front line”. A search of Scopus (with no date restrictions was made. A total of 56 papers were identified. After review of titles and abstract 34 papers were rejected as not being relevant. The remaining 22 papers were reviewed and their findings synthesised. Most of the papers (n=9) were reflections on relevant experiences. Five were commentaries, six reported the findings from qualitative studies and two reported findings from cross sectional surveys. The findings and recommendations can be presented in terms of strategies that can be employed when preparing for a crisis, when in the midst of a crisis and following conclusion of a crisis. There are two key recommendations identified from the review: 1. There are things we can do now: The importance right now of ensuring that staff have both time and a safe space in which they can reflect on their experiences either with fellow staff or via peer- facilitated reflections. That way those staff who wish to, could be supported to reflect on and process their after a difficult shift. 2. There are things we can think about in the mid to longer term: the importance of organisations learning from the pandemic. Post pandemic, perhaps developing simulations, but also formally engaging in structured reflective practice to ensure practical skills are honed and lessons learned and b) paying attention to the organisational culture and ensuring that reflective practice is embedded as ‘business as usual’ Underpinning these two points are a range of issues organisations should bear in mind. These include: • Supportive and encouraging organisational culture that recognises the importance of embedding reflective practice in everyday work • Leaders should themselves be involved in the process of reflection as participants • Staff need time and space to reflect on their experiences
    1. If you’re a controller based outside of the EU, you’re transferring personal data outside of the EU each time you collect data of users based within the EU. Please make sure you do so according to one of the legal bases for transfer.

      Here they equate collection of personal data with transfer of personal data. But this is not very intuitive: I usually think of collection of data and transfer of data as rather different activities. It would be if we collected the data on a server in EU and then transferred all that data (via some internal process) to a server in US.

      But I guess when you collect the data over the Internet from a user in a different country, the data is technically being transferred directly to your server in the US. But who is doing the transfer? I would argue that it is not me who is transferring it; it is the user who transmitted/sent the data to my app. I'm collecting it from them, but not transferring it. Collecting seems like more of a passive activity, while transfer seems like a more active activity (maybe not if it's all automated).

      So if these terms are equivalent, then they should replace all instances of "transfer" with "collect". That would make it much clearer and harder to mistakenly assume this doesn't apply to oneself. Or if there is a nuanced difference between the two activities, then the differences should be explained, such as examples of when collection may occur without transfer occurring.

    1. The bare idea of this is no doubt horribly repulsive to us, but at the same time I think that we should remember how repulsive our carnivorous habits would seem to an intelligent rabbit.

      GANGNES: The text beginning with "I know it is..." and ending with "But I wander from my subject" several paragraphs later was cut from the 1898 volume. See text comparison page.

      STOVER argues, "The reason Wells cut this passage from the book version is probably aesthetic. He did not wish to give away too much, if he were to keep with the novel's deepest artistic ambiguity" (188). However, this assessment risks oversimplifying an extensive edit. Apart from "giving away too much"--offering a lot of information that the narrator would not find out until much later and therefore informing the reader of details about the Martians relatively early--this passage can come off as "preachy" or overly philosophical in a way that Wells may have later decided he disliked.

      This omitted section tells us a great deal not only about the Martians' grisly study of a live human subject, but also about the narrator's ideologies. Looking back on his first glimpses of the Martians from a later time of safety, the narrator offers a kind of persuasive philosophical essay (he is, by trade, a professional writer of similar essays) on the ethical and moral lessons to be gleaned, from the Martians' behavior, about humans' treatment of other animals.

      While the passage may "wander from [the narrator's] subject," it offers an intriguing dissonance between the narrator's terror of being killed by the Martians--to the point where he sacrifices others' lives--and his cool, high-minded defense of their consumption of human beings.

      In the end, Wells retains only the first sentence of this passage in the volume to speak very briefly to the narrator's philosophical thoughts on the matter. What we gain in narrative flow and "artistic ambiguity" we may lose in characterization.

    1. We must never forget that we may also find meaning in life even when confronted with a hopeless situation, when facing a fate that cannot be changed. For what then matters is to bear witness to the uniquely human potential at its best, which is to transform a personal tragedy into a triumph, to turn one's predicament into a human achievement. When we are no longer able to change a situation— just think of an incurable disease such as inoperable cancer —we are challenged to change ourselves.

      I think this is the question that logotherapy answers the best, there are other ways of understanding Love, there are other ways of understanding your own individual life, but suffering is by far the hardest as we constantly reject suffering. I think what Frankl is trying to say here is that for our individual situations where we are faced with the worst moments in our lives, that only we can determine how we deal with it, And in doing so find an understanding of life through that Hell, in which we must compel ourselves to suffer through and triumph.

    1. When a storm subsides, the air is washed clean of whatever particulate matter has been obscuring the view, and you can often see farther and more sharply than at any other time. When this storm clears, we may, as do people who have survived a serious illness or accident, see where we were and where we should go in a new light.

      I think that after this pandemic everyone will think differently .

    2. A new awareness of how each of us belongs to the whole and depends on it may strengthen the case for meaningful climate action, as we learn that sudden and profound change is possible after all.

      Disasters can make us think about how we as a whole can better develop and cope with the huge changes that may occur in the future.

    3. When a storm subsides, the air is washed clean of whatever particulate matter has been obscuring the view, and you can often see farther and more sharply than at any other time. When this storm clears, we may, as do people who have survived a serious illness or accident, see where we were and where we should go in a new light.

      I think after this, many countries would take this as a lesson.

  11. Apr 2020
    1. Note: This rebuttal was posted by the corresponding author to Review Commons. Content has not been altered except for formatting.

      Learn more at Review Commons


      Reply to the reviewers

      We thank the reviewers and the editor for the insightful and thorough assessment of our manuscript. In this response to review letter, we have listed the original review (black text) and responded to each critique after it.

      Reviewer #1 (Evidence, reproducibility and clarity (Required)):

      Yang et al. submitted a manuscript describing the detection of pseudogenes ("retrocopies") of APOBEC3 (A3) genes in primates. The evolutionary history and relationship to specific A3s was analyzed and speculated that the maintained A3 retrocopies had a functional role at least early in the evolution and some may have still now. Functional data on some of the expressed retrocopies are presented on L1 and HIV.

      The authors claim that "retrocopying expands the functional repertoire of A3 antiviral proteins in primates". While almost of the genetic findings were published recently (Ito et al. 2020), the authors should more clearly describe how their data differ or confirm the data of Ito et. al.

      We thank the reviewer for their helpful comments which have guided revisions to our manuscript. We have taken steps to clarify the dramatic differences between our work and the recent publication from Ito, Gifford, and Sato.

      Foremost, we respectfully disagree with the reviewer that the genetic findings in our work were contained within the Ito, et al manuscript. Using a computational screen of assembled mammalian genome, the Sato group cataloged the gain and loss of APOBEC3 genes during the evolution of mammals. They found a fascinating correlation between the dynamics of A3s and ERVs that formed the precis of the paper. From their genome-wide search for A3s, Ito et al describe several retrocopies of A3s in two New World Monkey species, one of which retains a full-length open reading frame, leading to the statement that this gene may be functional.

      We note that the retrocopies found in the Ito et al paper span only two of the more than 20 species in which we identify A3 retrocopies. Further, as a result of the breadth of our search for A3s, we find additional retrocopies in the same two New World Monkey species that were examined in the Ito et al paper. Finally, our study also examined functional capabilities of these additional A3s. These differences are highlighted by reviewer 3 who writes that relative to Ito et al, our manuscript studies the phenomenon of A3 retrocopies “more deeply both by in silico analyses and cell culture experiments.” Reviewer 3 also summarizes the most important difference in our studies – our work presents a “conceptual advance that the antiviral gene expansion has achieved not only via tandem gene duplication but also via gene retrocopying”.

      Lastly, we want to point out that the findings of our manuscript and Ito et al. 2020 were made concurrently. Indeed, throughout the preparation process of this manuscript, we were both aware of each other’s findings and shared preprints with each other. Most of the participating journals in Review Commons have “scoop protection” mechanisms that typically extend 6 months after the publication of the first article (Ito et al was published Jan 2020), and our article was first submitted to Reviewer Commons on February 14, 2020. Therefore, we feel confident that the ‘no scoop’ policy applies to the minimal overlap between our paper and that of Ito et al.

      Nevertheless, we have modified the text to more clearly acknowledge the parallel finding of some New World monkey retrogenes in the Ito, et al. paper.

      The functional data (Fig. 6) are interesting, but in the current form not complete. The authors have to show protein expression in the transfected cells (A3, L1, HIV) and level of encapsidation into viral particles. In addition, please analyze if the retrocopies express cytidine deaminase active enzymes.

      We thank the reviewer for this comment, and we have added a Western blot of the six long-ORF-containing retrocopies as Figure S5. In this blot (from early in the project), we detected protein production in 293T cells for 3/6 retrocopies. In later optimizations of subsets of this blot, we were able to detect expression of the marmoset A3G and the other two marmoset retrocopies (marmoset-2 and marmoset-4). Despite optimization attempts, we were unable to detect protein for one of the retrocopies that restricts HIV-1ΔVif (capuchin-C1). Unfortunately, at this time the included blot is the only one we have in which all 6 constructs are included on a single blot. Optimally, all 6 constructs would be side-by-side in a single blot with optimized conditions, and we are happy to complete this experiment as soon as we are able to return to our lab after the SARS-2 quarantine is lifted. However, we think the added blot shows that some of the retrocopies produce protein and the absence of detectable protein from capuchin-C1 could suggest that this retrocopy is especially potent in its restriction function or an idiosyncratic problem with detecting this protein using Western blot analyses.

      We have not previously tested our lentiviral particles for levels of encapsidation of protein from each retrocopy. The value we see in this experiment is in explaining why some of the retrocopies that are expressed in producer cells may not restrict in target cells. While we note that precedent in the literature suggests that A3 proteins which restrict HIV-1ΔVif are invariably encapsidated, we would be happy to carry out this experiment when our lab reopens.

      In response to the reviewer’s request to test deaminase activity for each retrocopy, we note that Figure 4 shows the intactness of the deaminase motif in each retrocopy. However, we feel that a description of the mechanisms of restriction of these retrocopies is not a major point of this paper and is beyond the scope of the current investigation.

      Reviewer #1 (Significance (Required)):

      Minor advance compared to Ito et al. 2020.

      We respectfully and rigorously disagree with this assessment. Please refer back to the reviewer’s first comment. We defer, again, to Reviewer 3’s assessment that our work presents a “conceptual advance that the antiviral gene expansion has achieved not only via tandem gene duplication but also via gene retrocopying”. Moreover, we must point out that the Ito et al 2020 paper was entirely computational; indeed, several retrogenes that could computationally be predicted to be ‘dead’ were confirmed by us as having antiviral activity.

      Reviewer #2 (Evidence, reproducibility and clarity (Required)):

      **Summary:**

      Yang et al. study the expansion of APOBEC3 (A3) cytidine deaminases genes in primates. Authors find A3 retrocopies in several lineages in primates using Blast searches. Some are old and some are species specific. Some have disablements and some have intact ORFs. Authors study their mode of evolution, expression and functionality. Authors have performed detailed analyses including functional analyses. Some A3 retrocopies are broadly expressed and some have retained ability to restrain retroelements. I agree with the authors that their data supports that retrocopying has contributed the turnover in the repertoire of host retroelement restriction factors. Authors show that some retrocopies have remained active for long periods of time and they still show that they can restrict retroelements/retrovirus. This work provides an interesting example of immune system diversification. This study of the A3 family of proteins that are part of the vertebrate innate immune system and the data supporting turnover of these kind of immune system genes is strong. The work underscores that this is a way immunity genes evolve and it has parallels in the evolution of the TRIM gene family of immune genes. I just have a few comments. I think the work can gain from analyzing some aspects of the data in more detail and presenting the big picture in a summary table, even if it is just supplementary.

      **Major comments:**

      A3I is in many species. Does this mean it was preserved (i.e., functional for a while)? For how long have disabling mutations been accumulating? Can we get a sense of that? Even for other retrocopies, do we have a sense of how recent has the pseudogenization been? If it is very recent that means that the gene was active until not long ago.

      Our analyses suggest that A3I was born in the common ancestor of simian primates and pseudogenized before the Catarrhini/Platyrrhini split. It is possible that A3I was functional within this extended period (~12-15 million years), but the presence of a shared truncating stop codon amongst all simian A3Is suggests the gene was no longer full-length at the time of diversification of the simians. Instead, the simian LCA likely encoded an A3I with a predicted ORF of 261 codons; if this truncated ORF were functional, it was then further truncated/pseudogenized with additional frame-breaking mutations which follow the phylogeny of primates.

      We estimated the timeline of pseudogenization of each retrocopy using the species distribution of each syntenic retrocopy. We also note that we find full-length ORFs in three retrocopies which have been retained for a period of time at least as long as the age of the last common ancestor of the four New World monkeys. These old but intact retrocopies motivated our simulations of ORF retention rates (Figure 5).

      In the PAML analyses test could be performed to test if the rate of evolution that are higher or lower than 1 for particular genes are actually significantly higher or lower than 1 for the particular gene comparing the likelihoods of the modes with the given rate with the one with the rate fixed to 1. Is there enough power to do this?

      We thank the reviewer for pointing out this omission in our analysis. We did perform these tests and find a significant p-value for two of the nodes p=0.058 and p=0.025 respectively). We have updated the legend for figure S4 to incorporate these p-values

      Page 9. It seems to me that the synteny data Figure S2 reveals they are derived from independent retroposition events and not duplications of segments because those would include flanking genes. Is this correct? Authors could comment on that.

      Yes, we think that each retrocopy we show in Figure S2 is likely created via an independent retrotransposition event. We have clarified in the text that Figure S2 shows the genes used to establish synteny to support orthology of the retrocopies shared amongst multiple species and that each of these ortholog groups presumably originated via distinct retrotransposition events.

      In figure S4, I am not sure why orthologous genes are not grouped together in the phylogeny and why p is smaller than 0.05. How should that figure and the probability be interpreted?

      We thank the reviewer for their comments on this figure. First, the reviewer identified an error in the tree in which the branch labels for ‘night monkey-C2’ and ‘night monkey-SS1’ were inadvertently switched. The corrected tree now follows the pattern expected by the reviewer. Second, we employed RELAX to “determine whether selective strength was relaxed or intensified in one of these subsets relative to the other” (Wertheim, et al. MBE 2014). In this case, the p-value corresponds to the finding that the retrocopies (test branches) show intensification of selection relative to the intron-containing A3Gs (reference branches).

      We have modified Figure S4 and the associated text to more clearly explain the specific hypothesis test we report.

      It would be good to have a summary table that summarizes what genes have support for past or current functionality (preservation for long time or recent pseudogenization, expression, purifying or positive selection, ability to restrict retroelements) and in what lineages.

      We agree with this reviewer suggestion. We have added the additional information including the number of frame disrupting mutations as a measure of age, intactness, and ability to restrict retroelements to Table S1. Thanks to this suggestion, Table S1 now serves as the master table to summarize the analyses of each retrocopy.

      **Minor comments:**

      1. Page 3. Authors say "...the exons and UTRs..." but UTRs are part of exons. Authors could talk about exons only that include protein-coding regions and UTRs.

      Changed the text to "exons".

      Page 7. I would say disabled instead of "... becoming degraded by mutation."

      Fixed according to the reviewer's suggestion.

      I would say neutral evolution not neutral selection.

      Fixed according to the reviewer's suggestion.

      Reviewer #2 (Significance (Required)):

      This work provides an interesting example of immune system diversification. Authors study the APOBEC3 family of proteins that is part of the vertebrate innate immune system and the data supporting turnover of these kind of immune system genes. The work underscores that this is a way immunity genes evolve and it has parallels in the evolution of the TRIM gene family of immune genes revealing patterns in the mode of evolution of immunity genes. The audience of this work will be people interested in evolution of immunity, arms races and gene diversification and all evolutionary biologists interested in adaptation. I work in the field of comparative genomics and molecular evolution.

      Reviewer #3 (Evidence, reproducibility and clarity (Required)):

      **Summary:**

      This manuscript by Yang et al. is an well-written, intriguing paper highlighting the evolutionary significance of the gene creation via "retrocopying". The authors investigated the expansion of antiviral A3 genes via retrocopy in Primates, and found that A3G-like retrocopies have been generated repeatedly during primate evolution. A part of A3 retrocopies found in New World monkeys retained full length open reading flames and anti-lentiviral capacities. Interestingly, the spectrum of anti-retroelement activity of A3 retrocopies was different from the original (i.e., intron-containing) A3G gene in these species, suggesting the occurrence of the functional differentiation followed by gene amplification. However, one of the main findings that many A3 retrocopies are present in New World monkey is in-line to a previous report (i.e., Ito et al., 2020, PNAS), and the experimental validations were based on the human (not New World monkey's) retroelements. Nevertheless, this study deeply investigated the possible importance of A3 retrocopies for the host defense system evolution both by in silico analyses and cell culture experiments. This study provides the findings that can potentially expand our knowledge on the evolutionary arms races between retroelements and the hosts.

      **Major:**

      To strengthen the impact of this work, it would be better to increase the numbers of retroviruses in which the anti-retroviral capacities are investigated. I understand that it is difficult to examine retroviruses or L1s that are colonized naturally with New World monkeys, but I suppose it is not so difficult to investigate a variety of representative retroviruses such as murine leukemia virus (MLV) or the reconstructed human endogenous retrovirus K (HERV-Kcon). This additional experiment would be helpful to highlight that the spectrum of anti-retroviral activity of A3 retrocopies is divergent from the original A3G gene in these species and strengthen the concept to be proposed by this study.

      The reviewer raises a fascinating question about whether retrocopies might have different restriction abilities relative to the other A3s in a given species. First, we feel that showing activity against one pathogen is sufficient for our claim that some of the A3 retrocopies have antiviral potential. Second, we discuss in the paper the idea that HIV-1 is not the actual target of these (or any) innate immune genes in New world primates. We argue that any other targets we might test would also be surrogates for the ‘true’ target of these genes.

      **Specific:**

      1, Since the authors found the expansion of "functional" repertoire of A3 retrocopies specifically in New World monkey, it would be better to rephrase the title as "Retrocopying expands the functional repertoire of APOBEC3 antiviral proteins in New World monkeys".

      We thank the reviewer for this comment but point out that a large portion of our manuscript presents our work on primates outside the New World monkeys. The reviewer is correct to note that our finding of restriction activity is limited to New World monkey retrocopies, but we feel that the current title will attract a broader audience and reflects the broader relevance of this work.

      2, It might be better to add a figure summarizing which A3 retrocopies in which species retain nearly full length ORFs. For example, how about making a figure like Fig. 2A for all the four representative New World monkey species?

      We agree. We have added the length of the longest ORF for each retrocopy to Table S1.

      3, Fig. 3

      It would be helpful to clarify that which cell of the heatmap corresponds to the intact A3 retrocopies.

      We have added labels to indicate the intact A3 retrocopies and adjusted the legend accordingly.

      4, Page 4, line 5

      It would be better to replace the word "protected" with "escaped" because this retrocopy subset should include the ones that are intact but not functional.

      Changed as suggested.

      5, Page 4, line 25

      It would be better to rephrase "the common ancestor of mammals" as "the common ancestor of placental mammals" because A3 gene is absent in Marsupial.

      Changed as suggested.

      6, Page 5, line 5

      Please rephrase "ongoing" as "recently-occurred".

      Changed as suggested.

      7, Page 6, line 19

      I checked the multiple sequence alignment in File S1 and suspect that the codon (alignment) position of the shared premature stop codon is 261 (not 264).

      We thank the reviewer for pointing out this discrepancy. We have revised the text to reflect the correct position of the shared stop.

      8, Page 6, line 23

      I could not understand the meaning of the sentence "Intriguingly, one lineage-specific mutation...".

      Please specify the position of mutation which the authors mentioned (in File S1 or Fig. 1B).

      This portion of the text refers to a reversion of a stop codon in the orangutan A3I; specifically, the stop codon shared in all simians acquired a second mutation that created a longer ORF in only this species. We have removed this sentence from the text for the sake of clarity.

      9, Page 12, line 8

      Please refer Fig. S4 here.

      Changed as suggested.

      10, Page 12, line 8

      Please say "Significant relaxed selection was not detected" rather than "Our analysis detected no relaxation...".

      Changed as suggested.

      11, Page 12, line 8

      Fig. S4 indicates "p=0.015", but the authors regard it as "not significant"?

      We thank the reviewer for pointing out this confusing wording. We employ RELAX to “determine whether selective strength was relaxed or intensified in one of these subsets relative to the other” (Wertheim, et al. MBE 2014). In this case, the p-value corresponds to the finding that the retrocopies show intensification of selection.

      We have modified Figure S4 to more clearly explain the specific hypothesis test for this p-value. We have also modified the text to clarify this point.

      12, Page 12, line 9

      Please here refer the data showing the claim "Instead, these A3G retrocopies have evolved more rapidly than...".

      Changed as suggested; see previous point.

      13, Page 12, line 11

      Did the authors perform the statistical test on the dN/dS ratio analysis? If so, please mention the result of the test.

      Yes we did. Please refer to Reviewer 2’s ‘Major Point 3’.

      14, Page 12, line 15

      It would be better to modify the phrase "show evidence of recurrent selection for functional innovation"

      Changed as suggested.

      Reviewer #3 (Significance (Required)):

      This study provides a conceptual advance that the antiviral gene expansion has achieved not only via tandem gene duplication but also via gene retrocopying.

      Compare to existing published knowledge.

      Although one of the main findings that many A3 retrocopies are present in New World monkey is in-line to a previous report (i.e., Ito et al., 2020, PNAS), this study investigated the above finding more deeply both by in silico analyses and cell culture experiments.

      Audience.

      Evolutionary biologists and researchers in the field of viruses (particularly retroviruses including HIV-1) and transposable elements would be interested in this work.

      Your expertise.

      Bioinformatics, genome biology, viruses, and transposable elements

    1. Technological literacy is when you are completely competent with the use of technology and are open to learning new things about newer devices. There are three aspects to technological literacy which are capabilities, knowledge, and critical thinking. I would say capabilities and knowledge are the most prevalent and the ones outmost known. Some people would know all the features within their device or piece of technology and utilize all of it. I know my friend’s mom who always gets the latest IPhones and hasn’t used Google maps at all. The next important thing is knowledge which can be mentioned as an another word is basically knowing what technology to use at which moments. Sometimes your laptop may be better to type and print things. While purchasing a laptop knowing more about the specifications allows you to know what exactly you are buying. After this course ends, I would say I would focus on learning more and just getting the mere exposure of new technology would be a good start.

      I think the thing that struck me the most in this class would probably be the amount of readings and the wide array of readings we were exposed to. I am lucky to have chosen this class this semester my final semester at the U. I believe as young millennials we do inherently follow some of the principles that were taught in this class. In the future though I will be more open minded about technology and have learned about how technology literacies can change throughout other peoples lives.

    1. Josh Rushton writes: I’ve been following your blog for a while and checked in today to see if there was a thread on last week’s big-splash Stanford antibody study (the one with the shocking headline that they got 50 positive results in a “random” sample of 3330 antibody tests, suggesting that nearly 2% of the population has been infected “under the radar”). I didn’t see anything, so I thought I’d ask if you’d consider opening a discussion. This paper is certainly relevant to the MrP thread on politicization of the covid response, in that the paper risks injecting misinformation into an already-broken policy discussion. But I think it would be better to use it as a case study on poor statistics and questionable study design. I don’t mean to sound harsh, but if scientists are afraid to “police” ourselves, I don’t know how we can ask the public to trust us. Simply put, I see two potentially fatal flaws with the study (full disclosure: I [Rushton] haven’t read the entire paper — a thousand apologies if I’m jumping the gun — but it’s hard to imagine these getting explained away in the fine print): The authors’ confidence intervals cannot possibly be accounting for false positives correctly (I think they use the term “specificity” to mean “low rate of false-positives). I say this because the test validation included a total of 30+371 pre-covid blood tests, and only 399 of them came back negative. I know that low-incidence binomial CIs can be tricky, and I don’t know the standard practice these days, but the exact binomial 95% CI for the false-positive rate is (0.0006, 0.0179); this is pretty consistent to the authors’ specificity CI (98.3%, 99.9%). For rates near the high end of this CI, you’d get 50 or more false positives in 3330 tests with about 90% probability. Hard to sort through this with strict frequentist logic (obviously a Bayesian could make short work of it), but the common-sense take-away is clear: It’s perfectly plausible (in the 95% CI sense) that the shocking prevalence rates published in the study are mostly, or even entirely, due to false positives. So the fact that their prevalence CIs don’t go anywhere near zero simply can’t be right. Recruitment was done via facebook ads with basic demographic targeting. Since we’re looking for a feature that affects something like 2% of the population (or much, much less), we really have to worry about self selection. They may have discussed this in the portions of the paper I didn’t read, but I can’t imagine how researchers would defeat the desire to get a test if you had reason to believe that you, or someone near you, had the virus (and wouldn’t some people hide those reasons to avoid being disqualified from getting the test?)…
    1. According to Global News, Canada may have a cure but we do not know that for sure or if they do if they will share it.

      Frankly, I don't think this is accurate as is a misreading of the piece. There is no cure, but we can be sure that if any one is discovered by most any country, it would be widely known and reported on. Anything reported now as a 'cure' is undoubtedly unproven and anecdotal.

    1. The ability to see oneself in the monument would lead us to consider the piece as a living/breathing monument that is not static or staid, but as one that reflects today’s America and our current political and social moment. Monuments are established with the assumption that we as a nation have collectively decided that something should be remembered, honored, and celebrated.

      Karyn Oliver confirms the idea that a monument can reflect the viewer's beliefs and values. I think she is challenging the idea of collective memory here by saying "monuments are established wth the assumption that we as a nation have collectively decided..." She thinks that not all people have equal power to shape societal narratives, so the monuments are often one sided and not as "collective" as they may seem.

    1. At a time when we are all thinking about how best to respond to the present global crisis, it seems timely to think also about how we, as the Cognitive Science community, can be most effective. What kind of science can we do, and how should we go about doing it? This blog post is an attempt to help fuel discussion on these issues in order to formulate the best community response. It offers a starting point for thinking about cognitive science and coronavirus.  Though thoughts first turn to medicine, virologists, and epidemologists, the CogSci community has many potential contributions to make. Research areas that are established cognitive science topics, ranging from e-learning, e-delivery, media literacy, through risk analysis, risk perception, decision-making, behaviour change, argumentation, or communication are suddenly in high demand. But as cognitive scientists, we also possess key skills: the ability to interface with AI, handle ‘big data’, engage in computational social science, and maybe first and foremost, modelling skills and an ability for model thinking. And, finally, the sheer disciplinary breadth of Cognitive Science, from computer science, through to anthropology and philosophy, can offer much needed, complementary, perspectives and views. While cognitive scientists around the world consider how their own research skills and ideas may usefully be applied, we should also spend some time rethinking and looking to adjust, how we go about doing science. 
    1. There is a paradox in our desire to be seen as virtuous. If we do not overtly display our virtues, others will not be able to see them; yet, if we do overtly display our virtues, others may think that we do so only for social credit. Here, we investigate how virtue signaling works across two distinct virtues—generosity and impartiality—in eleven online experiments (total N=4,586). We demonstrate the novel phenomenon of differential virtue discounting, revealing that participants perceive actors who demonstrate virtue in public to be less virtuous than actors who demonstrate virtue in private, and, critically, that this effect is greater for generosity than impartiality. Further, we provide evidence for the mechanism underlying these judgments, showing that they are mediated by perceived selfish motivations. We discuss how these findings and our novel terminology can shed light on open questions in the social perception of reputation and motivation.
    1. It was not too long ago, that we were living in a place that everyone could call heaven. Everyone took advantage, not realizing how great it was to live back then. Nearly everyone would complain about how life may had sucked then, all because they were bored that day. Everyone could remember those days like it could have been yesterday. Jessica was still a young teenager back then, young, and happy. Her best friend was named Stephanie. She could remember when they would just spend the whole day talking about what boy was cute, what clothes we liked, what our favorite music was, and other things that just made us happy. Everything was so innocent and happy back then. No one had to even think about tomorrow, because they knew that tomorrow would be another day they would live in peace. Everyone knew that they were in great hands since they had the resources to support themselves and their families to encourage everyone to keep going. Everyone thought that this was ironic thinking about this now. People lived in a life that they could possibly even call heaven on earth.

      Again, the intro is highly disconnected from the rest of your story. A good introduction should not only introduce us to who the main characters are, it should also demonstrate what the theme of your story is going to be. As it is, the characters Jessica and Stephanie do not appear anywhere again, while the main characters Akilles and Mike appear far later on.

    1. We are talking about Whiting Petroleum, and Brad serves as both its Chairman of the Board and Chief Executive Officer. On March 26, 2020, that board paid him and his fellow executives $14.6 million in bonuses. Holley himself pocketed $6.4 million. Six days later, that same board sent Whiting Petroleum into Chapter 11 bankruptcy with a proposal that would wipe out 97% of the equity in the company. According to the Board of Directors of the Whiting Petroleum Company, these bonuses were “intended to ensure the stability and continuity of the company’s workforce and eliminate any potential misalignment of interests that would likely arise if existing performance metrics were retained.” If you are a layperson, this explanation may sound to you like a very large crude carrier full of horseshit. I understand why you might think that. But let me assure you as a non-layperson that this explanation is an ultra large crude carrier full of horseshit.

      Disgusting

    1. The importance of self-compassion in tempering the brittleness of self-efficacy

      The main reason is that most people’s risk tolerance is very low, because self-efficacy (defined as “a person’s conviction or confidence about his or her abilities to mobilize the motivation, cognitive resources or courses of action needed to successfully execute a specific task within a given context”) is remarkably fragile. When it comes to trying and learning new things, people have difficulty transferring success in one arena to even highly related ones. Even small failures lead to learned helplessness so quickly, we learn to protect against that eventuality by not trying new things unless success is guaranteed.

      The primary risk of entrepreneurship and other free agent lifestyles is not financial or even social — it is the risk to a person’s very self-concept as someone who does what they set out to do.

      What we need if we want to change behavior at this fundamental level is to replace predictive models of behavior change—do this and you’ll get that —with exploratory models.

      Stories may actually be a more accurate way of describing how people think about and use mental models of behavior change. Stories, like emergent systems, only move in one direction. They cannot be rolled back and played again. This irreproducibility suggests the importance of another form of psychological capital that is also highly correlated with successful behavior change: self-compassion. They are two sides to the same coin — you need self-efficacy to believe you can do it, but you equally need self-compassion to be ok when you don’t. Self-compassion aids change by removing the veil of shame and pain that keeps you from examining the causes of your mistakes (and often, leads you to indulge in the very same bad habit as a way of forgetting the pain). Self-forgiveness is the first step in fostering an invitational attitude that is open to feedback and learning, from yourself and others.

      There is something about the turning of this coin — between efficacy and compassion — that I believe lies at the heart of the experimentation framework I’m envisioning. And the more I think about it, the more I suspect compassion is the far more radical and important side.

    1. Taken from a graduation address delivered at West Point, which is just so good and worth quoting many sections of at length

      That’s really the great mystery about bureaucracies. Why is it so often that the best people are stuck in the middle and the people who are running things—the leaders—are the mediocrities? Because excellence isn’t usually what gets you up the greasy pole. What gets you up is a talent for maneuvering. Kissing up to the people above you, kicking down to the people below you. Pleasing your teachers, pleasing your superiors, picking a powerful mentor and riding his coattails until it’s time to stab him in the back. Jumping through hoops. Getting along by going along. Being whatever other people want you to be, so that it finally comes to seem that, like the manager of the Central Station, you have nothing inside you at all. Not taking stupid risks like trying to change how things are done or question why they’re done. Just keeping the routine going.

      We have a crisis of leadership in America because our overwhelming power and wealth, earned under earlier generations of leaders, made us complacent, and for too long we have been training leaders who only know how to keep the routine going. Who can answer questions, but don’t know how to ask them. Who can fulfill goals, but don’t know how to set them. Who think about how to get things done, but not whether they’re worth doing in the first place. What we have now are the greatest technocrats the world has ever seen, people who have been trained to be incredibly good at one specific thing, but who have no interest in anything beyond their area of exper­tise. What we don’t have are leaders.

      What we don’t have, in other words, are thinkers. People who can think for themselves. People who can formulate a new direction: for the country, for a corporation or a college, for the Army—a new way of doing things, a new way of looking at things. People, in other words, with vision.

      That’s the first half of the lecture: the idea that true leadership means being able to think for yourself and act on your convictions. But how do you learn to do that? How do you learn to think? Let’s start with how you don’t learn to think. A study by a team of researchers at Stanford came out a couple of months ago. The investigators wanted to figure out how today’s college students were able to multitask so much more effectively than adults. How do they manage to do it, the researchers asked? The answer, they discovered—and this is by no means what they expected—is that they don’t. The enhanced cognitive abilities the investigators expected to find, the mental faculties that enable people to multitask effectively, were simply not there. In other words, people do not multitask effectively. And here’s the really surprising finding: the more people multitask, the worse they are, not just at other mental abilities, but at multitasking itself.

      One thing that made the study different from others is that the researchers didn’t test people’s cognitive functions while they were multitasking. They separated the subject group into high multitaskers and low multitaskers and used a different set of tests to measure the kinds of cognitive abilities involved in multitasking. They found that in every case the high multitaskers scored worse. They were worse at distinguishing between relevant and irrelevant information and ignoring the latter. In other words, they were more distractible. They were worse at what you might call “mental filing”: keeping information in the right conceptual boxes and being able to retrieve it quickly. In other words, their minds were more disorganized. And they were even worse at the very thing that defines multitasking itself: switching between tasks.

      Concentrating, focusing. You can just as easily consider this lecture to be about concentration as about solitude. Think about what the word means. It means gathering yourself together into a single point rather than letting yourself be dispersed everywhere into a cloud of electronic and social input. It seems to me that Facebook and Twitter and YouTube—and just so you don’t think this is a generational thing, TV and radio and magazines and even newspapers, too—are all ultimately just an elaborate excuse to run away from yourself. To avoid the difficult and troubling questions that being human throws in your way. Am I doing the right thing with my life? Do I believe the things I was taught as a child? What do the words I live by—words like duty, honor, and country—really mean? Am I happy?

      So it’s perfectly natural to have doubts, or questions, or even just difficulties. The question is, what do you do with them? Do you suppress them, do you distract yourself from them, do you pretend they don’t exist? Or do you confront them directly, honestly, courageously? If you decide to do so, you will find that the answers to these dilemmas are not to be found on Twitter or Comedy Central or even in The New York Times. They can only be found within—without distractions, without peer pressure, in solitude.

      “Your own reality—for yourself, not for others.” Thinking for yourself means finding yourself, finding your own reality. Here’s the other problem with Facebook and Twitter and even The New York Times. When you expose yourself to those things, especially in the constant way that people do now—older people as well as younger people—you are continuously bombarding yourself with a stream of other people’s thoughts. You are marinating yourself in the conventional wisdom. In other people’s reality: for others, not for yourself. You are creating a cacophony in which it is impossible to hear your own voice, whether it’s yourself you’re thinking about or anything else. That’s what Emerson meant when he said that “he who should inspire and lead his race must be defended from travelling with the souls of other men, from living, breathing, reading, and writing in the daily, time-worn yoke of their opinions.” Notice that he uses the word lead. Leadership means finding a new direction, not simply putting yourself at the front of the herd that’s heading toward the cliff.

      So solitude can mean introspection, it can mean the concentration of focused work, and it can mean sustained reading. All of these help you to know yourself better. But there’s one more thing I’m going to include as a form of solitude, and it will seem counterintuitive: friendship. Of course friendship is the opposite of solitude; it means being with other people. But I’m talking about one kind of friendship in particular, the deep friendship of intimate conversation. Long, uninterrupted talk with one other person. Not Skyping with three people and texting with two others at the same time while you hang out in a friend’s room listening to music and studying. That’s what Emerson meant when he said that “the soul environs itself with friends, that it may enter into a grander self-acquaintance or solitude.”

      Introspection means talking to yourself, and one of the best ways of talking to yourself is by talking to another person. One other person you can trust, one other person to whom you can unfold your soul. One other person you feel safe enough with to allow you to acknowledge things—to acknowledge things to yourself—that you otherwise can’t. Doubts you aren’t supposed to have, questions you aren’t supposed to ask. Feelings or opinions that would get you laughed at by the group or reprimanded by the authorities.

      This is what we call thinking out loud, discovering what you believe in the course of articulating it. But it takes just as much time and just as much patience as solitude in the strict sense. And our new electronic world has disrupted it just as violently. Instead of having one or two true friends that we can sit and talk to for three hours at a time, we have 968 “friends” that we never actually talk to; instead we just bounce one-line messages off them a hundred times a day. This is not friendship, this is distraction.

    1. Casca Indeed he is not fit. Decius Brutus Shall no man else be touched but only Caesar? Cassius CassiusThen leave him out.CascaIndeed, he's not a good fit.Decius BrutusAre we just going after Caesar?CassiusGood question Decius. I don't think it's a good idea for Mark Antony, who is so beloved by Caesar, should be left alive. You'll find him a cunning strategist, and if he builds up his power, it may grow so much as to harm all of us. To prevent this, let's kill Caesar and Antony together.Decius, well urged.  I think it is not meet, Mark Antony, so well beloved of Caesar, Should outlive Caesar.  We shall find of him A shrewd contriver; and, you know, his means, If he improve them, may well stretch so far As to annoy us all; which to prevent, Let Antony and Caesar fall together.

      When I was reading this I was using the word bubble whenever I needed it. This was one of the moments that I used it to help me understand what this was saying and that it was telling me that Cassius , Casca, and Brutus were going after Caesar and to also kill Antony when they killed Caesar.

    2. Then, lest he may, prevent. And since the quarrel Will bear no color for the thing he is, Fashion it thus: that what he is, augmented, Would run to these and these extremities; And therefore think him as a serpent's egg — (Brutus)Then, in case he is, we should prevent his ascent. Since this accusation would not be supported by how he is now, the argument against him should be fashioned in this way: given what he is now, with the addition of the power of a crown, will lead to excessive behavior. Therefore, think of him as a snake's egg, which, while not dangerous as it is, will inevitably become deadly as all snakes are. So it would be better to kill him in the shell.[Re-enter Lucius]LuciusThe candle's been lit in your study, sir. While I was looking on the windowsill for a piece of flint, I found this paper sealed up like this, and I'm positive it wasn't there when I went to bed.[Gives him the letter]Which, hatched, would, as his kind, grow mischievous — And kill him in the shell.

      i understand now that they think that Julius Caesar will turn on them all, and be like a snake, which as an egg is harmless, but it can be harmful once it gets out of its shell.

    3. Decius, well urged.  I think it is not meet, Mark Antony, so well beloved of Caesar, Should outlive Caesar.  We shall find of him A shrewd contriver; and, you know, his means, If he improve them, may well stretch so far As to annoy us all; which to prevent, Let Antony and Caesar fall together.

      The word bubble on the left helped me understand this a whole lot more. When I first read it, it seemed that they really liked Antony and were going to try to get him on board with the plan but if he disagreed they'd kill him. When reading the further in context, they more feared him and were worried that if he were alive he'd know about the plan to kill caesar so if they were to kill caesar they have to kill Mark Antony too.

    1. Note: This rebuttal was posted by the corresponding author to Review Commons. Content has not been altered except for formatting.

      Learn more at Review Commons


      Reply to the reviewers

      Reviewer #1:

      **Summary**

      Jang et al., address the important question of spatially localized or compartmentalized metabolic enzymes with a focus on the glycolytic enzyme PFK1. Using a good strategy of inserting a fluorescent tag at the endogenous PFK1 locus with tissue-specific inducible expression in C. elegans, combined with strong quantitative longitudinal imaging and innovative bioengineered microfluidic-hydrogels to control oxygen availability as well as optogenetic approaches, they show PFK1 condensates, which are not stress granules and not seen in normoxia, assemble with hypoxia. PFK1 condensates are dynamic, reversible, localized at the synapse in neurons, and recruit aldolase, another glycolytic enzyme. Although glycolytic proteins were previously shown to compartmentalize near the plasma membrane, and PFK1 was previously shown to assemble into filaments in vitro and be punctate at the plasma membrane in mammalian cells, evidence for cellular localized PFK1 condensates in animals is highly significant. The work includes strong biophysical characterization of PFK1 phase-separated condensates, but no clear indication of the composition of condensates. More significantly, the findings lack functional significance related to PFK1 activity or glycolytic flux with hypoxia vs normoxia. Despite previous work by this group showing that disrupting subcellular localization of glycolytic enzymes impairs neuronal activity in response with hypoxia, the reader is left with questions on the importance of localized and PFK1 condensates and their make-up .

      **Major comments:**

      Key conclusions are convincing, and most experimental approaches, biophysical characterization including thermodynamic principles, and data analysis are exemplary and well described. However, as indicated above, the work is limited to a descriptive analysis of cellular localization of PFK1 condensates and their biophysical properties without insights on functional significance relative to enzyme activity - or at least glycolytic flux or metabolic reprogramming with hypoxia. At best, only correlations can be drawn from hypoxia-induced localized PFK1 condensates and the authors' previous report (Jang et al., 2016) on hypoxia-regulated neuronal activity. Some insight or at least prediction in the discussion on the differences in spatially localized PFK1 in muscle vs neurons with regard to metabolic or energy distinctions should be included.

      We have added additional discussions on the differences of the spatially localized PFK-1.1 in muscles versus neurons, explaining that in both tissues the cellular enrichment appears to be at sites predicted to have high ATP consumption (lines 128-133; 482-484).

      Despite the strong biophysical analysis of condensates, several important features are not determined. First is at best a rudimentary analysis of the composition of condensates and also how PFK1 is assembled into these structures. For the former, is the core of the condensate predominantly PFK1 with perhaps aldolase only recruited to the periphery or is aldolase an integral component of the structure. Hence, is it a PFK1 condensate or a glycolytic condensate? For the latter question, is there a particular orientation for PFK1 in condensates, i.e a collection of filaments as previously reported, which might provide insight on assembly? Finally, and less critical but also important is the criterion for spherical, which is not well defined, and at least some idea or speculation on determinants for a spherical morphology - compared with filaments that have been reported for other non-glycolytic metabolic enzymes.

      We have now co-expressed PFK-1.1 and ALDO-1 and examined their dynamic formation during hypoxic conditions. We observe PFK-1.1 and ALDO-1 form condensates simultaneously, with gradual enrichment of both molecules. We now include this new data in Figure 7E and Video 8; lines 422-441, 964-989). We also include genetic data demonstrating the ALDO-1 requires pfk-1.1 to form condensates, and that PFK-1.1 requires aldo-1 as well. Therefore, the enzymes are interdependent on each other to form condensates (Figures 7G, 7H, S7B, and S7C).

      The spheroid geometry reflects liquid-like properties, which arises from surface tension of molecules loosely held together via multi-valent interactions. Filamentous arrangements reflect crystalline-like structures resulting from more stable interactions between molecules into solid-like states. While we did not perform high resolution studies, like Cryo-EM, to resolve this question, the spheroid geometry of PFK-1.1 condensates, along with its fluid-like properties, suggest the condensates are liquid-like compartment distinct to filamentous structures. We now add this discussion in lines 467-470.

      The work is an important advance in our understanding on the self-assembly of metabolic enzymes by showing hypoxia-induced PFK1 condensates in vivo, their spatially-restricted subcellular localization in muscle cells and neurons, and their biophysical properties, the latter being distinct from those of stress granules. Taken together, these findings are more extensive than many previous reports on the assembly of metabolic enzymes into filaments or condensates, but fall short for new insights on functional significance.

      We focus this study on the biophysical characterization of the condensates, and how that results in compartmentalized enrichment of glycolytic proteins. Examination of the functional significance of the phase separation to the enzymatic reactions in vivo is not currently possible because we lack probes we can use in vivo to measure the metabolites resulting from the reaction. We have now added discussion acknowledging this and framing its significance in the context of what has been published in the field (lines 484-492). For example, a recent manuscript in ChemRxiv demonstrated, in vitro, that the enzymatic activity of glycolytic proteins, hexokinase and glucose-6 phosphate dehydrogenase, promote these enzymes condensing into liquid droplets. The authors further found that the condensation accelerated the glycolytic reactions (Ura et al., 2020). This raises the question whether glycolytic proteins compartmentalize, and form condensates, in vivo, which we address in this manuscript. We capture this point in (lines 444-464) where we explain that, while it has long been hypothesized that glycolytic proteins like PFK-1 could be compartmentalized, this remained controversial due to lack of dynamic in vivo imaging. In our study, and through a systematic examination of endogenous PFK-1.1 via the use of a hybrid microfluidic-hydrogel device, we conclusively determine that PFK-1.1 indeed displays distinct patterns of subcellular localization in specific tissues in vivo.

      Reviewer #2 (Evidence, reproducibility and clarity (Required)):

      This paper reports on the condensation of the glycolytic enzyme PFK-1 in response to hypoxic conditions in neurons of C. elegans. The authors employ a microfluidic-hydrogel device to dynamically monitor the relocalisation of PFK-1 from a mostly diffuse state to clusters in response to hypoxia and show that PFK-1 can undergo multiple rounds of PFK-1 clustering and dissolution. The authors work through the key features of a liquid-like compartment (sphericity, fusion, fast internal rearrangements) and give evidence that PFK-1 may have all three. Finally, the authors tag PFK-1 with the light-inducible multimerization domain Cry2 and find that even without light PFK-1 will constitutively form clusters that sequestrate endogenous PFK-1 as well as other glycolytic proteins. The strength of this work is that it is characterizing what appears to likely be phase separation in the context of a whole animal experiencing a stress that it could encounter in the natural world. A limitation of the work is that it is unclear what the functional implications are of condensates of PFK-1 at the molecular or cell scale.

      **Major comments:**

      -All experiments were performed using fluorescently tagged PFK-1 expressed from endogenous promoter or from the native genetic locus which is important for excluding overexpression artifacts. However, there is still risk that the GFP tag is driving the assembly process. In order to exclude tag-specific effects that may cause aggregation of the tetrameric PFK-1, ideally a control would be done in which PFK-1 is visualized through immunofluorescence experiments of WT cells. Alternatively, a short tag (e.g HA, His) as epitope for is an alternative .

      We used fluorescent tags to observe the dynamic relocalization in vivo. While in the study we have not performed immunofluorescence, we established the validity of the labeling method by: 1) using monomeric versions of GFP; 2) using different fluorophores to show the same condensation phenomenon; 3) performing CRISPR for single copy insertions; 4) Demonstrating that different glycolytic proteins form condensates; 5) demonstrating the GFP-tagged versions of the protein are capable of rescuing the loss-of-function alleles and 6) Now adding new data demonstrating the observed localization specifically depend on the presence of other glycolytic proteins. This last result supports that GFP tag is not driving the assembly process of glycolytic condensate and that the glycolytic condensate formation requires the presence of specific molecules in the pathway. I add that we routinely use fluorophore markers to over a dozen distinct proteins that label subcellular compartments, and we have never observed the dynamic relocalization reported here, with the exception of other glycolytic proteins that interact with PFK, suggesting this is a property specific to glycolytic proteins, and, based on the genetic studies, dependent on the glycolytic reaction. We add and discuss these findings in Figures 7G, 7H, S7B, and S7C; lines 422-441, 964-989.

      -For the Cry2-section, the complementation of the pfk-1 mutant supports functionality of the synaptic clustering phenotype. Are there other features of function that can be evaluated or could you look at how Cry-2 vs wt worms recover from different durations of stress or frequencies. Could you see if the Cry-2-fusion will rescue function to a partial-loss-of-function allele or a tetramerization deficient allele? A detailed analysis of the effects of constitutive presence of PFK-1-Cry2 clusters would be necessary to bolster claims that this is fully functional construct. Can enzyme activity be somehow monitored?

      We did not observe any difference between wild-type worms and CRY2-expressing worms with regards to their development, survival, locomotive behavior or synaptic phenotype. While we can not discard the possibility that this is not a full rescue, with available tools, we can not distinguish the recue with PFK-1-Cry2 from that of just PFK-1.

      -The analysis of the sphericity of clusters (4A) is limited due to the diffraction limit of light which limits an analysis of a compartment of this size. While this is a limitation of the live organism, this should be more clearly acknowledged.

      We have included in the Methods section our criteria for quantifying condensates and avoiding diffraction limit artifacts. Briefly, “Considering the resolution limit of a spinning disc confocal (approximately 300nm), any structure with a diameter less than 500nm and an area smaller than 0.2 µm2 was excluded from the analyses”. To better clarify this point, we also now add a description of the criteria used in the main text (lines 242-243).

      In addition, we observed that PFK-1.1 condensates are not perfect spheres, but constrained spheroids (which can not be explained by diffraction-limited point spread functions). We can explain the observed spheroid shapes based on liquid-like properties of the condensates, and the constrains of the diameter of the neurite. To better highlight this finding, we have now moved Figure S4E into the main figure (Figure 4B’).

      -Fusion experiments (4C) do not fully exclude that clusters overlap instead of merging. It would be beneficial to show the foci for several subsequent frames. One would expect that upon fusion, the condensate size would increase, but video 3 suggests the opposite. It would be useful to quantify condensate size before and after fusion for several separate fusion events. -an alternative possible experiment would be the tagging of PFK-1 with a photoconvertible fluorophore (e.g. Dendra2) and subsequent analysis of fusion events

      To better show the fusion events in Figure 4C, we now include all xy, yz, and zx plane views of before and after fusion events of Figure 4C (Figure S5B). We also added a quantification of four independent fusion events in which we compare the sum of the areas of the two puncta before fusion and the size of the area of the single punctum after fusion (Figure S5C). These data support that we are observing fusions events.

      -4D). It is unclear if foci are indeed undergoing fission or if two clusters next to each other are moving apart.

      For Figure 4D, in all the frames we had recorded, a single structure maintains a continuous signal until fission occurs and splits into two structures. To better present this event, we now include an unabridged version of figure of 4D in the supplement that shows all the frames captured (Figure S5D).

      -The analysis of side-by-side growth and dissolution kinetics are interesting and a novel view into the non-equilibrium aspects of phase separation in cells.

      -Purification of PFK-1 and in vitro reconstitution of condensates would be supportive of liquid-like characteristics although I don't think it is necessary however it would add a lot to the relevance to show enzyme activity is different +/- condensate state but I am not sure if an easy enzymatic assay exists in vitro.

      We agree. But the significance of this particular paper, specifically in the context of the in vitro enzymatic work on glycolytic proteins, is to examine the dynamic in vivo localization and the biophysical characteristics of the condensates. To better underscore this in the context of the field, we add discussion of a recent in vitro manuscript demonstrating that liquid droplet formation of glycolytic proteins affect their enzymatic activity (Ura et al., 2020) (lines 444-464; 484-492). While we see the value of future studies reconstituting the glycolytic particles, we believe that is beyond the scope of this particular in vivo study.

      **Minor comments:**

      -Stress granules in other organisms (yeast paper) have different composition depending on stress type. To make the claim that the PFK-1 compartments are independent of SGs one would ideally test multiple different SG markers.

      We selected the stress granule protein TIAR-1 because it is one of the most studied stress granule markers in C. elegans and it is reportedly one of the core proteins and universal components of stress granules irrespective of a stress type (Buchan et al., 2011; Gilks et al., 2004; Huelgas-Morales et al., 2016; Kedersha et al., 1999). Although we did not include images in the manuscript, we had tested a total of three stress granule markers: TIAR-1, TDP-43, and G3BP1 with similar results. We now added that as data not shown (lines 193-194).

      -it should be stated in the main text that the microfluidic-hydrogel device was fabricated following previously published protocols

      We have added the reference in the main text (line 170) to supplement what we had written in the Methods section: “A reusable microfluidic PDMS device was fabricated to deliver gases through a channel adjacent to immobilized animals, following protocols as previously described (Lagoy and Albrecht, 2015)”.

      -Figure 4b: Y-axis should be changed from probability to fraction of occurrence

      We have corrected this in both the figure and the figure legends (Figure 4B).

      -The discussion should be less speculative concerning any effects seen in PFK1-Cry2 expressing C. elegans

      We have modified the discussion as suggested.

      -it is perplexing that a protein known to tetramerize with no disordered or RNA-binding domains forms condensates like this. Is there anything known from other systems of additional interacting proteins that may have features that promote liquidity and serve to fluidize these assemblies?

      Condensates can form via multivalent interactions, which include, but is not exclusive, to disordered or RNA-binding domains. Because glycolytic proteins have dihedral symmetries that can facilitate multivalent interactions, we believe these structural properties, in combination with regulated conformational changes, promote multivalent interactions leading to their condensation. We had a statement in the discussion (lines 494-519) now add this more clearly in the results (lines 395-398).

      Reviewer #2 (Significance (Required)):

      Stimulus-induced phase separation has been observed for dozens of metabolic enzymes from various different pathways (reviewed in Prouteau, 2018). Several studies have published the formation of condensates through PFK-1 in diverse organisms (C. elegans, Yeast, human cancer cells) in response to hypoxia or in some cancer lines also without hypoxia (Jin, 2017, Jang, 2016, Kohnhorst 2017, etc.). A yeast study showed that PFK-1 condensates contain various other glycolytic enzymes and that condensate formation enhances glycolytic rates (Jin, 2017).

      This study gives the advance of analyzing the dynamics of PFK-1 condensate formation in vivo in the context of a live animal using a microfluidic-hydrogel device and showing that PFK-1 relocalizes to reversible condensates within minutes of hypoxia. If further appropriate experiments (as mentioned above) are performed, this study would strongly suggest that the underlying process of PFK-1 condensate formation is liquid-liquid phase separation. Ideally, if at all feasible, it would be strengthened if there was some insight into the functional consequences of the condensed assemblies formed in hypoxia. These findings may be interesting to researchers working on glycolysis and metabolism in different cells but particularly in neurons.

      Field of expertise

      -Phase separation, microscopy, in vitro reconstitution

      -no experience with C. elegans biology and do not have a practical handle on ease or difficulties of genetic manipulation of C. elegans or metabolic assays for PFK-1

      Reviewer #3 (Evidence, reproducibility and clarity (Required)):

      **Summary:**

      In this manuscript, the authors focus on the subcellular localization of the key glycolytic enzyme PFK-1.1 in C. elegans, initially in whole animals through GFP tagging of the endogenous locus and subsequently in single cells/tissues using a clever genome editing strategy that permitted tissue-specific expression of GFP-tagged PFK-1.1 from its endogenous locus. They observe that PFK-1.1 localization differs from cell-type to cell-type and can be dynamically reorganized in response to exogenous cues. Focusing on hypoxia, they observe that PFK-1.1 forms foci near synapses in neurons under this stress condition. These foci are not stress granules and they are dissolved upon re-oxygenation. These condensates have properties of liquid droplets and can mature (harden) over time. PFK-1.1 fused to the CRY domain can trigger condensate formation under normoxic conditions, which can co-recruit WT PFK-1.1 as well as aldolase.

      **Major comments:**

      The conclusions are convincing but the impact could be increased if the authors were able to demonstrate the physiological role that the observed phase separation plays in this stress response. Would it be possible to assess glycolytic flux under hypoxia vs normoxia?

      It is currently not possible to assess glycolytic flux in vivo in our system, as we lack metabolic sensors (an active area of work we are trying to address, but will take several years to perform correctly). We have added discussion of new in vitro studies examining the consequences of metabolic flux due to glycolytic compartmentation into liquid droplets (Ura et al., 2020), and the significance of those findings in the context of our in vivo studies (lines 444-464; 484-492).

      The authors should comment on viability during the hypoxia time course.

      C. elegans can survive anoxic condition for a day (Powell-Coffman, 2010). Our hypoxic conditions last minutes, and we can rescue live C. elegans upon completion of the assays. We now include a description of this in the Methods (lines 1216-1218).

      The co-clustering of ALDO-1 and PFK-1.1::mCh::CRY2 in Figure 7 should be properly quantified/statistically analyzed

      We quantified the fraction of animals that displays ALDO-1 clustering in PFK-1.1::mCh::CRY2 co-expressing animals, as suggested (Figure S7C).

      A control of mCh::CRY2 + ALDO-1::EGFP is missing from the experiments shown in Figure 7. Is the presence of mCh::CRY2 sufficient to drive ALDO-1::EGFP clustering?

      As a control for the CRY2 tag promoting the formation of glycolytic condensates, we had co-expressed mCh::CRY2 with PFK-1.1::EGFP, which is insufficient to cause the formation of the condensate (Figure 7C). We have now added a new data where we show that in pfk-1.1 deletion mutants, ALDO-1 condensate formation is suppressed, which further demonstrates the dependency between PFK-1.1 and ALDO-1 (Figures 7H and S7C).

      Does hypoxia trigger co-clustering of ALDO-1 and PFK-1.1?

      To answer this question, we examined the dynamic formation of ALDO-1 and PFK-1.1 condensates by co-expressing the two proteins together and observed that hypoxia triggers their co-clustering. We now include this in Figure 7E and Video 8.

      The authors speculate that hypoxia acts via diminished energy (altered ATP AMP ratios). Can this be measured? To support this hypothesis, the authors may wish to test if similar phase separation is triggered by mitochondrial poisons.

      We currently lack sensors that can reliably measure, in vivo, the subcellular changes in energy or metabolic flux in C. elegans neurons. However, we previously did test mitochondrial mutants and observed that in those mutants we observe glycolytic condensates (Jang et al., 2016), supporting the idea that defects in energy production promotes the formation of glycolytic condensates.

      **Minor comments:** Is 21% O2 not hyperoxic for worms?

      While C. elegans are known to prefer lower percentage of oxygen than those in air, in the lab animals are reared in normal air. We therefore used 21% oxygen present in air as our normoxic conditions.

      Can the authors speculate more on how do these condensates exhibit "memory" (how they're able to cluster in the same place repeatedly)? Is there any role for the cytoskeleton in mediating nucleation and/or condensation of PFK and glycolytic enzymes?

      When we were testing the reversibility of PFK-1.1 condensates, we were not expecting the reappearance of PFK-1.1 condensates in the same place repeatedly. Our current speculation is that, because many glycolytic enzymes, such as PFK-1.1, are allosterically regulated by nucleotides, AMP/ATP ratio may play a role on where glycolytic condensates appear. In other words, the specific synaptic areas, where PFK-1.1 condensate repeatedly reappeared, may have different AMP/ATP ratio that may trigger the condensation of the glycolytic proteins in those locationsupon conformational changes in PFK-1. We can’t exclude, currently, the presence of nucleating factors at synapses that facilitate PFK-1 clustering, but we have not yet identified them. We now include a discussion of this (lines 494-519).

      Do the authors think that these clusters are effectively G-bodies from yeast?

      G-bodies from yeast also shows glycolytic proteins changing from its diffuse localization to punctate localization in response to hypoxia (Jin et al., 2017). G-bodies, like C. elegans glycolytic condensates, are forms of subcellular glycolytic organization within eukaryotic cells. Yet, G-bodies take 24 hours to form, while we observe the glycolytic clusters in C. elegans within minutes of hypoxic conditions. We will need to understand the composition and function of both to determine if these forms of glycolytic subcellular organization represent the same structure. We note that glycolytic clusters have also been observed in some human cancer cell lines (Kohnhorst et al., 2017). Observation of glycolytic compartments in multiple different species and cell types suggest that, although the regulation, composition and formation kinetics of the glycolytic condensates may differ, compartmentalization of glycolytic enzymes may be a conserved feature. We now add a sentence discussing this (line 535-537).

      Reviewer #3 (Significance (Required)):

      It is much appreciated that this study tackles the cell biology of signaling and metabolism, which is a fascinating but difficult to study aspect of molecular biology. This work conclusively documents the dynamic reorganization of metabolic enzymes in vivo in response to physiological stimuli. Such reorganization had been proposed previously but was controversial and difficult to study in a controlled way. This work not only confirms previous observations but further demonstrates that the dynamic reorganization is mediated by a liquid-liquid phase separation. What is lacking is a demonstration that this phase separation is physiologically important. Such observations would generate interest from a much broader audience; the present audience presently targeting people specifically interested in non-membrane organelles per se. The reviewer has expertise in cell signalling and its regulation by phase separation.

      As we explain for Reviewer 1, we focus this study on the biophysical characterization of the condensates, and how that results in compartmentalized enrichment of glycolytic proteins. Examination of the functional significance of the phase separation to the enzymatic reactions in vivo is not currently possible because we lack probes we can use in vivo to measure the metabolites resulting from the reaction. We have now added discussion acknowledging this and framing its significance in the context of what has been published in the field (lines 484-492). For example, a recent manuscript in ChemRxiv demonstrated, in vitro, that the enzymatic activity of glycolytic proteins, hexokinase and glucose-6 phosphate dehydrogenase, promote these enzymes condensing into liquid droplets. The authors further found that the condensation accelerated the glycolytic reactions (Ura et al., 2020). This raises the question whether glycolytic proteins compartmentalize, and form condensates, in vivo, which we address in this manuscript. We capture this point in (lines 444-464) where we explain that, while it has long been hypothesized that glycolytic proteins like PFK-1 could be compartmentalized, this remained controversial due to lack of dynamic in vivo imaging. In our study, and through a systematic examination of endogenous PFK-1.1 via the use of a hybrid microfluidic-hydrogel device, we conclusively determine that PFK-1.1 indeed displays distinct patterns of subcellular localization in specific tissues in vivo.

      **REFEREES CROSS-COMMENTING** Globally it seems that all reviewers feel that impact would be increased if the physiological consequence of PFK-1.1 condensates was examined. Other, specific comments seem fair.

    1. Through mutations, these genes may produce proteins that cause a spectrum of inflammatory diseases such as ulcerative colitis and Crohn disease

      Crazy to think that diseases we have today were caused by mutations in evolution.

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      Reply to the reviewers

      Reviewer #1 (Evidence, reproducibility and clarity):

      Summary:

      In this work the authors investigate the dynamics of cell morphogenesis in a convenient in vivo system. They use the terminal cells of the embryonic tracheal system and comprehensively address how cell shape change (elongation in this case) takes place and how membrane is remodeled during the process. By combining different high-resolution techniques, i.e. in vivo imaging of terminal cells expressing different membrane markers and serial-section electron tomography, they describe the organelle organization (ER, Golgi, different types of vesicles) in the terminal cells during their elongation. They identify the presence of membrane structures/vesicles particularly abundant at the tip of the cell ahead of the growing tube. When they block endocytosis they find increased tube membrane and lack of basal membrane growth. In addition, in tomograms, they observe clear membrane defects, like invaginations that could even connect the tube and basal membranes. This correlates with the absence of vesicles at the tip observed in normal conditions.

      The analysis of the nature of the vesicles observed indicated the accumulation of late endosomes and MVB, particularly at the tip of the growing terminal cells. Interfering with the formation of these MVB led to defects in the growth of both the tube and the basal membrane.

      Altogether the authors propose a model in which the newly formed membrane (and transmembrane proteins) passes through the ER and Golgi and reaches the apical membrane. The incorporated membrane is then rapidly endocytosed and follows a maturation pathway through MVB, from where different cargoes and membrane would be sorted and recycled back to the apical (tube) membrane, or to the basal membrane through a transcytosis mechanism

      Major comments:

      Are the key conclusions convincing?

      Most of the conclusions presented are convincing and supported by the results observed.

      However, to my understanding, one of the key conclusions of this work (that membrane is transcytosed from the apical to the basal domain) is not fully convincing. A critical result to support the author's conclusion of apical-basal transcytosis is to find clear evidence of basal accumulation of a transcytosed marker.

      1. The authors show accumulation of FGFR-GFP and Myospheroid as evidence. However, I find the results presented not very convincing. The accumulation of FGFR-GFP at the basal membrane in the control is not very clear in the images and movie presented. In addition, in the shibire mutant, some basal accumulation of FGFR-GFP seems to be detected (particularly in the movie). In the figures the authors show an increase of FGFR-GFP intensity when endocytosis is blocked, but this is not explained in the text.

      If we understand the referee correctly, there are two parts to her/his concern:

      1. Are the proteins we use in fact present basally in normal tracheal cells, i.e. are they good candidates for transcytosed cargo?

      2.Do they change their localisation when endocytosis is disrupted? And this point can be divided into two aspects: a. do they change at the basal membrane? b. do they change at the apical membrane (this latter point is not questioned the referee)?

      1. The FGFR and beta-integrin are the only known basal markers in tracheal cells. A major reason for being confident of their presence in the basal membrane, even though they are difficult to visualize, is that the biological function of both is at the basal membrane, with the FGFR receiving growth or chemotactic signals from the surrounding tissue, and integrins anchoring the branches on the underlying tissue. However, it is indeed the case that their expression levels are very low, and it is difficult to visualize them, whether by expression of GFP-labelled constructs or by immunofluorescence. We have pushed to the limit a number of methods to improve the detection, but we seem to be constrained by the biology of these molecules.

      In addition to the low but detectable signal at the outer boundary cell, some signal is always visible within the cell, which we had in the past always interpreted as an artefact or background, but for which our findings here might provide alternative interpretations.

      1. a. We agree with the referee’s assessment that FGFR::GFP is still detectable in the basal membrane after blocking endocytosis. This is, in our view, no contradiction to our model. The most parsimonious interpretation is that this is the FGFR that had already been delivered basally before we interfered with endocytosis, and which remains there after endocytosis is blocked.

      b. In addition to this basal pool of FGFR, cells with blocked endocytosis accumulate abnormally high levels of FGFR at the apical membrane, in fact at much higher intensities than at the basal membrane. This is the more dramatic aspect of the phenotype, and our conclusions therefore rely not so much on a possible reduction of basal signal after blocking endocytosis (which would not be possible to demonstrate reliably), but rather on the abnormally enriched presence in the apical membrane.

      A technical point: The increase in FGFR::GFP on endocytosis blockage that we show in Figure S4 corresponds to the cytoplasmic + apical pool of the FGFR. We used the Dof signal in the cell to create a mask of the total cell volume to 3D-segment the FGFR signal. Therefore, this analysis does not take into account the FGFR that is present in the basal membrane. We had explained this only in the methods section but will now describe it more explicitly in the Results section.

      On the other hand, and very importantly, how does FGFR localization relates to its activity? The authors show that when endocytosis is prevented dpERK (i.e. a reporter of FGFR activation) is not decreased, indicating that FGFR is normally active. Wouldn't this suggest that FGFR is still localized basally to receive Bnl signal?

      Indeed, and this is also what we see. This is not in conflict with any of the results or known functions of the receptor. If endocytosis is blocked, FGFR cannot be internalized and removed from the basal membrane, where it is needed to receive the FGF from the surrounding cells.

      Our concern, and the reason for doing the experiment, had been that endocytosis might be required for FGF signaling, and that this might account for the failure of the cell to grow. But it turns out that our results show that this is not the case, at least not for the terminal cell in this time frame.

      Actually, as the authors indicate, in larval tracheal cells the intracellular accumulation of the FGFR leads to a reduced FGF signal transduction (Chanut-Delalande et al., 2010), suggesting that reduced FGF signaling activity in these cells is due to less FGFR reaching the basal membrane.

      That is true, and again, not inconsistent with our own results. In the cited study, trafficking was blocked at a step downstream of endocytosis. In this experimental situation, the internalisation of the FGFR would therefore occur as normal in these cells, but due to the impaired function of the ESCRT complex, intracellular processing, and therefore potential re-delivery to the basal membrane would be impaired. Furthermore, if (as we now propose) FGFR is also delivered via late endosomes in this context, blocking the ESCRT pathway should also impair initial FGFR delivery. In either way, initial delivery or re-delivery of the receptor being blocked, it is reasonable to assume that reduced signal transduction is the result of reduced basal FGFR.

      Thus in our study we see no reduction in basal FGFR and no reduction (and even an increase) in signaling, while Chanut et al see reduced basal FGFR and reduced signaling, and the reason for this is that they interfere with a different step of the membrane trafficking pathway.

      1. The results with Myospheroid are not very convincing either, as the authors just show a single confocal section of control and shibire mutants. In summary, I consider that this very important point needs to be better documented before concluding that apical membrane material containing basal cargoes is transcytosed to the basal membrane.

      We observed this phenotype in several instances. We will quantify it for the resubmission.

      1. Another conclusion that, to my opinion, should be better explained and documented, is the coordination of tube and basal membrane growth. Following the movements of the vesicles the authors conclude that there is a net displacement of these vesicles to the tip of the cell. This correlates with the presence of mature endosomes there. So the results postulate that transcytosis occurs at the tip, and therefore the growth of the basal membrane would occur preferentially at the tip. It has been demonstrated, as the authors indicate, that the tube membrane grows all along the length of the tube. How is then coordinated the tube and basal membrane growth? If, as the authors propose in their model, the tube membrane also grows after a process of endocytosis and recycling, wouldn't it be expected to have preferential tip growth? How do the authors reconcile all these observations with previously published results (Gervais and Casanova, 2010)?

      The elegant study by Gervais and Casanova (2010) used a very clever method, which was however entirely non- quantitative (and did not make any claims to the contrary, either). The conclusion that material is delivered to the tube throughout its length was based on looking at the displacement relative to the base and the tip of the cell of short and transient secondary branches (seen for example in Fig. 1H in our paper). If the tube would primarily receive material at the tip, these secondary branches would not change their position with respect to the base of the cell. Instead, these branches tend to be displaced towards the tip, which shows that material is also added between the branch and the base of the cell. These branches are seen only in a fraction of wild type terminal cells and quantification is therefore difficult. Thus, the experiment shows convincingly that material is also delivered behind the transient branch and excludes a model by which all growth occurs only at the tip. But it does not discuss what proportion of the total is delivered along the length vs the tip of the branch.

      Our model also does not contest the idea of ubiquitous membrane delivery over the length of the tube, either in the initial delivery step, or during redistribution. On the contrary, the presence along the length of the cell of vesicles carrying FYVE::GFP and Rab7, and of smaller MVB-like bodies in the EM sections, suggest that the pathway can also be deployed at a distance from the tip.

      1. The serial-section electron tomography analysis is very interesting and identifies different sorts of vesicles. However, it is very unclear what the different vesicles referred in the models correspond to (in the in vivo imaging for instance). For instance, the small granular or the dense-core vesicles correspond to endocytic vesicles at different stages of maturation?

      Based on distribution alone, it is almost impossible to determine which of these vesicles correspond to endosomes or to secretory vesicles, even for the extremely experienced EM experts in the team. We would require high resolution CLEM, and a wide range of fluorescent markers to be able to determine which population of vesicles found on EM correspond to each marker. Due to the broad distribution of these vesicles within the cell and their small sizes right now it would be extremely technically challenging to pursue this question (even though we too would love to know)

      1. If there is a constant endocytosis from the apical membrane to generate the basal and build the definitive apical membrane, wouldn't it be expected to find many more vesicles around the tube? Wouldn't it be expected to find coated vesicles around or budding from the tube, as the coated vesicles observed budding from the basal membrane in Fig 2D? Or is the endocytosis observed mediated by non-clathrin coated vesicles?

      We agree, and we had observed this to be the case, but had not quantified this. We have now analysed the distribution of coated pits and their density in the apical or the basal membrane of the cell. Overall, we found a higher density of endocytic events in the apical membrane than in the basal membrane. As the reviewer noticed, we also found that the majority of endocytic events in the basal membrane occur towards the tip of the cell. We will add these data to Figure S3.

      Should the authors qualify some of their claims as preliminary or speculative, or remove them altogether?

      The results of Serp accumulation upon MVB interference can lead to confusion (from line 404). The authors seem to suggest that Serp protein is exclusively produced in the FB and transported by transcytosis to reach the tracheal lumen. However, Serp is also produced in the tracheal cells themselves. In fact, serp expression in the FB seems to be detected by late embryogenesis, while expression in tracheal cells is detected much earlier (Dong et al. 2014; Luschnig et al. 2006; Wang et al. 2006). It was also shown that Serp undergoes a recycling mechanism from the lumen to the lumen, through the endosomes-TGN retrograde trafficking, that may also require Shrub (Dong et al, 2014; Dong et al 2013). Thus, it is unclear (and even unlikely) that the Serp found in the vesicles in Shrub-GFP mutants is derived exclusively from the transcytosed component from the FB. I suggest to better explore this issue or to remove this part.

      We agree that the notion that the Serp we see is exclusively derived from the fat body is not correct. We observed Serp accumulation around Shrb::GFP sites in embryos at early and late stages, so it is likely that what we see is the result both of apical-to-apical redelivery of Serp (as reported in Dong et al., 2014a), and transcytosis of Serp from the basal membrane to the apical. We will therefore rewrite this. But regardless of whether we are looking at transcytosis, or apical-to-apical cycling, this experiment still reinforces the idea of our work that late endosomes serve as stations that collect material from and re-deliver it towards various compartments in the cell.

      Would additional experiments be essential to support the claims of the paper? Request additional experiments only where necessary for the paper as it is, and do not ask authors to open new lines of experimentation.

      As indicated before, more conclusive results for transcytosis should be provided.

      I suggest that the authors determine the presence of apical and basal cargoes (FGFR) in the late endosomes found when Shrub activity is impaired. According to his model, both types of markers should accumulate there. A high accumulation of these markers in those endosomes would reinforce the hypothesis proposed.

      We agree with the reviewer that this would be a great experiment. Since we only have an FGFR tagged with GFP we cannot do this experiment using the Shrb::GFP line, so instead we would have to use shrb mutants. This in itself is not a problem (see also below – we will be adding some data), but the experiment would require multi-generation crosses which could only be started once the current Covid-19 restrictions are lifted and labs opened again. Instead, we propose to cite the following supporting evidence. Chanut-Delalande et al., (2010) showed that mutants for components of the ESCRT pathway hrs and stam show intracellular accumulation of overexpressed FGFR::GFP in tracheal cells of the air sac primordium, and Dong et al., (2014a) show that shrb mutants accumulate Crb in late endocytic compartments in tracheal cells of the dorsal trunks. We would suggest it is very likely that terminal cells therefore also accumulate Crb and FGFR in late endosomes in the absence of Shrb. An experiment would be nicer, but we fear this is the best we can do at the moment.

      While it has been reported that shrub-GFP act as a dominant negative in different contexts (Dong et al, 2014; Sweeney et al 2006) it is unclear why. So it would be desirable to confirm the results with a loss of function condition (either mutant or RNAi line).

      We will now add data on shrb mutants where we find a phenotype that is similar as in Shrb::GFP overexpression.

      Are the data and the methods presented in such a way that they can be reproduced?

      The materials and methods section would benefit from more detailed explanations.

      We agree.

      Are the experiments adequately replicated and statistical analysis adequate?

      Many of the experiments presented in this work are technically very challenging, like the in vivo analyses and particularly the serial-section electron tomography. This prevents having high numbers of replicates on occasions.

      Minor comments:

      1. In the abstract the authors state: "We show that apical endocytosis and late endosome-mediated trafficking determine the membrane allocation to the apical and basal membrane domains". I think that the authors show that "that apical endocytosis and late endosome-mediated trafficking is required for correct membrane growth", but I am not that sure that they show that it determines the membrane allocation

      We agree and will change this.

      1. References for the PH-GFP localization in cells should be provided. Which is the evidence that it only localizes to plasma membrane?

      Apical localization of PH-GFP is preferential rather than exclusive. The construct has nevertheless been widely used as an ‘apical’ marker, based on the fact that this PH domain binds to PIP2, which is enriched in the apical domain of epithelial cells (e.g. Pilot, et al., 2006; Román-Fernández, et al., 2018).

      1. It would be more adequate to always use the same terms to facilitate the reading. For instance, in several figures the membranes are referred as basal plasma membrane and tube membrane, but in others outer and inner membrane

      We will go through the entire manuscript and use a consistent nomenclature.

      1. Figure 3C,D and corresponding text are difficult to understand. The increase of fluorescence of the inner membrane seems to be very high, even higher than the corresponding to the outer membrane. Can the authors explain better this point and also describe better the method applied in the materials and methods section?

      Rather than absolute amounts the graphs show fold increase over the amount of membrane at the beginning of the recording. We had used this representation for two reasons. First, the overall signal intensity can vary from one imaging set to the next, so comparing and representing absolute amounts from different datasets is not easily possible. However, we understand now why the representation in our plots was confusing. We will now show in Figure 3D how the total amount of plasma membrane in shibirets cells increases in the same way as in controls. For Figure 3C, we have found a better way of representing what percentage of the total membrane is present in each compartment as the cell grows. We will rewrite this part to make it clearer, and we will describe more thoroughly in the methods section how the analysis was done.

      Significance:

      This work represents an important advance for the field for several reasons. First of all it represents a technical advance because the authors are able to combine the traditional genetic analysis with two powerful techniques (in vivo imaging and serial-section electron tomography) to analyze single cell behavior at high resolution (temporal and spatial). In addition it represents a conceptual advance as it proposes a mechanism through which membrane growth is coordinated to regulate cell morphogenesis. The mechanism presented (endocytosis and transcytosis) is not new but they find evidence in an in vivo system.

      It was previously known that tracheal terminal cells undergo a process of intracellular tube formation and cell elongation at the same time, but the mechanisms coordinating these two cell events were not known. The proposed mechanism may not only be relevant for the morphogenesis of tracheal terminal cells, but could represent a general mechanism of cell morphogenesis. Therefore, the paper should be relevant for research in the morphogenesis area but also in the cell biology field, as it shows how regulated membrane trafficking can control tissue morphogenesis

      REFEREES CROSS-COMMENTING:

      I agree with reviewer #4 on her/his comments and suggestions about analyzing the involvement of the recycling endosome in the process.

      Reviewer #2 (Evidence, reproducibility and clarity):

      In their manuscript, Mathew et al present a model in which transcytosis is utilized to deliver endocytosed apical membranes to supply basal membrane growth. The authors examined the developing terminal cell of the fruitfly tracheal system, which is a well established tubulogenesis model, as these cells form subcellular tubes by apical plasma membrane invagination. The authors show that basal membrane growth stops when endocytosis or endosomal transport is blocked, while the apical membrane grows excessively or membrane material accumulates in the cytosol, respectively.

      Significance:

      The authors used high-end microscopy (including CLEM and electron tomography) to support their model and in my opinion, the quality and the quantity of the presented data are indeed adequate for this. The text is well written, the figures are of superb quality, and several cartoons help to understand the presented data/experiments. Therefore I highly recommend submitting the manuscript to a cell biology journal in its present form.

      Reviewer #3 (Evidence, reproducibility and clarity):

      This paper investigates the role of membrane trafficking in growth of the polarized tracheal cell that forms a cellular projection containing a subcellular tube. The authors show that apical endocytosis and late endosome-mediated trafficking determine the membrane allocation to the apical and basal membrane domains. Basal plasma membrane growth stops if endocytosis is blocked, whereas the apical membrane grows excessively. Plasma membrane is initially delivered apically, and then appears to be continuously endocytosed, together with apical and basal cargo. The sorting and recycling of apical and basolateral membrane appears to occur in a novel organelle carrying markers of late endosomes and multivesicular bodies (MVBs). Inhibiting endocytosis eliminates this compartment.

      The work in this paper is generally of high quality, and with one exception, quite comprehensive. The writing and figures are clear.

      Major concerns:

      1. A central focus of the paper is that balance between apical and basal membrane and the role of transcytosis in moving membrane from the apical compartment to the basolateral compartment. The current view is that transcytosis in mammalian cells usually goes through the recycling endosomes which are marked by rab11, although there is evidence for some trafficking through MVBs as well. In Drosophila, Rab11 positive recycling endosomes are frequently examined as part of endocytic system analyses. However, rab11 is not used as a marker in this paper and indeed there is no mention at all of recycling endosomes, even though recycling is at the core of the work. Since the authors do not examine rab11 or other possible markers of recycling endosomes, it is unclear whether the organelle they identify as carrying markers of late endosome and MVBs is some MVB/recycling endosome hybrid, or whether the organelle is completely distinct from the recycling endosome. Consequently, it is not possible to assess whether the observed trafficking either uses or does not use involve the recycling endosome. This ambiguity make is difficult to relate the observed trafficking to other systems. Minimally the authors should stain for rab-11 in WT and in some of the conditions where trafficking is perturbed and determine if the MVB-like compartment they are observing is rab11 positive, and whether the recycling endosome are affected by the perturbations. Further experiments may be needed to resolve whether any trafficking is going via the recycling endosome or this new MVB-type structure, but without even preliminary data on the relationship between the MVB compartment and the recycling endosome, its hard to say what might be appropriate or exactly how long addressing this will take. But just staining for rab11 in WT and a few mutant conditions to get a handle on what is up with the recycling endosomes in these cells should take less than a month.

      We had done a number of experiments on Rab11 but did not include them because we felt they did not add any crucial insights on the mechanism we describe here. However, as the reviewer rightfully points out, Rab11 is a classical marker for recycling and transcytosis and we agree that the reader should know our results. We found that overexpressed Rab11::GFP as well as endogenously tagged Rab11:YFP are both highly enriched around the tube. Unlike Rab5 which is seen in widely spaced discrete vesicles, Rab11 forms a cloud of small puncta. We found very low overlap of Rab11 with CD4::mIFP-positive vesicles at the tip of the cell. This suggests that Rab11 is unlikely to be directly involved in the transcytosis pathway we describe here.

      Loss of Rab11 was harder to analyse; Rab11-RNAi did not show any obvious phenotype, which could be due to high maternal contribution or to low knockdown efficiency, none of which we analysed in detail. Expression of a dominant negative Rab11 resulted in very early defects (reported by Le Droguen et al., 2015) which prevented us from analysing the role of Rab11 in tube formation. But because Rab11 does not localize to the compartment at the tip of the cell, we believe that this structure does not rely on Rab11 to transfer material from the apical to the basal membrane of the cell.

      We will add the data on Rab11 distribution in Figure S6.

      1. In addition to the above, I would recommend more discussion of how the authors' results relate to membrane trafficking and transcytosis in other systems. The recycling endosome should be considered, and it may be appropriate to draw comparisons to membrane trafficking in neurons that goes through MVBs (e.g. reviewed in VON BARTHELD and ALTICK Prog Neurobiol. 2011 Mar; 93(3): 313-340.). Although neurons are not hollow, they have definite morphological resemblance to tracheal terminal branches.

      We thank the reviewer for the observation and will expand the discussion on MVB-mediated transcytosis in other systems.

      1. line 204-208 "To test whether raised levels of Crb were responsible for the excessive apical membrane, as reported in other contexts (Pellikka et al., 2002; Schottenfeld-Roames et al., 2014; Wodarz et al., 1995), we knocked down Crb (Fig. S4G-H)." According to the legend for Fig s4, the authors express an RNAi construct against crbs. However, there does not appear to be any quantification of the amount knockdown of crb that was achieved. This is a concern for two reasons: 1) RNAi in the embryonic trachea works poorly for most genes (for unknown reasons) 2) This does not appear to be a clonal experiment but rather a pan tracheal driving of Crb RNAi. Loss of crbs would be expected to have very negative effects on tracheal morphogenesis (although this hasn't been rigorously tested to this reviewer's knowledge), but there doesn't appear to be any adverse effects of pan-tracheal crbs RNAi, suggesting that little if any knockdown of crb was actually achieved.

      The authors either need to document the reduction of crbs or remove this paragraph. Preferably, they would be able to document the reduction of crb because they are trying to address an important point and if they can show the apical expansion is crb-independent, that would be an nice result.

      Loss of Crumbs definitely is detrimental to embryonic epithelia, to different degrees (Tepass and Knust, 1990). We therefore could not use mutants but expressed an RNAi, which does not abolish Crb completely. We have now determined the degree of crb knockdown in our experiments. We stained Crb in embryos that expressed crb-IR in the entire tracheal system (but leaving the epidermal expression intact) and quantified the amount of Crb in the tracheal dorsal trunks, normalized to the signal in the epidermis. We found that in crb-IR embryos, Crb levels were reduced by around 50% compared to control siblings. We will add these results to Fig. S4 in the new version of the manuscript.

      Minor concerns:

      1. Lines 250. "By this interpretation, unscissioned membrane invaginations protruding from the subcellular tube would occasionally have touched the basal plasma membrane or its protrusions and fused with it, as transcytosing vesicles would have done in the normal situation."

      I am not convinced by the argument that the bridging invaginations are fusing analogously to transcytosing vesicles because a protrusion/nascent vesicle coming from the apical surface should have rabs and V-SNAREs that should dock the protrusion/nascent vesicle with an endosomal compartment, not the basolateral surface. A transcytotic vesicle would have the rabs and V-SNAREs for the basolateral membrane. So it would seem that a fusion of the apical surface directly to the basolateral surface would have to be an ectopic event outside of the normal situation.

      We agree that the fusion events could also be unrelated to the normal physiology of the animal. In other contexts (e.g. the embryonic epidermis, the synaptic bouton) blocking dynamin results in long membrane invaginations as a result of failure in membrane scission. In terminal cells, the apical and basal plasma membranes are very close to each other, and we believe this increases the chance of membrane invaginations meeting and fusion to take place. In addition, the long membrane invaginations we see seem to have been stripped of their clathrin coat suggesting that at least some aspects of ‘vesicle’ maturation proceed even though scission had failed. We also find evidence of small vesicles that resemble the contents of MVBs being deposited within the aberrant membrane invaginations. This suggests that MVBs are able to fuse with these unscissioned tubes and sheets, again indicating that the appropriate molecular markers are present, and the machinery in charge of generating these vesicles is active at the invaginated pits directly. In either case, we will rephrase our interpretations of these data and present it as speculation in the discussion section.

      1. Significant figures. This is not a big deal, but the authors are over reporting their significant figures. E.g. "a 7.05-fold increase (+/-2.98 SD)" . With an SD that is 50% the value of the measurement, reporting to hundreds is definitely beyond the accuracy of measurement. Rounding to tenths would be more appropriate.

      We agree with the reviewer. We will rephrase this section and use more appropriate metrics. As prompted by Reviewer #1, we modified the analysis that corresponds to this sentence, which also modified the way data is normalized also reducing the spread. This happened because in the new analysis we compare apical and basal signal for each timepoint, which allows better comparison between different cells.

      Significance:

      As there have not been that many studies on the dynamics of membrane trafficking during morphogenesis, the results should be of broad interest to those studying the endocytic system and the role of membrane trafficking in morphogenesis. However, the paper would be greatly strengthened if the authors considered the recycling endosome in their analysis and write up. As a well-known compartment for trafficking cargo and membrane to both the apical and basolateral surface, it is hard to know how to interpret the observed trafficking without knowing the involvement, or lack thereof, of recycling endosomes in this system.

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      Referee #1

      Evidence, reproducibility and clarity

      Summary:

      In this work the authors investigate the dynamics of cell morphogenesis in a convenient in vivo system. They use the terminal cells of the embryonic tracheal system and comprehensively address how cell shape change (elongation in this case) takes place and how membrane is remodeled during the process. By combining different high-resolution techniques, i.e. in vivo imaging of terminal cells expressing different membrane markers and serial-section electron tomography, they describe the organelle organization (ER, Golgi, different types of vesicles) in the terminal cells during their elongation. They identify the presence of membrane structures/vesicles particularly abundant at the tip of the cell ahead of the growing tube. When they block endocytosis they find increased tube membrane and lack of basal membrane growth. In addition, in tomograms, they observe clear membrane defects, like invaginations that could even connect the tube and basal membranes. This correlates with the absence of vesicles at the tip observed in normal conditions. The analysis of the nature of the vesicles observed indicated the accumulation of late endosomes and MVB, particularly at the tip of the growing terminal cells. Interfering with the formation of these MVB led to defects in the growth of both the tube and the basal membrane. Altogether the authors propose a model in which the newly formed membrane (and transmembrane proteins) passes through the ER and Golgi and reaches the apical membrane. The incorporated membrane is then rapidly endocytosed and follows a maturation pathway through MVB, from where different cargoes and membrane would be sorted and recycled back to the apical (tube) membrane, or to the basal membrane through a transcytosis mechanism

      Major comments:

      - Are the key conclusions convincing?

      Most of the conclusions presented are convincing and supported by the results observed. However, to my understanding, one of the key conclusions of this work (that membrane is transcytosed from the apical to the basal domain) is not fully convincing. A critical result to support the author's conclusion of apical-basal transcytosis is to find clear evidence of basal accumulation of a transcytosed marker. The authors show accumulation of FGFR-GFP and Myospheroid as evidence. However, I find the results presented not very convincing. The accumulation of FGFR-GFP at the basal membrane in the control is not very clear in the images and movie presented. In addition, in the shibire mutant, some basal accumulation of FGFR-GFP seems to be detected (particularly in the movie). In the figures the authors show an increase of FGFR-GFP intensity when endocytosis is blocked, but this is not explained in the text. On the other hand, and very importantly, how does FGFR localization relates to its activity? The authors show that when endocytosis is prevented dpERK (i.e. a reporter of FGFR activation) is not decreased, indicating that FGFR is normally active. Wouldn't this suggest that FGFR is still localized basally to receive Bnl signal? Actually, as the authors indicate, in larval tracheal cells the intracellular accumulation of the FGFR leads to a reduced FGF signal transduction (Chanut-Delalande et al., 2010), suggesting that reduced FGF signaling activity in these cells is due to less FGFR reaching the basal membrane. The results with Myospheroid are not very convincing either, as the authors just show a single confocal section of control and shibire mutants. In summary, I consider that this very important point needs to be better documented before concluding that apical membrane material containing basal cargoes is transcytosed to the basal membrane.

      Another conclusion that, to my opinion, should be better explained and documented, is the coordination of tube and basal membrane growth. Following the movements of the vesicles the authors conclude that there is a net displacement of these vesicles to the tip of the cell. This correlates with the presence of mature endosomes there. So the results postulate that transcytosis occurs at the tip, and therefore the growth of the basal membrane would occur preferentially at the tip. It has been demonstrated, as the authors indicate, that the tube membrane grows all along the length of the tube. How is then coordinated the tube and basal membrane growth? If, as the authors propose in their model, the tube membrane also grows after a process of endocytosis and recycling, wouldn't it be expected to have preferential tip growth? How do the authors reconcile all these observations with previously published results (Gervais and Casanova, 2010)?

      The serial-section electron tomography analysis is very interesting and identifies different sorts of vesicles. However, it is very unclear what the different vesicles referred in the models correspond to (in the in vivo imaging for instance). For instance, the small granular or the dense-core vesicles correspond to endocytic vesicles at different stages of maturation?. If there is a constant endocytosis from the apical membrane to generate the basal and build the definitive apical membrane, wouldn't it be expected to find many more vesicles around the tube? Wouldn't it be expected to find coated vesicles around or budding from the tube, as the coated vesicles observed budding from the basal membrane in Fig 2D? Or is the endocytosis observed mediated by non-clathrin coated vesicles?.

      - Should the authors qualify some of their claims as preliminary or speculative, or remove them altogether?

      The results of Serp accumulation upon MVB interference can lead to confusion (from line 404). The authors seem to suggest that Serp protein is exclusively produced in the FB and transported by transcytosis to reach the tracheal lumen. However, Serp is also produced in the tracheal cells themselves. In fact, serp expression in the FB seems to be detected by late embryogenesis, while expression in tracheal cells is detected much earlier (Dong et al. 2014; Luschnig et al. 2006; Wang et al. 2006). It was also shown that Serp undergoes a recycling mechanism from the lumen to the lumen, through the endosomes-TGN retrograde trafficking, that may also require Shrub (Dong et al, 2014; Dong et al 2013). Thus, it is unclear (and even unlikely) that the Serp found in the vesicles in Shrub-GFP mutants is derived exclusively from the transcytosed component from the FB. I suggest to better explore this issue or to remove this part.

      - Would additional experiments be essential to support the claims of the paper? Request additional experiments only where necessary for the paper as it is, and do not ask authors to open new lines of experimentation.

      As indicated before, more conclusive results for transcytosis should be provided.

      I suggest that the authors determine the presence of apical and basal cargoes (FGFR) in the late endosomes found when Shrub activity is impaired. According to his model, both types of markers should accumulate there. A high accumulation of these markers in those endosomes would reinforce the hypothesis proposed.

      While it has been reported that shrub-GFP act as a dominant negative in different contexts (Dong et al, 2014; Sweeney et al 2006) it is unclear why. So it would be desirable to confirm the results with a loss of function condition (either mutant or RNAi line)

      - Are the data and the methods presented in such a way that they can be reproduced?

      The materials and methods section would benefit from more detailed explanations.

      - Are the experiments adequately replicated and statistical analysis adequate?

      Many of the experiments presented in this work are technically very challenging, like the in vivo analyses and particularly the serial-section electron tomography. This prevents having high numbers of replicates on occasions.

      Minor comments:

      -In the abstract the authors state: "We show that apical endocytosis and late endosome-mediated trafficking determine the membrane allocation to the apical and basal membrane domains". I think that the authors show that "that apical endocytosis and late endosome-mediated trafficking is required for correct membrane growth", but I am not that sure that they show that it determines the membrane allocation

      -References for the PH-GFP localization in cells should be provided. Which is the evidence that it only localizes to plasma membrane?

      -It would be more adequate to always use the same terms to facilitate the reading. For instance, in several figures the membranes are referred as basal plasma membrane and tube membrane, but in others outer and inner membrane

      -Figure 3C,D and corresponding text are difficult to understand. The increase of fluorescence of the inner membrane seems to be very high, even higher than the corresponding to the outer membrane. Can the authors explain better this point and also describe better the method applied in the materials and methods section?

      Significance

      This work represents an important advance for the field for several reasons. First of all it represents a technical advance because the authors are able to combine the traditional genetic analysis with two powerful techniques (in vivo imaging and serial-section electron tomography) to analyze single cell behavior at high resolution (temporal and spatial). In addition it represents a conceptual advance as it proposes a mechanism through which membrane growth is coordinated to regulate cell morphogenesis. The mechanism presented (endocytosis and transcytosis) is not new but they find evidence in an in vivo system. It was previously known that tracheal terminal cells undergo a process of intracellular tube formation and cell elongation at the same time, but the mechanisms coordinating these two cell events were not known. The proposed mechanism may not only be relevant for the morphogenesis of tracheal terminal cells, but could represent a general mechanism of cell morphogenesis. Therefore, the paper should be relevant for research in the morphogenesis area but also in the cell biology field, as it shows how regulated membrane trafficking can control tissue morphogenesis

      REFEREES CROSS-COMMENTING: I agree with reviewer #4 on her/his comments and suggestions about analyzing the involvement of the recycling endosome in the process.

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      Reply to the reviewers

      Review comments Rebuttal

      Reviewer #1 (Evidence, reproducibility and clarity (Required)): **Summary ** This manuscript describes the X-ray structure determination of two SMAD-DNA complexes confirm that SMAD family proteins bind at least two DNA sequences in a similar fashion, and explores dimer versus monomer formation of the non-DNA bounds forms of the proteins which could influence whether the proteins bind as monomers and dimers. This includes identifying a loop which appears to make a major contribution to this process. There is a lot of experimental work and analysis included. **Major comments: ** The overall conclusions of the manuscript are convincing, but some of the detailed analysis is not clear. The structures look good, the experiments look to be generally well controlled, although some details could be provided in the main text to be clear about what methodology is being used or how analysis was carried out and stepwise conclusions obtained. In particular the analysis of SAXS data is not clear. I'd like to see initial data analysis presented as per the guidelines of Trewhella et al 2017 (PMID: 28876235). There is some mention of data in the SASREF database, but it should be in the supplemental data.

      We have prepared a table following this recommendation.

      I can't see any evidence for the conclusions about open versus closed monomer state (how good were the fits obtained) - just a graph and a statement. If this can't be better justified please remove the conclusions about these states (they don't really add to the overall conclusions about monomer/dimer which are much less specific), but even the simple analysis supports mostly monomer and small amounts of dimer or higher aggregates. I would also like to see a clear explanation provided about why the MS data supports dimer over other oligomers

      We have revised and simplified the SAXS section to clarify the main points. We have re-analyzed the conformations in solution, and the values are presented in new Table S4 and new Figure 3D. We have also included new panels (Figure 3E) and explanations with respect to the IM-MS data (pages 8-9).

      State what thermal unfolding experiments are were carried out in the text (and why is the data biphasic?)

      The biphasic graphics were interpreted as the presence of dimers and monomers in equilibrium. As suggested by the other reviewer, we have removed these sections as they do not contribute to clarify the main points of our work.

      The concept of long versus short loops re domain swapping have been studied in the past but there isn't much reference to this.

      https://www.ncbi.nlm.nih.gov/pmc/articles/PMC2373619/

      https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6349918/

      https://journals.plos.org/plosone/article?id=10.1371/journal.pone.0039305

      https://www.ncbi.nlm.nih.gov/pubmed/22411444

      We now mention some related examples in page 8.

      **Minor comments: **

      The last couple of paragraphs of the introduction are a fairly comprehensive summary of the study overall and the conclusions of the paper. While presaging the key findings and conclusions is fairly common in an introduction this seems to be way too much detail. Unless it is a requirement of the journal, reduce these sections to a couple of sentences and use any other word count to explain your analysis better.

      Thank you for this recommendation. We have rephrased and reduced this part of the introduction.

      Figures are quite small and hard to see detail at 1X magnification (in both the main and Supplemental figures).

      We have removed some panels that were not necessary and increased the size of the figures and labels.

      NB.The difference in Tm of SMAD 5 over 8 doesn't seem particularly high as it’s only a couple of degrees (especially when SMAD4 is quite different). The explanation for the Ile>Cys mutation might be about competition of zinc ligation (except that it doesn't seem to cause issues for many zinc finger proteins) but more likely that you've replaced a reasonably bulky hydrophobic sidechain and therefore have lost a bunch of hydrophobic contacts.

      We have removed this section entirely.

      With respect to the Ile>Cys difference, the residue is located in a loop, and it does not participate in hydrophobic contacts. We still believe that its negative role in the stability of the domains arises from the competition for Zn coordination but we agree with the reviewer that quantifying its specific role is not obvious.

      Reviewer #1 (Significance (Required)):

      -This paper clarifies concepts about the state of isolated SMAD proteins (thought be largely monomeric in the absence of DNA) and DNA-binding preferences of these proteins. -I don't have specific expertise in the structure/function of SMAD proteins, but the study appears to include sufficient background to place the study in context.

      -Audience will mostly be those interested in structure/function of SMAD proteins, with some protein engineers interested in the manipulation of monomeric versus dimer.

      -I am a protein chemist and structural biologist with an interest in protein dimerization/oligomerization. I am familiar with most techniques presented, but don't have first-hand experience with IM-MS.

      Reviewer #2 (Evidence, reproducibility and clarity (Required)):

      **Summary**

      The manuscript by Ruiz et al examines how the receptor-activated SMAD (R-SMAD) transcription factors bind DNA, and specifically how the MH1 (DNA-binding domain) of the different classes contributes determining whether they bind as monomers or dimers.

      In the context of the full length SMAD proteins, it is thought that hetero-trimers with one SMAD4 plus two R-SMADs are the functional unit. In general, the SMAD1/5/8 R-SMADs respond to BMPs whereas SMAD2/3 respond to TGF beta. However, what is less clear is how the specificity of the gene responses is determined, since all SMADS are able to bind to each of the two sequence classes of response element (GTCT and GC-rich, or 5GC).

      Previous structural studies suggest that the major contacts between SMAD MH1 and DNA are very similar, irrespective of the particular SMAD or of whether they bind a TGTC or 5GC element. On DNA, MH1 domains have been observed as dimers, but there has been some concern as to whether this (at least in part) is a crystal artefact, or is perhaps forced by the specific DNA sequences use in these studies. For the BMP R-SMADS this may be less likely, since the amino-terminal helix 1 of one dimer is seen to be dislodged from its own intramolecular interactions allowing it to make contacts with the second MH1 domain in the dimer.

      Here the authors test this question of MH1 dimerization and address differences between the BMP responsive and other SMADs. They first show by crystallography that SMAD5 and SMAD8 MH1 domains adopt similar dimeric conformations with the displaced helix 1, and bind to a single 5GC element via one of the MH1 domains. To get at whether these MH1 domains form dimers in solution, they use small angle X-ray scattering, NMR and mass spectrometry, to suggest that the SMAD5 and SMAD8 MH1 domains in solution do not fit with a single conformation, but are better modeled by a mixture of dimer and open monomer. Ion mobility MS also suggested a mix of dimer and open monomer for the BMP SMADs, whereas SMAD3 appeared to be primarily monomer. To test if the MH1 domains themselves encode this potential difference between SMAD5 and SMAD3, they swap loop 1 (6 versus 4 amino acids, between helices 1 and 2) from SMAD3 to SMAD5, and now in solution this chimera appears monomeric, and forms monomers when crystalized with DNA.

      Major comments

      1. Adding the SMAD3 loop to SMAD5 prevents the open dimer - does the reverse also work? Can you make SMAD3 form SMAD5-like open-dimers by adding the loop 1 sequence from SMAD5?

      We have prepared new Smad3 chimeric constructs and we are currently screening crystallization conditions in order to obtain diffracting crystals (if possible). Unfortunately, due to the COVID-19 pandemic, access to our laboratory is highly restricted, while access to synchrotron and mass spectrometry facilities is not available), therefore this work has been postponed until the end of April/May. For this reason, the revised version of the manuscript does not refer to this question. We hope that we will be able to address it in the future.

      1. Can the authors include similar schematic models for how the site spacing would be for SMAD2/3-SMAD4 complexes - adding the SMAD2/3/4 model to Figure 5C?

      We have incorporated new panels to Figure 5 (Figure 5E,F).

      1. The authors comment on the possibility that the dimer conformation dictates the spacing of the sites that will be bound in vivo. In this context, they refer to a previous paper (PMID: 29234012) to suggest differences in site clustering between BMP SMAD and TGF beta SMAD regions of the genome (from ChIP-seq) that fit with the spacing they imply here. However, the major difference shown in this work seems to be between the clustering of GC sites and GTCT sites irrespective of the pathway. Can the authors analyze existing ChIP-seq data to more specifically test the question they raise - ie that SMAD4 bound regions of the genome have different site clustering/spacing depending on whether they are BMP or TGF beta responsive?

      Thank you very much for this recommendation. We agree with the reviewer that this information is very valuable and can help support our hypothesis on the different binding preferences of monomers and dimers of MH1 domains. We have performed this analysis and is now included as two new sections. The results are displayed as new Figure 5A,B.

      1. I think Figure 2C,D is not really well described in terms of the importance to this work. As it is this data does not really seem to add very much, but perhaps I am missing the importance.

      We have entirely removed this section in the new version of the manuscript.

      5.Can the authors comment about the compressed GC element or BRE? This seems to be an unfavorable conformation. How might it be bound in vivo, is it an unusual element, or is it relatively widely found? Is it possible that in vitro it binds two MH1 domains, but in vivo might simply act as a normal 5GC, with an additional site nearby?

      The BRE domain is less abundant than 5GCs and SBE sites, and in fact, this sequence is not enriched in the ChIP-Seq datasets that we have analyzed. We have included a sentence refering to these findings in page 11.

      We have also revised the section comparing the 5GC and BRE-GC site and illustrate this interaction as well as the comparison to our 5GC complex by including two panel in Figure2 that before were displayed as supplementary information. The panels have been edited to clarify the similarities and differences between both complexes. Indeed, the protein-DNA complex made of one BRE motif bound by two MH1 domains as found in the PDB:5X6H crystal structure suffers from several issues, including compactness of the two overlapping Smad-binding motifs that led to distortion of DNA geometry, clashes at protein-protein interface and local cancelation of protein-DNA interactions.

      In this new section (page 8) we include the sentence that “we believe that the most probable binding mode in vivo should be that observed in the 5GC and SBE complexes. It seems very unlikely that two MH1 domains would interact with a reduced BRE motif —using half of their protein binding site and causing a high distortion to the DNA structure— if there is the possibility to interact with neighboring sites (Figure 2B,C) using the full protein binding interface and a perfect accommodation to the DNA”.

      **Minor comments: ** 1.In Figure 1B is one the two DNAs assumed? In the structure was it two MH1 to one DNA or two of each?

      One DNA was hidden for clarity. The crystallographic structure is now shown in full. The crystal structure was solved for a complex made of two MH1 domains bound to a dsDNA molecule that included two Smad-binding sites.

      2.Figure 2C and page 9: the stabilization of SMADs in the text and figure do not agree. Maybe just state the exact numbers from the figure in the text.

      We have entirely removed this section in the new version of the manuscript.

      3.In Figure S1C, can the authors label the retarded complexes on the gels?

      Done.

      4.Figure 4A - explain the asterisk (presumably the SMAD2 insert).

      Yes, it corresponds to the Gly rich region present in loop1. It is indicated now.

      5.In Figure 4B, C (and maybe D) can they color helix 1, loop 1, and helix 2 three separate colors, it might really emphasize the effect of the loop if it was more immediately visible.

      We have improved these figures but we did not change the colors because the figure was getting even more complicated.

      6.The legend to Figure 4 is missing F.

      Thank you. This has now been corrected.

      Reviewer #2 (Significance (Required)):

      The authors conclude that the length of the loop between helices 1 and 2 determines the dimer versus monomer state - a shorter loop as in the BMP SMADS hinders the intramolecular interactions needed for the closed monomeric form, whereas the longer loop in the other SMADS allows the flexibility for these interactions so favors a more closed monomeric form. Showing that the dimers are not forced by crystallography or by binding to fixed DNA elements clearly adds to our understanding of the mechanisms of SMAD function, and it is of interest that the BMP and TGF beta SMADS are different in this respect.

      They speculate that this may contribute to the specificity of the responses activated by BMP versus TGF beta signaling based on the requirements for different site spacing depending on whether an open (BMP) or closed (TGF beta) dimer of R-SMADS is present. This idea is likely to be of interest to anyone who studies the responses to the TGF beta superfamily of signaling molecules, and should spur additional experimentation to test it.

    1. We connected this notion to larger systems of oppression and how stereotypes help create and perpetuate those, noting how, for example, our justice system relies on humans who may harbor stereotypes which can result in severe consequences for those in their charge

      As teachers it is important for us to think about the stereotypes we hold of the students we teach. How does it influence teaching and learning?

    1. Finally, moving maintenance and repair back to the center of thinking around media and technology may help to develop deeper and richer sto-ries of relationality to the technological artifacts and systems that surround us, positioning the world of things as an active component and partner in the ongoing project of building more humane, just, and sustainable collec-tives.

      I think this last point is really excellent. If the ability to repair any infrastructure isn't built holistically into a system the ability for any option other than building over or removing and replacing isn't on the table.

      For example when I was a construction inspector for the Township of Lake Como I worked on their artificial aquifer that fed their lakes and houses. They had chosen to build a piping system that had no shut off connectors for outflow and further draining on the north half of the town and on the southern half they had built into those connectors. We were able to reline the southern half of the town's pipes in 5 weeks and we had to remove and replace almost every major water pipe in the town on the north half which took 6 months. A lack of any ability for maintence leads to greater problems.

    1. Children know this; they have the desire and the ability to search for the meaning of life and their own sense of self as soon as they are born.

      LOVE THIS! Children want to know 'why' they are here? Their purpose (not the 'purpose' their parents think they should have ;)! When we pay close attention we may find that the innate desires/interests that the children show us are in fact tied to their ultimate purpose. For example, a little boy whose passion for building is supported by providing him wood, tools, etc. so that he can build a table - later we come to find out that this same boy became an adult contractor.

    1. The discussion of privacy, intellectual property rights and freedom of information has shown that a good case can be made for the descriptive culture-relativity of these values. These values are central in information ethics, as it has been developed in the West. Moreover, it was argued that the uncovered cultural differences in the appraisal of these values can be placed in the context of a dichotomy between two fundamentally different kinds of value systems that exist in different societies: rights-centered and virtue-centered systems of value. Information ethics, as it has developed in the West, has a strong emphasis on rights, and little attention is paid to the kinds of moral concerns that may exist in virtue-centered systems of morality. In sum, it seems that the values that are of central concern in Western information ethics are not the values that are central in many nonwestern systems of morality. The conclusion therefore seems warranted that descriptive moral relativism is true for information ethics.

      This conclusion proves the point that I made in my alternate annotation. We are obviously able to see so many differences propagated by cultural relevancy phenomenon. In fact, each case study outlines this very thing. There are countless examples down to even when a person becomes an autonomous human entity. So why must we resort to categorizing in such a primitive other-ing way? I think that there could have been many stronger conclusions to this piece and I am disappointed to see the author resort to the insufficient cop out method of binary categorization.

    1. In their minds, clearly, writers fall into three fairly distinct groups: the talented, the average, and the others; and the Writing Center's only logical raison d'etre must be to handle those others- those, as the flyer proclaims, with "special problems."

      I think it is easy to classify writers if grammar is the only factor taken into consideration. Otherwise, everyone has a different idea of what makes someone a good writer. For some, it may be the ability to make the reader feel something. For others, someone who uses big words and long sentences may be thought of as the best writer in the world. I believe our personal classifications are dependent on our own relationship to writing and what we strive to improve in our own writing.

    1. Note: This rebuttal was posted by the corresponding author to Review Commons. Content has not been altered except for formatting.

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      Reply to the reviewers

      We thank all the Reviewers for taking the time to evaluate our manuscript and providing us with constructive feedback. We are pleased to hear that all Reviewers appreciate the importance and significance of our study, commenting that our conclusions are ‘convincing, are supported by the presented experimental results’ and that our study ‘will yield novel insights into the regulation and function of PALB2 in DNA repair’.

      Please refer to our point-by-point response to the specific points raised, in which we highlight a couple of key experiments to be conducted to refine our study in bold. We are grateful for all the reviewers’ remarks and suggestions, which will certainly lead to a substantial improvement in our manuscript.

      Point-by-point response:

      Reviewer #1 (Evidence, reproducibility and clarity (Required)): Fournier et al. detect acetylation within the chromatin association motif (ChAM) of PALB2 and demonstrate that KAT2 can acetylate these 7 lysine residues within this region. They then generate K to R mutations (7R) or K to Q mutations (7Q) at these sites and perform assays of fluorescence recovery after photobleaching (FRAP) to measure mobility as a measure of chromatin association, RAD51 foci, PALB2 recruitment at sites of laser-induced DNA damage, and sensitivity to olaparib. They find increased mobility of the 7Q mutant of PALB2 but not 7R in the absence of exogenous DNA damage, as well as defects in DNA damage-induced RAD51 foci and resistance to olaparib. On this basis, the authors conclude that acetylation is required for the association of PALB2 with undamaged chromatin and that deacetylation permits mobilization and association with BRCA1 to enable proper DNA repair. While the manuscript is generally well-written, many of the systems are rather elegant, and this study may yield novel insights into the regulation and function of PALB2 in DNA repair, there are some missing experiments to be added and important contradictions that should be resolved in order to fully establish the new model the authors propose.

      **Major comments:** 1.There are some concerns about the interpretation of experiments with the 7R and 7Q mutants of PALB2. For example, in the description of results in Fig. S2C, the authors state "K to R substitutions maintain the charge yet are unable to accept acetylation and hence mimic constitutively non-acetyl lysine". However, in Fig. 4B the association of the 7R mutant with chromatin is similar to WT and in Fig. 7D,E the relative immobility of the 7R mutant is very similar to WT PALB2. Thus, the conclusion that acetylation is required for PALB2 association with damaged undamaged chromatin and for release of PALB2 upon DNA damage does not appear justified. Perhaps the authors need to better consider whether the 7R mutant mimics acetylation because of its charge. Even so, the mutant then maintains the charge normally associated with acetylated PALB2, calling into question whether deacetylation indeed "releases PALB2 from undamaged chromatin".

      We agree with the Reviewer’s point that there is no or little difference between WT and the 7R mutant in regard to their enrichment on non-damaged chromatin, as detected by fractionation (Fig 4B), or their mobility, as detected by FRAP (Fig 4D and E). Note that Fig. 7D is our model and Fig. 7E does not exist. As the Reviewer suggests, it is possible that, in contrast to the 7Q mutant, which is defective in both nucleosome and DNA binding (Fig. 2E and F), the 7R mutant may maintain its electrostatic interaction with DNA, while lacking its acetylation-mediated nucleosome interaction, masking the impact of substitutions. This assumption is in line with our model in which ChAM DNA binding assists HR repair, which is supported by the 7R mutant but not by the 7Q mutant. To better dissect the question raised by the Reviewer, we will conduct biochemical analyses of the ChAM 7R mutant, testing its direct interaction with nucleosomes and DNA; the results will be included in the revised manuscript (Experiment 1).

      It is also worth noting that full-length PALB2 is enriched at a fraction of H3K36me3-marked exons (which comprise only 1-1.5 % of the whole genome), as shown in our previous genome-wide ChIP-seq analysis (Bleuyard et al., 2017, PNAS). Hence, it is also possible that bulk fractionation or FRAP analyses might not be sensitive enough to highlight the impact of the 7R mutation. Conversely, we foresee that the ChIP-qPCR method, detecting PALB2 association at defined genic regions as shown in Fig S5, will be more appropriate. Thus, in the revised manuscript, we will expand our ChIP-qPCR analyses to further validate our proposed model (Experiment 2).

      2.Related to questions of interpreting results utilizing the 7R and 7Q mutants of PALB2, in Fig. 7B,C the 7R mutant but not 7Q supports RAD51 foci and resistance to olaparib similar to WT PALB2. The authors then state in the Discussion that "our work also suggests that caution should be exercised in the use of K to Q substitutions for functional studies of lysine acetylation". Thus, which mutant is giving the correct and reliable results?

      We apologise for the miscommunication if this point was unclear. Using biochemical approaches, we established that ChAM acetylation, but not K to Q substitution, facilitates its association with nucleosomes (please compare Fig 2E and Fig 3B). This observation clearly demonstrates that K to Q substitution does not mimic acetylation at these residues, but instead renders PALB2 ChAM functionally null. The PALB2 7Q phenotypes therefore demonstrate the importance of the 7K patch for ChAM function in HR repair, rather than its acetylation status.

      Perhaps even more importantly, if results with the 7Q mutant are suspect, the conclusion that deacetylation is required for HR (or DNA repair) is suspect because that is the only case where the authors see a defect in RAD51 foci and resistance to olaparib. Similarly, if the 7R mutant "mimics non-acetyl-lysine" then the fact that it has normal RAD51 foci and resistance to olaparib contradicts the conclusion that deacetylation is required for DNA repair.

      Unfortunately, it is currently technically not possible to ‘lock’ the PALB2 7K patch in its acetylated status in vivo (i.e. preventing PALB2 dissociation from active genes). We thus agree with the Reviewer that it is difficult to draw definitive conclusions on the impact of constitutive PALB2 acetylation in HR, although the importance of the 7K-patch for the functionality of PALB2 is evidenced by the 7Q mutant phenotypes. Similarly, strictly speaking, our results using the 7R mutant support the notion that the ‘non-acetylated’ status of the 7K mutant, but not necessarily the dynamics of ‘de-acetylation’ events, can promote HR repair. In the revised manuscript, we will rephrase and clarify these points.

      3.There are multiple concerns about Figs. 5 and S5. In Fig. 5A-C, difference in cell cycle progression after synchronization are relatively small and no rationale/interpretation is given for how this may be related to PALB2 function is given. In Fig. 5D,E differences in the levels of gamma-H2AX as a marker of DNA damage between different forms of PALB2 do not become readily apparent until about 6 or more days after addition of doxycycline. As such, it seems that these could be indirect effects and it is unclear how strongly this supports the importance of PALB2 acetylation in the DNA damage response.

      We apologise for the miscommunication on these points. We have previously established that steady-state PALB2 chromatin association, jointly mediated by the ChAM and MRG15 interaction, protects a subset of active genes from DNA damage that may otherwise arise from replication-transcription conflicts (Bleuyard et al., PNAS 2017). The results presented in Fig 5 and S5 led us to propose that PALB2 chromatin association is, at least in part, mediated by the ChAM 7K patch, and its acetylation (hindered by 7Q and 7R substitutions, respectively) prevents DNA damage via a similar mechanism, i.e., protecting PALB2-bound genes during replication. This model nicely supports our observations that both 7Q/7R mutants exhibit slow S-phase progression and accumulation of gamma-H2AX over time. These points will be better articulated in the revised manuscript.

      In Fig. S5, it is interesting that there are differences in the association of different forms of PALB2 with 3 distinct active loci, but no error bars or measures of statistical significance are given. Further at 2 of the 3 loci, the association of the 7Q mutant is closer to WT than the 7R mutant. Taken together, neither Fig. 5 nor Fig. S5 strongly support the key conclusion that acetylation regulates the association of PALB2 with actively transcribed genes to protect them.

      We appreciate this constructive comment. The analysis was conducted once, albeit with three technical replicates, which explains why the results are presented without error bars. Nonetheless, we observe a consistent trend at three different loci, that both 7R and 7Q have chromatin association similar to the empty vector, which is background level (FLAG/IgG ChIP) and does not reflect real binding. The revised manuscript will include the results from three biological replicates with statistical evaluation (Experiment 2).

      4.Figs. 6D-G and S6A-D conclude that "DNA damage triggers ChAM deacetylation and induces PALB2 mobilization" based upon FRAP experiments utilizing WT PALB2. But there is no control to demonstrate that this is a specific effect driven by the state of PALB2 acetylation. For example, DNA damage might cause global acetylation changes resulting in relaxed chromatin in which proteins that are not subject to acetylation-deacetylation also show increased mobility.

      We thank the Reviewer for this valuable comment. It is true that we cannot formally exclude the possibility that changes in PALB2 mobility are indirect consequence of damage-induced chromatin reorganisation/increased chromatin mobility. However, our analyses clearly demonstrate that ChAM acetylation increases its association with nucleosomes (Fig. 3B), while non-nucleosome binding ChAM-null (7Q or deletion) increases PALB2 mobility (Fig. 2E, Fig. 4E and Fig. S4C). Further, WT PALB2 mobility increases after KAT2 depletion (i.e. reduction of chromatin acetylation of KAT2 targets, hence chromatin compaction) (Fig. 3F), but reduces upon KDAC inhibition (i.e. global increase in acetylation, hence chromatin relaxation) (Fig. 3G). Considering all these observations collectively, the increase in PALB2 mobility detectable upon DNA damage is unlikely to reflect global chromatin relaxation, and that PALB2 acetylation influences its mobility in both challenged and unchallenged cells. This point will be emphasised in the revised manuscript.

      5.Fig. 7B shows that the 7Q mutant has diminished RAD51 foci while Fig. S7C,D suggests based upon a different methodology (laser-induced damage) that the 7Q mutant does not affect PALB2 recruitment. Since the issue of recruitment is key to the mechanism proposed, the authors should examine PALB2 foci instead as this may be a more sensitive assay of PALB2 recruitment.

      We appreciate the Reviewer’s point. We would like to highlight, however, the well-documented role of BRCA1 in PALB2 recruitment to sites of DNA damage. This supports our notion that the 7Q mutant is recruited to sites of DNA damage, likely mediated via its interaction with BRCA1. As depicted in Fig. 7D, we propose that the 7K patch-mediated PALB2 engagement with damaged DNA, which is disrupted by the K to Q substitutions, is essential for proper RAD51 loading onto DNA, hence RAD51 foci formation and HR repair. This is in line with our observation that PALB2 ChAM deletion, similarly to the 7Q mutant, perturbs damage-induced RAD51 foci formation (Bleuyard et al., EMBO Rep. 2012). We believe that the laser-induced experiments provide high sensitivity and resolution for PALB2 recruitment kinetics, as the data were obtained with real-time live-cell imaging.

      6.The authors state in the last sentence of the Results section that "lysine residues within the ChAM 7K-patch are indispensable for PALB2 function in HR" but never test the mutants for HR using reporter assays. The manuscript would be strengthened by performing such assays.

      RAD51 foci formation and sensitivity to PARP inhibition are well-accepted readouts for HR repair. Conversely, we have been cautious about existing HR reporter systems, which evaluate gene-conversion or targeting events triggered by a ‘clean’ enzyme-induced DSB, but not an authentic repair of ‘dirty’ DSB induced by IR or olaparib.

      7.The model for the role of ChAM acetylation in regulating PALB2 function presented in Fig. 7D is not fully supported by the data presented. Critically, while association with RAD51 and BRCA2 is tested in Fig. S7B, the authors hypothesize that deacetylation is required to release PALB2 to enable association with BRCA1 but this is not tested utilizing the mutants.

      We appreciate the Reviewer’s point. It has been demonstrated that PALB2 interaction with BRCA1 is triggered by damage-induced PALB2 phosphorylation (Ahlskog et al., EMBO Reports, 2016), as well as removal of KEAP1-mediated ubiquitylation in S and G2 (Orthwein et al., Nature 2015). Our preliminary analyses further suggest that BRCA1-PALB2 interaction is highly dynamic, and we propose that damage-induced PALB2 modification and its mobilisation jointly facilitate this interaction.

      Also, there are some specific points that should be considered in the context of the model. This includes how DNA damage may trigger deacetylation, and whether it is the deacetylated state or the process of deacetylation of ChAM that is critical. Also, if acetylation is important for protecting active genes in the absence of DNA damage, is deacetylation necessary to release PALB2 local or global. This is important, because if it is local there needs to be a specific mechanism for local deacetylation, while if deacetylation is global that could result in transcriptionally active genes becoming unprotected.

      We thank this Reviewer for this valuable comment. We agree that, while this study establishes that ChAM is deacetylated upon DNA damage, it remains unclear whether the dynamic ‘de-acetylation’ of PALB2, rather than the ‘non-acetylated status’ of PALB2, is important for HR repair, and whether or not this is a local event. However, we would like to highlight that PALB2-bound genes are mostly periodic, e.g. those required for cell cycle progression (Bleuyard et al., 2017, PNAS). It would therefore be reasonable to speculate that DNA damage triggers the suppression of periodic gene expression as a part of DNA damage checkpoint signalling, possibly in a KDAC-dependent manner, which then allows release of PALB2 without risking DNA damage that could otherwise be caused by replication-transcription conflict. Mobilised PALB2 might then be recruited to sites of DNA damage for HR repair. Further study will be required to fully evaluate this model, for example by identifying the specific KDAC involved in ChAM deacetylation and tracking individual PALB2 molecules, which we consider to be beyond the scope of the present study. In the revised manuscript, we will better describe our model, and further detail the arising questions to be addressed in future studies.

      **Minor Comments:** a.Some parts of the Materials and Methods are overly long (such as the subsection on "Protein purification" and "immunofluorescence microscopy") and could be shortened by consolidating experimental details that are largely the same for related processes.

      We propose to move these experimental sections to the supplementary information.

      b.In the description of Fig. 1D, the statement "7K-patch, which is common to PALB2 orthologs" is misleading since there is not complete conservation of each lysine residue across each ortholog.

      We agree with this Reviewer’s comment and will amend the description in the revised manuscript accordingly.

      c.Figs. 3E,F and S3B,C perform FRAP in cells with knockdown of KAT2A/B as a surrogate for chromatin association. The authors note that this global reduction in acetylation increases PALB2 diffusion, but there is concern that this experiment is not very informative because the increased mobility may have nothing to do acetylation of PALB2.

      Please refer to our answer in response to the Reviewer’s point 4.

      Reviewer #2 (Evidence, reproducibility and clarity (Required)):

      This manuscript reports the control of PALB2 - chromatin interaction by the acetylation of a particular lysine-rich domain of the protein called ChAM. This acetylation is shown to be mediated by the acetyltransferases KAT2A/B. Following these investigations, the authors made an effort to place their findings in the context of DNA replication and DNA repair. The proposed model is that the acetylation-dependent interaction of PALB2 with chromatin could ensure the protection of the genome during DNA replication and control DNA repair. **Specific remarks** 1 - Based on different experiments, essentially the one shown in Fig. 3B, the authors conclude that the acetylation of the ChAM domain enhances its association with nucleosomes. However, taking into account the experimental setting, this conclusion should be largely tuned down. Indeed, this enhanced acetylation-dependent nucleosome binding was observed when the experiment was carried out in the presence of excess of free naked DNA. Under these conditions, the non-acetylated ChAM fragments became mostly trapped by DNA (clearly shown in Fig. 3C/D), and hence would not be available for nucleosome binding, while the acetylated ChAM fragments would remain available for nucleosome association because of their reduced DNA-binding ability. Consequently, the acetylation of the ChAM domain would only play a role on the availability of PALB2 for chromatin/nucleosome binding and not directly stimulate nucleosome binding. Therefore, the nucleosome-binding capacity of ChAM by itself should not be dependent on ChAM domain acetylation.

      If true, this hypothesis could also be relevant in vivo since the poly-K in the ChAM domain could also non-specifically interact with nuclear RNAs and hence its acetylation, by releasing it from nuclear RNAs, would make it available for chromatin-binding. The importance of RNAs in the regulation of PALB2 nucleosome-binding could be tested in the experiments shown in Fig. 2C and 2E by adding RNase to the pull-down medium (WT +/-RNase or addition of increasing exogenous RNAs).

      We are grateful for the Reviewer’s detailed comments and find the potential involvement of RNA very intriguing. Indeed, transcriptionally active loci, which are bound by PALB2, are enriched in nascent RNA, and such local RNA may play an important role in promoting the association of acetylated PALB2 with nucleosomes. However, we believe that investigating the role of RNAs in PALB2 nucleosome binding is beyond the scope of this study. As discussed extensively in response to this Reviewer’s point 2 below, we believe the mode of interaction of ChAM with nucleosomes to be highly complex, being jointly mediated by the N-terminal conserved region and the C-terminal lysine cluster. We will discuss these issues more extensively in the revised manuscript.

      2 - The real question is as follows. While acetylation makes the protein available for nucleosome binding, which part of the ChAM domain is actually mediating nucleosome binding and whether lysine acetylation could be directly involved in this binding. Another question would be to identify the elements in the nucleosome mediating this interaction, histones (core domain, tails, post-translational modifications, specific histone types), histone-DNA, etc...

      We entirely agree with the Reviewer’s question – despite the increasing recognition of the physiological importance of the PALB2 ChAM and our efforts in understanding the mode of association of ChAM with nucleosomes (including the potential involvement of histone tail modifications), this specific question remains enigmatic.

      Explicitly, our previous work demonstrated that substitutions of residues within the evolutionarily highly conserved N-terminal part of the ChAM perturb its association with nucleosomes (Bleuyard et al., 2017, PNAS; Bleuyard et al., 2017, Wellcome Open Research). A recent study by the laboratory of Prof Jackson proposed that basic residues across the ChAM are part of a binding interface with an acidic patch of histone H2A in its nucleosomal context (Belotserkovskaya et al., Nat Comm. 2020). Our results presented in this study introduced an additional complexity, showing that the C-terminal 7K basic patch is essential for ChAM-nucleosome interaction. Intriguingly, our study also suggests that the regions flanking ChAM, which are phosphorylated at multiple residues, play roles in regulating ChAM binding to nucleosomes (Fig 2B and C; please refer to our answer to the Reviewer’s minor point 6 too).

      We are currently working towards solving the structure of ChAM in complex with a nucleosome, which may help to clarify this very important question. At this point, we think that the question about complete elements for the ChAM interaction with nucleosome is out of the scope of this manuscript, and should be addressed in future work. To make this point clear, we will provide an updated overview of the ChAM elements affecting nucleosome interaction in the revised manuscript.

      3 - Taking into account the authors conclusions on the role of ChAM domain acetylation and its impact on PALB2 mobility, in Figure 4D/E, one should expect a difference of t1/2 when wild-type and 7R mutant are assayed by FRAP. At least the measures of t1/2 in the wild-type should have been more heterogeneous compared to the 7R mutant due to the acetylation of the wild-type PALB2 by the endogenous HATs (the impact of endogenous HATs on the wild-type sequence is shown in Fig. 3F). Could the authors comment on this?

      We appreciate this Reviewer’s point. As mentioned in our responses to Reviewer 1’s points 1 and 3, we are unable to exclude the possibility that the 7R mutant still maintains its DNA-binding capacity, masking detectable change in its chromatin enrichment and mobility. Also, PALB2 in vivo chromatin association is limited to a small fraction of periodic genes, hence FRAP assay may not be sensitive enough to detect minute but critical differences. We will conduct biochemical assessment of the ChAM 7R mutant and ChIP-qPCR analyses to assess PALB2 binding to specific genes, results of which will be included in the revised manuscript (Experiments 1 and 2).

      4 - It would be better to remove the data presented in Fig. 5 since, as currently presented, these investigations remain shallow and do not bring much information on what is happening. The presented data are rather confusing since, in the absence of further investigations, it is not clear which one(s) of the mechanisms involved in the control of DNA replication is controlled by PALB2 and many explanations, including artefacts, remain possible.

      The manuscript would gain in interest if the authors would devote the functional studies only to the repair part (Fig.6 and 7).

      We feel it is important to show Fig 5, as although the results may appear confusing, they highlight the importance of the acetylation of the 7K patch at the cellular level. Namely, the non-acetylatable 7R mutant fails to support normal cellular growth, likely due to its impaired association with active genes (Fig S5), which might provide in vivo evidence that non-acetylation of the 7K patch promotes PALB2 release from chromatin (please refer to our response to Reviewer 1’s point 3). We are confident that we will be able to clarify this point with our additional ChIP-qPCR analyses (Experiment 2).

      **Minor points** 5 - High background of non-enzymatic acetylation of PALB2 fragments makes the identification of KAT2A/B specific acetylation not very convincing. The immunoblot detection of acetylation fragments shown in Figure S1 is much more convincing. Therefore, the authors may consider to present Fig S1 as a main Figure and Fig.1B as a supplementary one.

      We will swap or add Fig S1B with or to Fig 1B in the revised manuscript.

      6 - It would be interesting if the authors would comment on why the presence of regions flanking the ChAM domain (Fig. 1A, construct #5) significantly reduces chromatin (Fig. 1B) and nucleosome binding (Fig. 1C).

      We are grateful for this Reviewer’s comment. Indeed, we noticed that the inclusion of the ChAM C-terminal flanking region perturbs its chromatin association. This region is highly enriched with serine and threonine residues which could be targeted for phosphorylation by cell cycle regulators (CDKs and PLK1) and DNA damage-responsive kinases (ATM and ATR). It is therefore tempting to speculate that, when phosphorylated, this flanking region could mask the basic patch of the ChAM, hence facilitating the release of PALB2 from undamaged chromatin region and its recruitment to sites of DNA damage. In the revised manuscript, we will provide the complete list of PTMs and discuss this point.

      Reviewer #3 (Evidence, reproducibility and clarity (Required)):

      KAT2-mediated acetylation switches the mode of PALB2 chromatin association to safeguard genome integrity

      The authors describe a series of experiments examining the consequence of acetylation, within a defined motif (Chromatin Association Motif; ChAM), on the cellular roles of the protein PALB2 (Partner and Localizer of BRCA2).

      The key conclusions drawn by the authors are generally convincing and are supported by the presented experimental results, which indicate that acetylation of PALB2 by KAT2A/KAT2B modulates its cellular behaviour and response to DNA damage. However please see specific comments below:

      **Major Comments**

      Expression of full-length PALB2 in the heterologous host E. coli is highly problematic, as the WD40 domain is generally not correctly folded. The authors use the ArticExpress strain to try and solve/alleviate this problem - but it is clear from the materials and methods section that an ATP-wash step has had to be introduced in order to release the recombinant protein from the chaperone system encoded by the ArticExpress system; i.e. indicating poor / mis-folding. Whilst this does not strictly have an effect on the results presented in Figure 1 (detection of in vitro acetylation sites), they have implications for the wider scientific community, as this may lead to the erroneous assumption that is possible to produce functional / folded full-length PALB2 in this way.

      We apologise if the manuscript conveyed the message that we are able to produce functionally active, full-length PALB2 in bacteria, which was clearly not our intention. Our aim was to test whether KAT2A was able to acetylate PALB2 in vitro. We agree that the folding and the biochemical properties (e.g. WD40-mediated BRCA2 binding) of the bacterially produced full length PALB2 were not fully assessed. We believe that this does not affect the overall conclusions of this study. In the revised manuscript, we will correct this error to make this point clear.

      In vitro modification assays are prone to producing post-translational modifications that are not fully reflective of those observed in vivo, and therefore need to be treated with some caution. This is highlighted by the relatively low modification of K438 in vitro by KAT2A; esp. as this is an acetylation site that has been previously mapped in vivo (by the authors). It would have been useful to include / see the effects on PALB2 function in vivo by modification / alteration of this single site.

      We appreciate the Reviewer’s constructive comment. Redundancy of acetylation acceptor residues within a lysine cluster is common, as is also the case for many ubiquitination events, hence we analysed the 7K patch mutant for phenotypic studies. For the same reason, we trust that the outcome of the characterisation of a K438 mutant would not significantly change our conclusions.

      Figure 3C and Figure 3D do not fully support or reflect the conclusions drawn by the authors - any peptide containing a cluster of positive charged residues are likely to interact with DNA through charge neutralisation of the phosphodiester backbone, concomitantly any alteration to this region of charge (i.e. via acetylation) will perturb this interaction.

      We totally agree with the Reviewer’s view and state, in the main text referring to the results shown in Fig. 3C and D, that “As anticipated, lysine acetylation, which neutralises the positive charge on the lysine side chain, conferred reduced affinity for negatively charged DNA”. In the revised manuscript, we will make this point clearer.

      Furthermore, experiments performed with the synthetic acetylated peptides do not agree with those carried out with the GST-ChAM constructs - GST-ChAM interacts with the nicked and linear forms of the pBS plasmid (Figure 2F) but does not interact with the supercoiled form. The WT synthetic ChAM peptide, in contrast, interacts with all three plasmid states at high concentrations. It is suggested that these two figures are removed.

      It is true that we cannot exclude the potential difference between GST-ChAM and synthetic ChAM peptide: for example, 26 kDa of GST, which can form a dimer, might mask the full biochemical properties of ChAM in DNA binding. However, we believe that the difference is more likely caused by the concentration of ChAM used. While we used the synthetic ChAM peptides at concentrations of 2.97, 5.94, 29.3 µM for Fig. 3C, we used 5.94 µM of GST-ChAM for Fig. 2F, for which we apologise for the omission of the experimental conditions used. This notion is supported by the side-by-side experiment, which was not shown in the original manuscript. In the revised manuscript, we will make these points clear.

      p. 18 : the authors used a PALB2 variant, where the lysines in the 7K patch are mutated to arginine - but don't fully characterise the effects of introducing these particular mutations on the ability of the ChAM fragment to bind to DNA, or indeed to nucleosomes; this is an important control.

      We appreciate the Reviewer’s comment. Biochemical analyses of the 7R mutant were not conducted, as ChAM produced in bacteria is not expected to be acetylated. Nonetheless, as also linked with the concerns of Reviewers 1 and 2, we recognise the importance of 7R biochemical characterisation for accurate interpretation of in vivo phenotypes. We will assess the DNA and nucleosome binding of the ChAM 7R mutant, which will be included in the revised manuscript (Experiment 1).

      Figure 6 : it would be good to show a second supporting example for deacetylation of PALB2 in response to DNA damage - perhaps treatment with MMC?

      We appreciate the Reviewer’s comment. Indeed, we have conducted the analysis upon MMC and Olaparib exposure. Curiously, however, no clear change of ChAM acetylation was detectable. Note that, for this experiment, we assessed the acetylation level of a GFP-fusion of ChAM, exogenously expressed in HEK293, along with endogenous gamma-H2AX as a readout of DNA damage signalling. Unlike ionising radiation, which triggered strong induction of gamma-H2AX (Fig. 6), no clear increase of gamma-H2AX was detectable upon MMC/Olaparib exposure. Hence, we propose that the reduction of ChAM acetylation reflects the cellular response to DNA damage. We will make these points clear in the revised manuscript.

      **Minor Comments**

      p. 16 : 'Our MS analysis of the chromatin-associated GFP-ChAM fragment identified actelyation of all seven lysines within the 7K-patch (Fig. 3A, marked with arrows).

      This part of the manuscript is potentially a little confusing, as Fig. 3A references a series of synthetic peptides rather than the GFP-ChAM fragments themselves.

      We appreciate the Reviewer’s point. Indeed, Fig. 3A shows 1) MS of the chromatin-associated fraction of GFP-ChAM (the top part with arrows) and 2) a schematic diagram of synthetic peptides that we used for biochemical analyses (the bottom part). In the revised manuscript, we will clarify this point and indicate the MS result and the schematic of synthetic peptides in two separate panels and refer each of them appropriately.

      p. 20 : Furthermore, using the FRAP approach, we observed clear differences in diffusion rates of FE-PALB2 following damage by IR, MMC, or olaparib treatment... FE-PALB2 = FL-PALB2?

      We apologise for the confusion. In our study, FE-PALB2 refers to Flag-EGFP tagged PALB2 (full-length). This is defined in the text “To this end, a tandem FLAG- and EGFP-tagged full-length wild-type (WT) PALB2 (FE-PALB2)” (p. 17).

    1. Overall, I think you have a great story. There is definitely a beginning middle and end that work cohesively. I am most interested in the organization and it taking over the government. It then eventually did a lot to change society which was cool, and you described the different things well. I know you probably did it on purposely, but congress, state and local governments were left out. I’m guessing in Healthment’s takeover those institutions ended? I also think that you could consider removing your “prologue.” It may not be necessary given that you do mention many of the things in that part throughout the story. For me if I read the story without that part, I think I would focus mostly on the environmental and health affects that Healthment’s formation and governance had on society. You did a pretty good job referencing and including the city as a character as Dr. Bush “asked” for. I think the chapter about Lairen going to get her mom may not be necessary to your overall plot. Lairen and Xavier talking and becoming even closer could probably happen anywhere or anytime in your story. Regardless of some of my “critiques” you have a story that resonates with me and probably many others when it comes to health and that if the government forced it on people, we would be better off. I think though that maybe it would be more than 4% of the US that would say no to it. Great conflicts and resolutions and an interesting outlook and story of a future where a health/environmental group runs the country.

    1. Late-twentieth-centurymachines have made thoroughly ambiguous the difference betweennatural and artificial

      Machines have progressed in such a way though that the previously formed boundary between human and machine is not clear. No longer is the distinction so clear as: humans=consciousness/subjective experience/natural machines=coded devices/bound to man's limitations/artificial Though...I kind of don't think machines are close to consciousness (have the ability to experience "what it is like" to exist) in these progressive technologies, neither when Haraway was writing nor 2020. This may not be all about consciousness but I still think it is at the root of this distinction. It moreso seems like we are just attributing complexity to machines which are human created nonetheless...e.g. I may just not know enough about machines but how are they "self-developing"? Or is she saying they just seem self-developing and that is enough to have social implications?

    1. And yet I need to say here that silence is not always or nec-essarily oppressive, it is not always or necessarily a denial or extinguishing of some reality. It can be fertilizing, it can bathe the imagination, it can, as in great open spaces-I think of those plains stretching far below the Hopi mesas in Arizona-be the nimbus of a way of life, a condition of vision.

      Beautiful! Silence as a condition of vision. This is brilliant!!!! It made me so happy when I found a way, a phrase that could express why I love the classes Prof. Annabel teaches. I wrote in a piece of paper in order not to forget: "Prof. Annabel makes me believe that the feeling I sometimes have --the feeling of having something to say-- is not false, is not illusory." But now, having read this passage, I may add more to it! I can explain it better. Your classes, the texts you choose for us to engage with, they allow for us to find a path, an always unique path, in which we can celebrate our silence. Not to suffocate with what is not said, but to "bathe [our] imagination" so that when we do break it, it is never to further harm our human relationships. That's so beautiful!

    1. Note: This rebuttal was posted by the corresponding author to Review Commons. Content has not been altered except for formatting.

      Learn more at Review Commons


      Reply to the reviewers

      Reviewer #1 (Evidence, reproducibility and clarity (Required)):

      **General comments**

      The manuscript 'Second messenger control of mRNA translation by dynamic ribosome modification' is a very interesting follow up on the research performed by the authors published in 2016. Here, the authors continue their study by determining the impact of the intricate RimABK pathway in Pseudomonas fluorescens on translational reprogramming by controlled modification of ribosomal protein S6 in response to environmental signals. The manuscript is interesting and well written, and the results are sound. However, in my opinion the general conclusion is not supported by experimental data and leaves several potential explanations open. Thus, I suggest to either perform in vitro translation experiments using ribosomes equipped with glutamated S6 to validate translational selectivity, or to soften the language on the working model shown in Figure 12.

      The authors would like to thank reviewer 1 for their detailed review of our manuscript. We agree with the reviewer that alternative explanations are possible for the translational changes linked specifically to glutamation, as opposed to rimBK deletion. Our intention when writing the discussion was to clearly distinguish glutamation-specific effects from the large number of indirect translational changes associated with Hfq disruption and other uncharacterised consequences of rimBK deletion. With hindsight, we acknowledge that the discussion and the model in figure 12 should more clearly outline the possible alternative causes for the observed glutamation-specific translational regulation. We have modified the discussion and figure 12 (now figure 10) accordingly.

      The reviewer further suggests that we perform in vitro translation experiments using ribosomes equipped with glutamated S6, to prove that glutamation controls translation directly. This is an excellent suggestion that would clarify this important point, and we will certainly attempt it as part of our future analysis of the Rim system. However, at this stage we feel these experiments are beyond the intended scope of this paper, which is to describe the signal inputs and mechanism of the RimABK system and to show evidence for both specific and secondary translational effects of ribosome modification.

      **Specific comments**

      Figure 1 and S1: The RT-PCR analysis shown here does not allow excluding transcription initiation at alternative promoters downstream of the one determined by 5'-RACE. However, an alternative promoter might contribute to relative ratios between the rimA, rimB, and rimK mRNAs. A Northern blot and/or primer extension analysis could clarify this assumption and would give more detailed insights into the specific regulation.

      The reviewer is correct that alternative rim promoters could exist downstream of the amplified 5'-RACE region. To test this hypothesis, we conducted additional RT-PCR experiments to measure expression of rimA (the third gene of the polycistronic rimABK operon) in the same set of conditions as tested for rimK. Relative levels of rimA mRNA do not substantially differ from those seen for rimK, strongly suggesting that the promoter upstream of rimK controls expression of all three rim genes. We have added this dataset to figure S1 and have modified the relevant sections of the text.

      Figure 2B: I'm confused by the results shown here! I do only see a reduction of RpsF in the presence of RimA, RimK and cdG. What indicates the modification? Please, explain the interpretation of the result in more detail. Shouldn't the modified RpsF shift due to the addition of glutamate residues?

      The uncontrolled activity of RimK acting in the absence of RimB (e.g. the experiment represented in Fig 2B) typically results in a reduction of the unmodified RpsF fraction in the reaction, replaced with RpsF proteins with widely varying numbers of glutamate residues attached to their C-termini. The resulting modified RpsF fraction can appear as a smear of protein density throughout the gel. We have clarified the text surrounding figure 2 to make this more explicit.

      Figure 2C: Why does the RpsF modification lead to a supershift? How many glutamate residues are added? Is the smear visible in lane 4 (RpsF+RimK) representing already the slightly modified RpsF protein, which upon addition of RimA results in a supershift? For all SDS-Page analyses shown in the manuscript the validation of the glutamation using the antibodies specific against poly-glutamate would be a great asset to facilitate their interpretation.

      Pseudomonas fluorescens RimK appears to have unregulated ligase activity, with many hundreds of glutamates being added to each RpsF protein in the absence of RimB cleavage. In our 2016 paper (Little et al., PLoS Genetics) we use radiolabelled glutamate incorporation and mass spectrometry to show that the supershifted protein smear is composed entirely of RpsF units with C-terminal glutamate tails of varying length. (It is interesting to note that E. coli RimK, which does not have an accompanying RimB protease, can only add 4-15 glutamates to each RpsF protein). We have modified the text slightly to make this clearer.

      The reviewer’s suggestion to stain the supershifted RpsF with the poly-E antibody is interesting but would likely only reiterate our published results with radiolabelled glutamate (Little et al. 2016).

      Lines 236-238: '...strongly suggesting that the proteomic changes we observe are an active response to modification of ribosomally-associated RpsF proteins.' This is an important suggestion as it allows a flexible and very fast integration of the external signals into a specialized protein synthesis. Thus, it definitely deserves further analysis! Considering that the purified RimA and RimK proteins are available, in vitro modification of RpsF in the context of the purified ribosome would be an important experiment and would greatly increase the quality of the paper. Up to now the selective or specialized translation is pure hypothesis and might also be explained by indirect effects via e.g. increased interaction between the ribosome and HFQ that might mediate interaction with certain mRNAs and thus stimulate their translation.

      We agree with the reviewer that direct measurement of translational changes in vitro would tell us a great deal about the mechanism of RimK regulation. This would enable us to confirm whether the glutamation-specific effect is direct, or if it functions through an as-yet uncharacterised indirect mechanism (such as interaction with another translational regulator). As stated above we feel these major experiments are beyond the scope of the current manuscript, although we are keen to do them (as part of a planned structural biology investigation of modified ribosomes). As stated elsewhere in our response, we have extensively revised the discussion text and figure 12 to clarify the limits of our current understanding and highlight the different potential regulatory routes for RpsF glutamation.

      Lines 322: '...into a single output: the proportion of all ribosomally-associated RpsF proteins that have C-terminal poly-glutamate tails.' Considering the identification of a group of genes whose translation is altered by rimBK deletion, but not by RpsF glutamation (Class 1, Fig 11B), I would suggest softening this statement. If I interpret the data correctly, they pinpoint to a moonlighting function of the rim-pathway that does not target RpsF!

      The genes whose translation is affected by rimBK deletion, but not by RpsF glutamation specifically, include all those genes whose translation is indirectly affected by downstream translational regulators, or through interaction with another affected gene target. As expected, there is substantial overlap between the rimBK and hfq translatomes (Grenga et al. 2017): this analysis can be included in the manuscript as a supplementary table if requested. Importantly, there is very little overlap between the Hfq translatome and those genes that are affected specifically by RpsF glutamation. One possibility is that Hfq interacts with RimK at the ribosome, and the loss of the RimK protein is a major factor in destabilising Hfq function in the ∆**rimK mutant. We have modified figure 12 (now figure 10) and expanded the discussion to include this hypothesis.

      While we cannot exclude the possibility that RimK has other cellular targets in SBW25, we think this is unlikely to be a major cause for the results we see here. We have carefully examined the C-terminal peptides of proteins detected in our various proteomic assays and are confident that RpsF is the sole target of RimK in SBW25 under the conditions we tested. We also directly tested RimK interaction with purified Hfq and confirmed that Hfq is not a direct target of RimK modification.

      Lines 377-76: '...distinguishing features in the primary or predicted secondary structures of the Rim-mRNAs...' As mentioned already above several indirect options are still open that could confer selectivity to the ribosome.

      As stated above, the discussion has been rewritten to more completely reference the possible mechanisms by which RpsF glutamation may lead to translational regulation.

      Reviewer #1 (Significance (Required)):

      The key concept of the manuscript namely the impact of the intricate RimABK pathway in Pseudomonas fluorescens on translational reprogramming by controlled modification of ribosomal protein S6 in response to environmental signals is novel and will significantly impact the field.


      Reviewer #2 (Evidence, reproducibility and clarity (Required)):

      **Summary**

      The main question addressed by this research is how bacteria adapt to rhizospheric niche through the RimK ATPase glutaminase. This enzyme post-transcriptionally modifies the ribosomal protein RpsF in a process of complex regulation. Regulation is mediated by c-di-GMP that is degraded by the phosphodiesterase RimA and the protease RimB exerts a role opposite of RimK. Novel findings include the finding of RimK acting as a four-state ATPase, depending on the binding of RimA, c-di-GMP or both. Another important finding is the opposite roles of RimK and RimB on the glutamation/deglutamation of RpsF and the tendency to a steady state of four glutamate residues in the RpsF protein. The authors also use proteomics to determine the effect of glutamation, specially at low temperature and under nutrient limitation.

      We thank the reviewer for their positive review of the manuscript and address their comments below.

      **Major comment**

      In my opinion, the results obtained with the Hfq regulation by RimK blur the message. I firmly think that the Ms is very solid with the results obtained in relation with the RimABK/RpsF regulation in P. fluorescens shown as a model in the Figure 12. Moreover, in this final model presented by the authors (fig. 12) they not included the results related with Hfq. These results could be part of another paper.

      We agree with the reviewer that the Hfq independent effects of RpsF are an exciting finding and should be a major focus of the paper. That said, we feel that the additional work we have done showing how Hfq is affected by RimK should also be retained in the manuscript in some form. Our data (e.g. figure 8) indicate that Hfq is responsible for a large (indirect) fraction of the ∆rimK phenotype, so understanding how it is affected is important to understand how RimK functions. Based on comments from reviewers 2 and 3 we have reviewed the manuscript text (including data on Hfq) to make the narrative as focussed and clear as possible. We have also redesigned figure 12 (now Fig 10) to consider comments from all three reviewers and have changed the text in the discussion to match this.

      **Minor comments**

      In figure 4A, what is lane 5?

      Lane 5 contains RimB without ADP. The figure legend has been modified accordingly, and we thank the reviewer for highlighting this error.

      Line 159 change "suppression of RimK band-shifting" by "suppression of RpsF band shifting"

      This has been fixed.

      Reviewer #2 (Significance (Required)):

      The Ms. is very interesting and deeply describes the relation between environmental conditions, c-di GMP second messenger and the RpsF ribosomal protein posttranscriptional modification in order to respond to low temperatures and changes in nutrient availability. The research developed in this manuscript is original and novel in the field and includes new advances in the signal transduction pathways implicated in the regulation of bacteria adaption to the environment. Besides, the research design and technical methodology is original and includes multidisciplinary approaches of interest to the research community in general.


      Reviewer #3 (Evidence, reproducibility and clarity (Required)):

      **Summary** Post-transcriptional control of protein abundance is an important yet poorly examined regulatory process by which bacteria respond to signals found in the environments they grow in. The authors' team have previously identified, described and published details around a novel regulatory pathway involving the ribosomal modification protein RimK, regulator proteins RimA and RimB, and the widespread bacterial second messenger cyclic-di-GMP (cdG). In the current manuscript builds considerably on those previous findings and goes several steps further, through various approaches including protein biochemistry, computational modelling, quantitative proteomics and ribosomal profiling, the authors described how the RimABK pathway as a novel translator system that controls bacterial adaption to the rhizosphere in the bacterium Pseudomonas fluorescens. They show that the system achieves this through specific controlled modification of the ribosomal protein RpsF. I read the article with excitement and overall the manuscript describes an extensive data set that will be of considerable interest to many readers in several fields. However, I have made a few points below that the authors need to take on board and address. If these issues are addressed, I believe it will make the presented data much clearer to the reader, tidy up a few ambiguities and make the article a little more accessible to many non-specialist readers.

      We thank the reviewer for their thorough and positive assessment of the manuscript. We address their specific points below.

      **Major Comments**

      1) The major finding described in the manuscript and the one that will be of significant interest to reader is that a novel post-translational ribosomal modification regulatory mechanism involving Rim system controls bacterial adaption. The second messenger cdG only plays a small part in this complicated process. Therefore, I believe the title needs to be revised to capture the scope and key findings of the manuscript.

      We are happy to change the title along the lines suggested by the reviewer. We propose: Control of mRNA translation by dynamic ribosome modification as a new title.

      2) The authors present a lot of interesting data; however, I found the manuscript a bit of a dense read. I find the key findings are diluted within the text. I would ask that the authors to make it a little more focused. For example, on the regulatory role of RimK and its influence on Hfq and RpsF has been detailed previously so could be placed in supporting information and briefly mention when required. Also, the experiments on the pvdIJ pathway could be removed or placed in supporting information as they are not the main focus of the manuscript. Fig 5 and 6 could be combined as one figure as well.

      We have modified the manuscript throughout to make it clearer, more concise, and to focus as much as possible on new findings rather than reiterating what we showed in our last manuscript. In line with the reviewers’ recommendation, we have moved the pvdIJ data into the supplementary material (Fig S3) and merged figures 5 and 6 into one. In addition, to support our data on the importance of RpsF glutamation for ribosomal regulation we used Western blotting to confirm that RpsF4/10glu variants incorporate normally into SBW25 ribosomes in vivo (added as supplemental data Fig S5).

      As stated elsewhere, we feel that key data on the relationship between Hfq and RimK should remain in the main manuscript, although we have reviewed the text thoroughly to try to ensure it is as focussed as possible and have moved some results to supplementary material as suggested.

      3) The authors propose a four-state kinetic model for RimK ATPase activity with RimA and cdG (described in Fig2 and Table S1). However, later in the manuscript the authors demonstrate that RimB also stimulates RimK ATPase activity, but this seems to have smaller impact than RimA and cdG (Fig 2E, Fig 3A). Why RimB was not included in the ATPase kinetic model of RimK? Does including the RimB data suggest there might be more conformational states for RimK?

      Thank you for raising this point. The reviewer is correct in that this data does indeed suggest another level of ATPase activity of RimK. We have added text to the manuscript to reflect this. We have also extended the supplemental Table S1 to include these equations.

      4) The authors claim that the suppressive effect of cdG on RimK was depended on the enzyme activity (PDE domain) of RimA. This was tested using an enzymatically inactive RimA variant (RimA-E47A). However, in Fig 3E the amount of RimA-E47A used in the assay seems to be significantly less than wildtype RimA. Additionally, in Fig 2B, the authors show that addition of cdG also stimulates RpsF modification with or without RimA (lane 4-6). I would ask the authors to clarify these points.

      It is difficult to directly compare protein variants due to differences in solubility post-purification. Due to difficulties in purifying this (less soluble) form of RimA, co-purifying contaminants have also probably influenced the determination of RimA-E47A concentration to some extent. This restricts us to making largely qualitative statements about protein function, as we do here. Despite its poor solubility and low concentration, RimA-E47A is still able to stimulate RimK. Furthermore, the relatively low concentration of RimA-E47A in our assays would render it at least as susceptible to any effects of cdG addition as WT RimA, meaning we can be confident that cdG has no effect on RimK stimulation by this variant.

      Our model incorporates direct stimulation of RimK by cdG alongside its effect on RimA. We show evidence for this in this manuscript and in our 2016 paper.

      5) The authors claim that high levels of cdG increase the ratio of RimB protease activity to RimK glutamate ligase activity. However, there is no experiment to provide direct evidence to support this. Please tone down the language used or provide evidence. On the same point Fig 6 was not explained in the main text to support this conclusion. Please include an explanation.

      The hypothesis that high cdG levels favour RimB activity over RimK stems from the observation that cdG suppresses RimK activity (by abolishing RimA stimulation) but does not affect RimB. We have data showing that increasing cdG levels suppresses RpsF band shifting in vitro in an assay containing all three Rim proteins (Fig 4). However, we agree the hypothesis that cdG controls the ratio of RimB to RimK activity by controlling the activity of RimK currently lacks explicit, direct evidence and we have modified the text to tone down the language.

      An explanation for Figure 6 (now 5b) has been added to the manuscript as requested.

      6) In some of the figures/images, for example, Fig2B and Fig 3E, RimA is shown as a major band. However, in other figures/images, for example, Fig 2D, Fig 3D, RimA seems to be two bands. The authors should explain the reason for this.

      Based on extensive experimentation, we are confident that the second band present in some of our assays is a cleavage product of RimA. This is an experimental artefact that is linked to concentration and protein stability in vitro. We must stress that the presence of an inactive fraction of RimA in our assays does not affect the conclusions we are able to draw from these experiments. A note has been added to the relevant section of the text.

      **Minor comments**

      • Line 151, should be RpsF band-shifting instead of RimK.

      • Fig 4A there is no legend for lane 5, which made it very difficult to understand the data presented.

      Please see above. These two minor errors will be fixed.

      • The layout of some figures could be improved.

      We have revised the layout of several figures, in line with the reviewer’s suggestion.

      • If it is possible to have Fig 11 as a Venn diagram or some intuitive diagram, it will help the readers gain access to the data and understand the results.

      We respectfully disagree with the reviewer here. We have tried several different presentation styles for these data, but ultimately considered scatter charts to be the most effective, in line with our previous study of Hfq regulation in Pseudomonas (Grenga et al. Frontiers in Microbiology 2017).

      Fig 12 is very neatly laid out. However, I don't feel it captures the dynamic nature of the system. I am just wondering if the authors could break it down so that it describes the changes relating to environmental conditions and/or different cdG levels?

      Figure 12 (now Figure 10) has been modified to reflect to comments of all three reviewers.

      Reviewer #3 (Significance (Required)):

      The manuscript provides detailed evidence to demonstrate a dynamic, post-translational ribosomal modification mechanism which is an important feature of prokaryotic (potentially archaeal and eukaryotic) environmental adaptation. This is an exciting manuscript and one many will wish to read. The data provided will be of interest to scientists working in many fields including microbiology, biochemistry and plant pathology.

      I have several areas of expertise including genomics, molecular microbiology, small molecule signalling and regulation, micro-host interaction, adaptation,

    1. Skule (1999) describes how an inter-organisational group of workers from five food-and-drink companies were taken through a training program that included "practice in other companies". Says Skule, "Most of the skilled operators described [the experience] in terms like "see things differently", “opened my eyes”, “think more about what I am doing”, “more alert” and “think more about the consequences”. These new perspectives or ways of seeing in turn made operators attend to features in their work situation in a new way. From a former habitual way of working according to minimum standards, many skilled operators developed a more reflectively skilled way of performing their job, within the limits of existing job structures and routines." We believe this kind of benefit may not be as often used as is possible.

      Having teachers participate in laboratory classroom residencies has had this impact, but communicating the importance to experts is a challenge, and the tyranny of experts is to deny the importance of practitioners gaining insight or seeing new possibilities.

    1. How do you think the “inner” and “outer” landscapes apply to the development of the teacher?
      From what I have been able to gather about the “inner” and “outer” landscapes as presented by Parker Palmer, Laura Rendon and the AACU suggests to me that good teachers are genuinely motivated by not only passion for their field and for the reward reflected in the academic successes of their students, but also the personal growth of their students.  Teaching isn’t something they happen to do but something they must do in order to facilitate students’ social consciousness and empowerment. When we encounter students who may be perceived as unmotivated or lazy, we kick it into gear and get creative with approaches to engaging students where they are. Instead of seeing our students from a deficit perspective, we look for their assets—the qualities of our students that can help them learn, such as their bilingualism, creative interests, hobbies or talents outside of our discipline. What we value in our students isn’t a reflection of what we want them to be, but what they want for themselves. We trust our students’ innate capacity to learn and to acquire confidence in being a college student.
      
    1. Dayadhvam: I have heard the key Turn in the door once and turn once only We think of the key, each in his prison Thinking of the key, each confirms a prison Only at nightfall, aethereal rumours Revive for a moment a broken Coriolanus

      Voices: The voices speak of a key that would either lock/unlock the prison they are in. Whether it is real and they believe it or just a fictitious rumor is either possibility. Yet it seems they want freedom from something. Un/natural time/Desire frustrated (maybe?): If their thoughts are mostly on this key, are they keeping track of the passing time other than day and night? How many days are spent in this prison they believe they are in? Exile: They seemed to be trapped in a prison, in search of a key that may or may not free them. Fragments: The lines go back to the key for three of the lines and prison for two lines. These moments seem to only pause when it is night. Text as plagarism/remixed (maybe?): Coriolanus was mentioned. After looking it up, one result was a tragedy Shakespeare wrote.

    1. Note: This rebuttal was posted by the corresponding author to Review Commons. Content has not been altered except for formatting.

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      Reply to the reviewers

      Reviewer #1 (Evidence, reproducibility and clarity (Required)):

      In this study by Wegwitz et al, the authors examine the tumour promoting properties of RNF40 (and the H2B monoubiquitinylation catalysed by it) in Her2 driven breast cancer.

      They report, using publicly available data, that increased RNF40 expression is associated with reduced overall and disease-free survival.

      Using a mouse model, where they crossed the Erbb2 (mouse Her2) under the control of the MMTV promoter with conditional Rnf40 deletion constructs, the authors found that deletion of Rnf40 simultaneous to Her2 overexpression resulted in a prolonged tumour-free survival, somewhat reduced tumour growth kinetics and tumour incidence.

      siRNA silencing of Rnf40 in two Her2 positive breast cancer cell lines resulted in reduced proliferation, clonogenicity and tumour sphere formation and cellular motility.

      Transcriptome analysis revealed pathways that could explain the phenotype, like increased apoptosis and actin cytoskeleton regulation. The authors then took further some candidates in the later pathway to investigate the mechanism. They find that Rnf40 loss impacts on actin cytoskeletal dynamics. They also investigate the impact on focal adhesion signalling integrity.

      Finally, they investigate the relationship between the transcriptome and H3K4me3 and H2Bub1 landscape in the presence or absence of Rnf40.

      The manuscript is convincing regarding the tumour promoting roles of Rnf40, but the key claim that H2B monoubiquitinylation is essential for activation of the Rho/Rock/Limk pathway, where genes are down regulated upon Rnf40 loss resulting in decreased tumourigenicity of cells, is so far not convincing.

      "Together, these findings support the hypothesis that the actin regulatory gene network is dependent on direct epigenetic regulation by RNF40 through modulation of H2Bub1 and a trans-histone cross-talk with H3K4me3 levels in HER2-positive BC cells."

      Although the correlation is apparent, at this point it's unclear if the phenotype is dependent on the catalytic activity of Rnf40 or it's a non-catalytic effect. Generating a catalytic mutant RNF40 and test it at least in the cell lines studied would be desirable.

      We thank the reviewer for this comment and agree that the addition of data with a catalytic mutant RNF40 could strengthen our findings and further clarify mechanisms involved. Thus, in a resubmission we will directly address this point by performing knockdown/rescue experiments with either wildtype or a RING finger mutant RNF40. This will be done by transfecting cells with expression constructs for either wildtype or mutant RNF40 proteins followed by knockdown of endogenous RNF40 using siRNAs targeting the 3’ UTR. Experiments central to our take-home message will be performed (e.g., cell migration, target gene expression, Western blot for H2Bub1, F-actin formation). Together, we hope these experiments will help significantly solidify the message of this paper and further clarify the individual role of RNF40 within the RNF20/40 heterodimer.

      **Other comments that need a response:**

      1."we investigated RNF40 expression and H2Bub1 levels by immunohistochemical staining of 176 primary BC tumors and 78 brain metastases."

      In Fig 1 I can only count 41 primary BC tumours and 73 brain metastases. Numbers don't add up. Also, how is "low" defined as opposed to negative? What is used as controls?

      We apologize for this mistake. We corrected the numbers of primary and metastatic HER2-positive specimens used in this study.

      2."Moreover, HER2-positive metastatic BC samples showed a particularly high expression of RNF40 compared to primary tumors"

      Figure 1 or Fig S1A does not contain data on HER2-positive metastatic BC

      We think there might have been a confusion regarding this point. The manuscript does provide information about RNF40 and H2Bub1-staining in primary HER2-positive breast cancer lesions as well as HER2-positive brain cancer metastasis specimens in Fig.1A-C as well in Fig.S1A (representative brain metastases are shown in IHC pictures). This is stated both in the main text as well as in the respective figure legends. However, if for some reason this remains unclear, we would certainly be open to suggestions as to how we can modify the respective sections to improve their clarity.

      3."tumors did not display a loss of either RNF40 or H2Bub1 (Fig. 1H) when compared to the adjacent normal mammary epithelium (Fig. S1F)."

      I don't understand what I see in Fig S1F, where is the tumour, what is adjacent?

      We agree with the reviewer that splitting tumor staining in the main Fig 1 and normal tissues in Fig S1 makes a comparison difficult. We will therefore edit the Fig S1F and provide there an overview of tumor and surrounding normal tissues together with magnifications of the respective areas. This should significantly ease a comparison of both RNF40 and H2Bub1 in tumor and adjacent normal tissues.

      4."homozygous loss of Rnf40 (Rnf40fl/fl) resulted in dramatically increased tumor-free survival of MMTV-Erbb2 animals (Fig.1E)." This is overinterpretation of the data, I would not call it dramatic, just significant.

      The MMTV-Erbb2 mouse model is a very reliable mouse model for the induction of HER2-positive lesions. In our hands, the tumor incidence in these animals was 100% with a median tumor free survival of 166 days. In comparison, approx. 20% of the Rnf40fl/fl animals (3 out of 14) never developed the disease during the 18 month observation. The animals that still developed lesions had a median tumor free survival of 241 days, which represents a delay of 75 days (45% delay). In light of this, it seems to us that the effect of RNF40 loss on HER2–positive lesions is, indeed, remarkably strong. However, we do not wish to give an impression of over-interpreting or misrepresenting the data. For that reason we modified the wording in the main manuscript according to the reviewer’s suggestion (line 140: “dramatically” was replaced with “pronounced”).

      5."loss of Rnf40 led to strongly reduced tumor growth kinetics (Fig.1G)." Is this result significant, I did not see an evaluation of statistical significance in this data.

      As suggested by the reviewer, we have performed additional analyses to examine the statistical significance. We have now included the results of these tests in the respective figure.

      6."Rnf40fl/fl lesions displayed a heterogeneous pattern of RNF40 expression (Fig.1H), suggesting that the few tumors that did develop in this model were largely caused by an incomplete loss of the Rnf40 allele." If this conclusion is suggested, the authors should check if the "escaper" cells have failed to flox the Rnf40 allele on the genetic/protein level. Otherwise it's not conclusive.

      The reviewer brings up an interesting and important point about the heterogeneous loss of RNF40 in “escaper” tumors. Very important to note is that these “escaper” tumors developed significantly later and three animals never developed tumors. Thus, the “escaper” phenotype is rare (at the cellular level) and is likely similar to the selective process that occurs in human tumorigenesis and tumor progression. It is well established through a number of publications that deletion of genes essential for tumorigenesis via Cre-based systems frequently results in a specific selection for the rare instance that the Cre-mediated excision is ineffective. These “escaper” cells can then grow out and proliferate because they do not suffer from deletion of the floxed allele. This effect has also been established when combining MMTV-HER2 and MMTV-Cre. For example, analogous findings were recently published by Costa, et al., in Nature Communications (doi: 10.1038/s41467-019-11510-4) where the MMTV-Cre-mediated deletion of Pak4 resulted in impaired MMTV/HER2 or MMTV-PyMT-driven tumorigenesis, but occasional tumors did appear, which all retained some degree of PAK4 expression. This effect, which we have also seen in our system, was also reported by Miao, et al. in Cancer Research (doi: 10.1158/0008-5472.CAN-11-1015) in 2011. In their work the authors observed that deletion of the Myb gene also impaired MMTV-HER2-driven tumorigenesis and those tumors that developed in Myb flox/flox mice displayed a late onset and invariably retained MYB expression. Similar findings have been reported in a number of other tumor types and with various Cre drivers. Thus, we posit that these findings provide further support for the essential role of RNF40 in HER2-driven tumorigenesis to the extent that rare, RNF40/H2Bub1-expressing “escaper” cells are positively selected for during tumorigenesis and tumor progression.

      In order to definitively establish this, we propose performing dual immunofluorescence staining of Rnf40 flox/flox tumors to verify that H2Bub1 is exclusively and universally lost together with RNF40 and that each case of a complete loss of RNF40 also results in a complete loss of detectable H2Bub1 staining. Additionally, we will assess the efficiency of the cre mediated deletion of Rnf40 exons 3 and 4 in Rnf40fl/fl animals by detecting their presence using a conventional PCR approach.

      1. Fig S4D - is this clonogenic assay? How many replicates were done, biological technical?

      We apologize for the imprecise description of this figure. We edited the manuscript accordingly and included details about the number of replicates.

      8."Additionally, treatment with either CYM-5441 (Fig.4J) or …"

      Fig 4J is missing! It makes this section rather hard to follow. Fig S4F-G, how many replicates were done, biological technical?

      We thank the reviewer for noticing this error. The figures were indeed inappropriately referenced in the text. This error has been corrected.

      9."Consistent with our analyses based on changes in H3K4me3 occupancy, genes downregulated upon RNF40 silencing displayed the most prominent decrease in H3K4me3 in the gene body (the 3' end of the peak)"

      The impact of these mods changes is hard to judge because they are rather small (I would not use the wording prominent).

      As implied by the reviewer, we will replace the word “prominent” with “noticeable”.

      Also, are there many other "peak narrowing" genes but they are not downregulated?

      The point mentioned here is very interesting. The bioinformatic analyses performed in this study solely focused on the relationship of significantly regulated genes and the H3K4me3 peak narrowing at their TSSs. However, we did not analyze the global regulation of genes showing H3K4me3 peak narrowing near the tSS. As this information might be of high relevance for this study, we propose to investigate this interesting aspect in the revised version of the manuscript.

      In fact, our analyses have revealed that a large fraction of genes with H3K4me3 narrowing peaks do not show an appreciable decrease in expression. To better understand the epigenetic features determining the sensitivity of genes to H3K4me3 peak narrowing, we studied the occupancy of several histone marks at differently behaving genes. We discovered that sensitive genes globally present lower occupancy of histone modifications which are known to positively influencing gene transcription. We therefore propose that the epigenetic context (i.e., presence of additional histone modifications) strongly determines whether the loss of H2Bub1, and ensuing narrowing of H3K4me3 near the TSS, results in decreased transcription.

      Statistical analysis missing: for example in Fig 2C, Fig 2E, Fig 3G what is n=?, how many technical, biological replicates were analysed?

      This information has been added to the revised manuscript.

      Fig 4E seems to be a partial duplication of Fig 3D!

      The samples in Fig. 3D and 4E originate from independent experiments. In Figure 4D, we indeed provide again PARP and Casp3 signal for siRNF40 samples in order to allow a direct comparison of the effect magnitude between RNF40 depletion and ROCKi treatment.

      **Minor:**

      Figure referencing: it can be quite confusing to see a different ordering of figures compared to the referencing in the manuscript, for example Fih 1H is referenced in the text before Fig 1F, G. The authors should change the order in the main figures....

      We thank the reviewer for pointing this out. We have updated the figure order in the revised manuscript accordingly.

      Reviewer #2 (Evidence, reproducibility and clarity (Required)):

      In this study, Wegwitz et al propose that the E3 ubiquitin ligase RNF40 is highly expressed in HER2+ breast cancer tumours and correlates with poorer survival, using their own and TCGA data. Contrary to observations suggesting a tumour-suppressive role in other cancers, authors show using RNF40-knockout breast cancer mouse models and in vitro data shat RNF40 promotes tumour growth. RNF40 depletion impairs proliferation, survival and sphere formation by inducing apoptosis. In addition, RNF40 promotes cell migration by upregulating expression of cytoskeletal proteins (ROCK1, VAV3, LIMK2) and their effectors such as phosphorylated cofilin. Authors show elegant partial rescue experiments of the effect of RNF40 depletion on apoptosis and survival.

      Given that RNF40 function seems to be context-dependent, findings from this study could have broad significance for other cancers with high RNF40. It also provides some mechanistic data (that should be improved as suggested below) linking this ubiquin ligase to the cytoskeletal machinery and, therefore, control of migration and also proliferation and survival.

      Data are well presented and most conclusions are supported by the data. However, there are some gaps at the mechanistic level. Since migration is controlled by RNF40 in vitro, evaluation of metastatic ability in vivo (local invasion for example as suggested below) should be evaluated and would strengthen this part too.

      **Major comments**

      1. Fig.1A-B, S1A. Specificity of RNF40 antibody should be shown, which could be done quite easily in the tumours from the knockouts. From the datasheets, antibodies recognize human protein only.

      We thank the reviewer for this suggestion and apologize for this mistake. The antibody utilized in the IHC studies is actually from Abcam (ab191309) and, in fact, recognizes both species. Table S5 has been corrected accordingly.

      It is unclear when the murine tumours were analysed, at endpoint? This should be stated.

      We thank the reviewer for this comment. Indeed, all IHC analyses were performed after dissection (endpoint). This information will be added to the manuscript. Kinetic analyses of tumor growth (i.e., Fig. 1G) were performed on the same mouse cohort.

      Could authors establish cell lines from the mouse tumours (knockout, partial knockout escapers..)? These could be very useful tools to evaluate key in vitro findings from the study.

      The reviewer makes an interesting suggestion. Unfortunately, we were not able to establish cell lines from this model and have since stopped breeding this mouse line (due to the relocation of the principle investigator). However, we did try to generate RNF40-deficient breast cancer cell lines using the CRISPR/Cas9 technology. Interestingly, all attempts failed, supporting the fact that the loss or RNF40 is lethal for the cancer cells. However, to further establish this, for the revision we propose to transfect HCC1954 cells with CRISPR/Cas9 constructs targeting exons 3 and 4, similar to our mouse model. We will then assess the evolution of RNF40-negative cells population over time (i.e., via immunofluorescence staining for H2Bub1). This assay should inform about the expected growth “disadvantage” following RNF40 loss.

      Fig.1F-G: since RNF40 controls the cytoskeletal machinery and therefore, migration (Fig. 2G) in the RNF40 knockout tumours, was metastasis (if observed) affected? Or if there was no growth in distant organs detected in the time frame of these experiments, was invasion (and/or pattern of invasion or mode of invasion (morphology of invading cells)) into adjacent tissues affected upon RNF40 depletion? This would add in vivo relevance to the in vitro mechanistic findings, especially since the authors later showed that p-cofilin was also decreased in the RNF40-depleted mouse tumours (Fig.4D).

      We agree that the metastasis data from our mouse genetic tumor model would significantly help solidify our findings. Unfortunately, the MMTV-Erbb2 mouse model (overexpressing wildtype Erbb2 gene) only rarely develops distal metastases. In our analyses, we only ever observed two macroscopically visible metastases (one wt/wt and one in an Rnf40flox/flox mouse). However, we feel that the reviewer’s suggestion is a one and will follow this suggestion and attempt to examine possible changes in local invasion of primary tumors into adjacent tissues.

      Fig.3: most results using HCC1954 cell line. Key findings should be validated in other cell lines.

      We agree with the reviewer about the importance of cross validation of findings using different cell lines. For this purpose, we have now generated data with an additional HER2-positive cell line. These data using the SKBR3 cell line were performed for several of the key experiments. Key findings about phenotypic changes (growth kinetics and colony formation), Ki67 protein levels differences and mRNA regulation of identified regulators of actin cytoskeleton (VAV3, ROCK1, LIMK2 and PFN2) will be included in the revision for both cell lines. Furthermore, as seen in the HCC1954 cell line, an increase of the apoptosis marker cleaved PARP as well as a loss of VAV3 and ROCK1 protein levels was also observed upon RNF40 knockdown in SKBR3 cells. These data will be included in the revised manuscript.

      Fig.3A: authors state "both pathways remained intact following RNF40 depletion". However, from those blots, siRNF40 clearly increases pERK and slightly pAKT, which would be unexpected according to previous data in Fig.2. Authors could show quantifications of different blots, or show a more representative blot if increase in pERK was not consistently observed. Was this also seen in SKBR3 cell line?

      We thank the reviewer for this comment. Initially, we had anticipated that oncogenic signaling may be decreased in the Rnf40 conditional knockout model. However, much to our surprise, the activity of the downstream signaling actually appears to be increased. In fact, the increase in AKT and ERK1/2 phosphorylation following RNF40 silencing was consistent across different experiments and replicates. While this finding is also consistent with our previous results in an ER-positive system (e.g., see Prenzel, et al., 2011), we do not understand the mechanistic underpinnings of this finding. Importantly though, while consistent, we do not feel that this increase explains the observed phenotype. Nevertheless, to more precisely show the overall change of p-ERK/ERK and p-AKT/AKT, in the revision we will provide a densitometry quantification for both cell lines. We will also modify the sentence to more precisely describe this finding and make the point that since these pathways are not reduced/impaired, they are unlikely to be responsible for the increased apoptosis observed upon RNF40-KD. Western blots assessing p-ERK/ERK and p-AKT/AKT levels in SKBR3 upon RNF40 knock-down will also be added into the supplementary data of the revised manuscript (Fig.S3).

      For Fig.3G and Fig.S3A, authors selected genes from this set, how was this done (fold change?). Was expression of the other family members (ROCK2, LIMK1, etc) or of Rho GTPases regulated too?

      This information was indeed missing in the manuscript. We have modified the figure legend and the main text accordingly in order to provide the information about the cutoff used in the Enrichr analysis. Regarding the expression of other family members of the actin regulatory network, in the past we performed a more, in depth and focused analysis of our RNA-seq data, broadening our view to other members of the RHO/RAC/CDC42 pathways. While we did identify a few further potentially regulated target genes (e.g. ROS1 or PAK6), these genes were either only weakly expressed or weakly regulated. For this reason, we presumed that these factors could only play a marginal role in the observed phenotype and have focused our attention on the robust part of the signature.

      Fig.4B: this may not help, decrease of p-cofilin by Vav3 knockdown is way less dramatic compared to RNF40 depletion or ROCK inh treatment. See comment below regarding other effectors such as Myosin.

      Indeed, the consequence of VAV3 loss on p-cofilin levels is less pronounced than the effect observed upon RNF40 knockdown or ROCK1i treatment. Given the fact that RNF40 loss not only affects VAV3 expression, but also has additional direct effects on the expression of other pathway members, this may be expected. We do, however, feel that the VAV3 regulation is likely one component of the effects of RNF40 loss. In addition, it has also been shown that VAV3 is not the only GEF regulating the activity of RHO kinases upstream of ROCK1. Therefore, we would also expect that VAV3 loss only partially reduces ROCK1 activity and therefore only partially phenocopies the effects observed. We will expand the description of these findings in the revised manuscript to reflect these views.

      Fig.4C: does ROCK inh reduce RNF40 levels? It may from the immunofluorescence picture.

      We thank the reviewer for this comment. In fact, we have examined this possibility. However, no significant changes in RNF40 protein levels were observed upon RKI-1447. If helpful, we can provide Western blot data demonstrating this in the supplemental figures.

      Fig.4H-I: the sphingosine 1-phosphate receptor-3 agonist (CYM-5441) partially rescued the effects of RNF40. Since S1P signalling involves Rho GTPase activation -presumably downstream of VAV3 -which is a GEF for Rho, Rac and Cdc42- and upstream of ROCK, LIMK, was activity of these Rho GTPases affected upon RNF40 depletion? This would strengthen the mechanism.

      The reviewer points at an interesting aspect of the actin regulation. Indeed we expect that the reduction of VAV3 levels upon RNF40 loss would significantly influence the activity of the downstream client GTPases. However, the measurement of RHO-GTPase activity is tricky and expensive. Furthermore, as mentioned in the previous comment (#7, part 1) VAV3 is only one component of the four major genes encoding critical actin cytoskeleton regulatory proteins regulated upon RNF40 loss, and the only factor upstream of RHO-GTPases. The reduction of downstream ROCK1, LIMK2 and PFN2 levels also influence the activity of this pathway downstream of RHO-GTPase activity. We therefore focused our efforts on assessing F-actin and p-cofilin levels as these may provide more sensitive readouts about the consequence of RNF40 loss on this signaling cascade. However, if the reviewer considers this information as indispensable, we would attempt to investigate changes in Rho-GTPase activity by commercially available Active Rho Detection Kits, although this will significantly delay the resubmission of a revised manuscript.

      Related to this, was Myosin II activity (phosphorylated MLC2) affected -since its upstream regulators, especially ROCK are controlled by RNF40?

      We thank the reviewer for this insightful suggestion. To address this possibility, we will test this hypothesis for the revised manuscript as suggested by performing Western blot analysis for phosphorylated MLC2.

      Fig. S5E:

      Authors should consider presenting data of decreased histone methylation of cytoskeleton regulators in main Fig. 5, since this is an important conclusion of this part.

      As suggested we will shift the information currently presented in figure S5E to the main figure 5.

      Statistics should be revised throughout the manuscript. Comparisons of more than 2 groups should be performed with ANOVA or similar multiple comparison test (instead of t-test).

      We thank reviewer for this comment. We replaced statistical tests with the appropriate ANOVA in the respective graphs and updated the legends accordingly.

      **Minor comments**

      Statement of significance mentions "Anti-HER2-therapy resistance", but this is a misleading since the paper does not deal with therapy resistance. Or are the cell lines used in the study resistant to anti-HER2?

      We thank the reviewer for this suggestion. While resistance to anti-HER2 therapy remains one of the major clinical challenges in the treatment of HER2 positive BC lesions, we agree that our data do not strictly address this point. Thus, we have modified the sentence accordingly.

      In line with this, authors could add some lines of thought on how RNF40 could be targeted in the clinical/pre-clinical context, which could inform further translational studies.

      This is a great suggestion. In the revised manuscript we will include additional text to specifically address this point.

      Line 117 "Moreover, HER2-positive metastatic BC samples showed a particularly high expression of RNF40 compared to primary tumors".

      Perhaps rephrase, was it that the expression level (intensity) was higher or that the % of positive cells/tumours was higher in the brain mets?

      This is a critical point that we will consider in the revised manuscript. We have modified the sentence accordingly to read, “Moreover, the incidence of RNF40-high specimens was higher in HER2-positive brain metastases compared to primary tumors (Fig.1A-B)”.

      Fig.1D and S1C. while S1C shows TCGA data, it is unclear which set of patients is Fig.1D (since text says publicly available data, line 118-119), are these their own set of patients (used in Fig.1A-B)? This should be specified in the text, legend.

      The origin of the data shown in Fig.1D for relapse free survival of RNF40high and RNF40low patients (KM plotter) is mentioned in the figure legend (kmplot.com) and in the material and method section. However, since this was not apparent, to increase the readability, we have now added a statement about the publically available database of origin for every output graph in the main text as well in the legend and supplementary material.

      Line 122. Authors should be careful with this conclusion so far, a correlation between expression and cancer stage/survival does not necessarily mean a tumor suppressive/supportive role.

      We thank the review for this comment. We agree with this statement. Therefore, we have carefully rephrased this sentence as following, “In summary, these data demonstrate that RNF40 expression is maintained in HER2-driven primary metastatic BC and that its expression correlates with poor prognosis in these patients.”

      Fig. S4E: there are missing labels in graph (control, siRNF40).

      The labels have been added.

      Panels in some figures are discussed in text randomly and not following same order. For example, Fig.1 (after panel D, then panel H, then back to E, F, G), S3, 4A-C,

      We reorganized the order of the different panels of Fig1 to increase the readability. We further screened the main text for similar problems and modified the respective figures accordingly.

      Fig.1E: I would suggest changing the line colours, so Rnf40wt-wt line is red and the fl-fl is black, therefore it is similar to panel D (high Rnf40 red, low in black).

      We thank the reviewer for this suggestion. Accordingly, we have now indicated low RNF40 expression in red (Figure 1D, 1E and S1B) in the same way that we have indicated RNF40 expression throughout the rest of the study.

      Supp Videos: for reviewers and readers, it would help that video has a label while it plays, otherwise after downloading it, video name does not tell whether it is control or RNAi.

      As suggested we have renamed the video files of for each condition and added a label informing about the identity of the sample while the video plays.

      Reviewer #2 (Significance (Required)):

      Given that RNF40 function seems to be context-dependent, findings from this study could have broad significance for other cancers with high RNF40, or even in other pathological contexts -if any- that cursed with high RNF40.

      It also provides some mechanistic data (that should be improved as suggested in comments) linking this ubiquitin ligase to the cytoskeletal machinery and, therefore, control of migration and also proliferation and survival. This will also advance the field.

      Area of expertise

      Actin-myosin cytoskeleton, Rho GTPases-ROCK, cancer, metastasis, cell signalling

      Reviewer #3 (Evidence, reproducibility and clarity (Required)):

      **Summary:**

      Wegwitz and colleagues present extensive and detailed data focussed on the role of the E3 ubiquitin ligase and ring finger protein RNF40 in HER-2 associated breast cancer. It is clear that the role of RNF40 and its major substrate histone H2B (monoubiquitination of histone H2B at lysine 120; H2Bub1) as part of a complex with RNF20, is not a simple one in the context of malignancy. This group, and others, have previously reported on the intriguing role of RNF40 that can in certain circumstances function to suppress tumorigenesis, and in other circumstances function to support tumorigenesis. While H2Bub1 has been shown to be lost in many different malignancies, these investigators show that in HER-2 associated breast cancer, this is not the case. In fact, the results presented in this study show that RNF40-mediated H2Bub1 is important for the expression of genes involved in the actin cytoskeleton and the downstream FAK signalling cascade. Supporting this, mining of a public database showed that RNF40 mRNA was high in HER-2 associated breast cancers and was correlated with a worse prognosis (overall and RFS, relapse free survival). The investigators also used a mouse model (MMTV-Erbb2) generating a tri-transgenic (MMTV-Erbb2; MMTV-Cre; Rnf40flox) that allowed breast tissue specific overexpression of HER2 at the same time as KO of Rnf40, so mimicking the human disease. In fact, mouse tumours recapitulated the human results, including in disease free survival (lower with higher Rnf40), and with less tumours seen when there was less Rnf40 (the Rnf40 floxed tumours appeared heterogenous in staining patterns for Rnf40 and H2Bub1, supporting the concept of "escaper" cells, positive for both Rnf40 and H2Bub1 that would be positively selected during tumorigenesis).

      The authors also took a cell biology approach to studying HER2 positive breast cancer and RNF40 using two HER2 positive cell lines (HCC1954 and SKBR3). RNF40 was down-regulated using siRNA and numerous functional studies showed that targeting RNF40 suppressed behaviours consistent with tumorigenesis (proliferation, migration, clonogenic survival, spheroid formation, growth kinetics). Furthermore, down-regulation of RNF40 in the HCC1954 cell line followed by GSEA identified gene signatures associated with apopotosis and the actin cytoskeleton regulatory pathway (e.g. ROCK1, VAV3, LIMK2, PFN2). They further showed that phospo - cofilin (that occurs downstream of ROCK1) was reduced in RNF40 down-regulated cells, also implicated in regulation of the actin cytoskeleton. Phalloidin staining for F-actin showed disruption of the cytoskeleton in RNF40 down-regulated cells. Additionally, the ROCK1 inhibitor, RKI-1447, showed similar effects to depletion of RNF40.

      The authors then sought to determine whether the RNF40 associated gene expression in HER2 positive cells was in fact happening through H2Bub1 and the active histone mark H3K4me3 it has been reported to cross-talk with. RNF40 regulated genes (up or down-regulated) showed lower levels of H2Bub1 occupancy compared to non-regulated genes. H3K4me3 was lost in most genes influenced by RNF40 down-regulation, including genes associated with the actin regulatory pathway. The overall conclusion is that RNF40 is a major epigenetic regulator of the actin regulatory gene network in HER 2 positive breast cancer and could be a therapeutic target.

      **Major comments: major issues affecting the conclusions.**

      (1) What is happening at the gene level for both H2Bub1 and H3K4me3 in the context of RNF20 down-regulation is complex and would benefit from inclusion of a schematic, or a series of schematics describing different scenarios, as the text is quite difficult to follow.

      This is a very constructive proposition. We will attempt to follow this suggestion in order to simplify the message of the respective section by providing to schematic illustrations depicting the cascade of events occurring upon RNF40 loss in the cancer cells.

      It is not entirely clear that the changes seen in H3K4me3 are a direct result of cross-talk with H2Bub1 (some literature reports that there is no cross-talk between these histone marks for instance). It is also not entirely clear how the other histone marks investigated support the main discoveries of the paper. The authors need to consider this in the way that they present the data and their interpretation of it.

      The reviewer addresses an important point about the mechanistic aspect of the RNF40-dependent epigenetic regulation. We and others have shown that RNF40-mediated H2B monoubiquitination is a central step for activation of the COMPASS complex and the TSS-proximal broadening of H3K4me3 (PMID:31733991, 19410543, 22505722, 28209164). However, the situation certainly is not as straight forward as it is in yeast, where the vast majority of H3K4 trimethylation is H2Bub1-dependent. To what degree global H3K4me3 levels are dependent upon the H2B ubiquitin ligases RNF20 and RNF40 appears to vary, depending upon the investigated system (probably the variation in the literature referred to by the reviewer). However, in our work, we reproducibly see widespread H3K4me3 peak narrowing specifically on RNF40-dependent genes, in a context-dependent manner (i.e., genes displaying these effects are different according to the system investigated). To support and consolidate the central function of the H2Bub1-H4K4me3 crosstalk in our system, we propose to perform rescue experiments: siRNAs targeting the 3’UTR of RNF40 will be co-transfected with an expression construct encoding for either a wild type or a ΔRING (catalytic inactive) form of RNF40 lacking the endogenous 3’UTR. The ability of ectopically expressed wild-type, but not catalytic inactive RNF40, to rescue the expression of the identified actin cytoskeleton genes and downstream signaling should provide a solid argument to support the hypothesis of our study. We will also include additional discussion about the potential different H3K4 methyltransferases that may potentially be involved.

      (2) RNF40 is known to work in a complex with RNF20 to monoubiquitinate histone H2B at lysine 120 (H2Bub1). In experiments where RNF40 has been down-regulated, did the authors also note down-regulation of RNF20 (as has been previously reported).

      This is an interesting question from the reviewer. We indeed observed a consistent reduction of RNF20 protein levels upon RNF40 knockdown (and vice versa) in different cell systems, including the HER2-positive cell lines HCC1954 and SKBR3.

      Is the data presented likely to be the result of abrogation of the complex rather than RNF40 specifically?

      Although particularly difficult to answer, the use of a catalytic mutant in key experiments should at least partially shed light on this aspect (as proposed in the answer to Reviewer #3’s question 1). In that case, the complex integrity can be maintained while specifically abrogating RNF40 ubiquitin ligase activity.

      While I am not asking for experiments to be repeated with down-regulation of RNF20, some consideration of this needs to be included in the Discussion. Is RNF20 also highly expressed in HER2 positive breast cancer (TCGA, KM Plotter data).

      We absolutely agree with the reviewer’s point of view. As an obligate binding partner of RNF40, RNF20 indisputably plays an important function in the phenotype caused through RNF40 loss. We will therefore carefully further discuss this aspect in the revised manuscript. Preliminary analyses based on the TCGA dataset point at a high expression of RNF20 in HER2-positive lesions. Furthermore, survival analysis of HER2+ BC patients based on the same dataset showed that patients with high RNF20 expression harbor an unfavorable prognosis, similar to what we have seen with RNF40. We may therefore implement these expression and survival data in the revised manuscript.

      **Minor comments: important issues that can confidently be addressed.**

      (3) It would appear that immunohistochemistry for RNF40 and H2Bub1 on human samples is only reported as "low" or "high". This is perhaps not dealing with the full spectrum of IHC scores, such as completely absent, although the methods do note a "null" value (no detectable staining). Were there no "null" results? Please define the criteria for "low" or "high".

      Indeed, specimens lacking H2Bub1 or RNF40 staining were attributed the “null” scoring. However, while we have observed null staining in other BC subtypes (e.g., see Bedi, et al., 2015), none of the HER2 positive BC samples were found to be negative for either RNF40 or for H2Bub1. However, for the revision, we will provide representative examples of null-stained tumor specimens (from other BC subtypes) for RNF40 and H2Bub1 from the same tissue microarray.

      (4) I think there might be some confusion in labelling of Fig 1A and B as the legend states that all breast cancers are on the left and the HER-2 positive on the right, for each of primary tumours and brain mets, but I think one is under the other? Labelling should be checked in this figure.

      We apologize for this mistake. This has been corrected in the figure legend accordingly.

      (5) What this IHC data doesn't show is whether RNF40 and H2Bub1 levels are always correlated in individual tumours (i.e. RNF40:H2Bub1, high:high OR low:low OR null:null). Can the authors please include and comment on this data.

      The reviewer has made a very interesting point here. We will comment on this point in the revision.

      (6) Please include overall survival data (KM Plotter) as a panel in figure 1, alongside RFS for RNF40 expression levels (currently in Supplementary).

      We added the OS as well the RFS data from the same database next to each other in the main figure.

      (7) Spheroid formation looks to only be shown in a single cell line (HCC1954). Was the other cell line not suitable for spheroid studies? Some comment should be made and care taken not to "overclaim" as text notes two cell lines.

      SKBR3 are unfortunately not suitable for tumor sphere formation assay. We may provide instead a soft agar assay with SKBR3 cells upon. If needed, we may replace the SKBR3 cell line with BT474 for this specific experiment

      (8) It would have been interesting to see results of a GSEA in the mouse mammary tumours as a complement to human. Is there a reason why this wasn't undertaken?

      Rnf40fl/fl tumors present a large fraction of “escaper” cancer cells retaining RNF40 expression. For this reason, bulk sequencing of such tumors would likely only provide a “diluted” molecular signature consequent to RNF40 loss. For this reason this experiment has not been done.

      (9) Conclusions are made about RNF40 in HER2 positive cells only in the context of H2Bub1 and H3K4me3. Without having conducted similar experiments in HER2 negative breast cancer cell lines / models, it is difficult to draw the conclusion that this is HER2 positive specific. Can the authors either soften some of their conclusions along this line, or consider repeating some of their data in HER2 negative models.

      The scope of the study has deliberately been set on HER2-positive malignancies, because former studies already extensively studied the impact of H2Bub1 loss in TNBC and Luminal BC (PMID 28157208, 18832071). We will therefore modify the manuscript text accordingly and soften the appropriate sections as suggested by the reviewer.

      (10) RNF40 likely has substrates other than histone H2B. There is a report describing interactions with RNF40 (STARING) and syntaxin for e.g., (Chin et al., 2002 J Biol Chem 277:35071-9). Can the authors please comment on other potential substrates of RNF40 in light of their data that focuses only on its epigenetic role as a regulator of the actin cytoskeleton.

      Our study was mainly focused only on the gene expression program driven by RNF40 in HER2+ BC. We therefore do not know nor have we focused on other novel non-histone substrates. We will, however, allude to this possibility in a revised manuscript.

      Reviewer #3 (Significance (Required)):

      Nature and Significance of the Advance:

      Clinically, this work provides a significant advance in that it is zeroing in on HER2 positive breast cancer and generating fundamental data that could underpin development of a new therapy for this malignancy. Conceptually, it is expanding knowledge of how the E3 ubiquitin ligase RNF40 is functioning as an epigenetic modifier of a specific type of malignancy by being important for the actin cytoskeleton.

      Work in Context of Existing Literature:

      As acknowledged by the authors, this work builds on a previous publication of theirs (Xie et. al., 2017 "RNF40 regulates gene expression in an epigenetic context-dependent manner." Genome Biol). They have other recent papers on RNF40 (Schneider et al., 2019 "The E3 ubiquitin ligase RNF40 suppresses apoptosis in colorectal cancer cells", Clin Epigenetics; Kosinsky et al., 2019 "Loss of RNF40 decreases NF-kappaB activity in colorectal cancer cells and reduces colitis burden in mice", J Crohns Colitis). H2Bub1 is one of the least well studied histone modifications and as such, this study of one of its key histone writers, RNF40, is significant in elucidating the significance of this histone mark.

      Audience:

      This paper will suit a discovery-based science audience interested in epigenomic regulation of malignancy. Further, it will suit those looking for new drug development strategies for malignancy.

      My Field of Expertise:

      Basic scientist with expertise in epigenetic/epigenomic regulation in malignancy; cell and molecular biology. I felt capable of reviewing all aspects of this paper.

  12. Mar 2020
    1. Note: This rebuttal was posted by the corresponding author to Review Commons. Content has not been altered except for formatting.

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      Reply to the reviewers

      Rebuttal_ Preprint RC-2020-00156

      We thank the editor for handling our manuscript and both reviewers for their constructive critiques. We provide below a detailed list of results already available and experiments we propose to perform to address the reviewers’ comments and improve the quality of our manuscript.

      Reviewer #1

      In this manuscript, Obacz et al. investigated the role of IRE1 signaling in regulating the recruitment of myeloid cells in glioblastoma multiforme (GBM) microenvironment. They show that inhibition of IRE1 signaling decreased polynuclear neutrophil (PN) infiltration to GBM tumors in an animal model; conversely, IRE1 activation correlated with higher expression of myeloid cells-attracting chemokines in GBM. They also show that IRE1-XBP1s pathway promotes proinflammatory chemokines in GBM tumor cells through upregulation of UBE2D3, which leads to degradation of the NFκB inhibitor IκB and activation of NFκB downstream signaling. Their finding of a novel IRE1/XBP1s/UBE2D3/NFκB axis is important for understanding the basis of pro-tumoral inflammation in GBM, potentially in other 'immune hot' cancers. The manuscript is well written and the conclusion is well supported by the experiments. However, there are a few critical points that need to be addressed to strengthen their study**.

      We thank this reviewer for his/her positive comments on our work and for the suggestions made to improve its relevance

      Review#1 point 1: In this study, the authors used the GBM primary cell line RADH87 with stable overexpression of wild-type (WT) IRE1 or a truncated IRE1 variant. The expression of wild-type IRE1 was confirmed by Western analysis (Figure S1D). However, the expression of truncated IRE1 variant was not shown.

      Response 1.1. The expression on truncated IRE1 variant (designated as Q780* - 80 KDa) is shown in Fig.S1D, following the expression on wild-type (WT) IRE1 (110 KDa). This point will be indicated in the revised version of the supplemental figure legends.

      In addition, without tunicamycin treatment, there was no visible difference in XBP1s expression between the cells expressing WT or the mutant IRE1.

      Response 1.2. Under basal condition, XBP1 splicing is indeed limited and therefore, there is no detectable difference in XBP1s expression level between IRE1 WT and Q780*. In contrast, under tunicamycin treatment (acute stress), reduced XBP1 mRNA splicing is observed (Fig.S1D) thus confirming the functionality of the Q780* truncated form. Of note, RNAseq was performed on these cell lines and basal splicing was quantified showing that even though it is an event that occurs at low frequency, it is decreased in cells expressing the Q780* mutant (this information will be added in the revised manuscript, data are available and analyses ongoing).

      In the Boyden chamber assay (Figure 1C, D), conditioned medium from these cells were used; it was not described whether the cells were treated (e.g. with tunicamycin) to activate the IRE1 pathway. ** Response 1.3. Cells were not treated with Tunicamycin to excluded the impact of other UPR arms in the induction of cytokines expression/myeloid cells attraction. As a consequence, it is the basal secretome (found in conditioned media) that was used in those experiments to evaluate cell migration. We have now strong evidences that blunting IRE1 signaling (either genetically or pharmacologically) has a strong impact on GBM cells proteome and in particular on their secretome even if under basal conditions (manuscript in preparation). This information together with the fact that basal XBP1 mRNA splicing is reduced in IRE1 signaling deficient (Q780* expressing) cells, indicate that in GBM cells, constitutive IRE1 activity contributes to modulate the composition of their secretome towards chemoattraction of myeloid cells. This point will be further detailed in the results and discussion sections of the revised manuscript.

      Review#1 point 2: The evidence that the mRNA expression of UBE2D3 positively correlates with IRE1/XBP1s pathway is weak. First, In Figure 3D, the correlation between the mRNA expression of UBE2D3 and XBP1 does not seem strong. In addition, as XBP1 mRNA level does not reflect IRE1 activation (as opposed to that of XBP1s), the level of XBP1s instead of total XBP1 should be assessed. Furthermore, such correlation should be validated in additional GBM cohorts/datasets.

      Response 2. We agree that the correlation between UBE2D3 and XBP1 mRNA levels in TCGA GBM cohort might not be strong. However data presented in Fig3D were significant. Values indicated in green were Pearson’s correlation values (r). This point will be included in the revised figure legends. Moreover, in the revised version of the manuscript we propose to directly correlate the levels of XBP1s mRNA with the expression levels of SYVN1, UBE2D3 and UBE2J1 mRNAs. These data are available from the RNAseq data obtained from the TCGA cohort and already used previously by us (Lhomond et al. Embo Mol Med 2018). In addition, following this observation we have carried out a number of experimental validations using both established and primary GBM cell lines with genetic modifications of XBP1/XBP1s expression as well as ER stress-dependent induction of XBP1s and we clearly demonstrated that XBP1s mRNA levels correlate with UBE2D3 mRNA expression levels (Fig.3G-H, Fig.S2D-E). In addition, in Fig3E using our IRE1 activity signature we have shown a strong correlation between UBE2D3 and XBP1s, which is even more robust than simply correlating the mRNA levels. Data are already available and analyses are ongoing.

      Review#1 point 3: The results in Figure 3 indicated that XBP1s acts as a transcriptional regulator of UBE2D3 expression. However, it is not clear whether this effect in GBM cells is direct or indirect. Further experiments such as chromatin immunoprecipitation and reporter assays are required to clarify this point.

      Response 3. We agree with this reviewer’s point. Although we have scrutinized the publicly available ChIPseq databases and found UB2D3 among potential XBP1-regulated genes, we did not validate this observation in our model. To address this point we propose to perform ChIP experiments in cells overexpressing a tagged form of XBP1s and validate the presence of UBE2D3 promoter fragments in our experimental system. Moreover, these experiments will also be carried out with endogenous XBP1s (in-house XBP1s antibodies Pluquet et al. Cancer Res. 2013) in our primary GBM lines under basal and ER stress conditions. At last, to further document this, luciferase reporter assays using the UBE2D3 promoter (whose length would be defined based on ChIP experiments and the presence of XBP1s binding sites) upstream the luciferase ORF could be performed. Both ChIP and reporter assays have to be performed.

      Review#1 point 4: In addition to UBE2D3, the two other ubiquitin-protein ligases, SYVN1 and UBE2J1, may also be implicated in the degradation of IκB. Did the authors assess their potential role on IκB degradation in their model system?

      Response 4. We thank this reviewer for this suggestion. We have previously tested the impact of SYVN1 on IkB degradation with results showing a lot of variation. Indeed even though the trend of our results indicated that SYVN1 silencing appeared to lead to a slight increase in IkB expression, they never reached statistical significance. Variability in the results might be due to the efficacy of SYVN1 silencing and as such we propose to repeat further these experiments with SYVN1 siRNA smart pools to improve silencing efficacy. Moreover, SYVN1 has been shown to also contribute to the ubiquitylation and degradation of IRE1 (Gao et al. Embo Rep 2008; Sun et al. Nat Cell Biol 2015) and has its expression regulated by IRE1 activity (Dibdiakova et al. Neurol Res 2019), it might represent as well a very interesting target to study. Regarding UBE2J1, the situation is less documented. However, it was shown that this E2 works together with SYVN1 in conserved manner to contribute to ERAD (Chen et al. Nat Plants 2016). As such it might also be interesting to test whether the silencing of UBE2J1 impacts on IkB expression. To sum up, we propose to test experimentally whether the silencing of UBE2J1 or SYVN1 or both together impacts on IkB expression (we need to perform the experiments).

      Review#1 point 5: The authors only used ectopic expression of relevant proteins to test their hypothesis in U87 and RADH87 cells. It is necessary to validate these findings using siRNAs/inhibitors for IRE1 and UBE2D3 in a GBM cell line that expresses high levels of endogenous IRE1 and UBE2D3.

      Response 5. We propose to test the effect of SYVN1 and UBE2J1 silencing on IkB expression in U87 and RADH87 cells in the revised version of the manuscript (see above). In addition to address this reviewer’s comment, we propose to use U87 and RADH87 cells overexpressing IRE1 (Lhomond et al. 2018) and treat them with MKC886, or with siUBE2D3 or with both and evaluate whether in those conditions the NFkB pathway is affected. These experiments should be carried out relatively easily provided that all the recombinant cell lines, drugs and siRNA are already available.

      Review#1 point 6: In Figure 3I: The protein expression of UBE2D3 should be shown.

      Response 6: We had included control experiments with UBE2B3 expression in FigS3B in the initial version of the manuscript. We will include UBE2D3 expression for Fig3I in the revised version of the manuscript (these data are already available).

      Review#1 point 7: In the right panel of Figure 3I: What do the labels #1, 2, 5 mean? Clear descriptions should be provided in the figure legend.

      Response 7. Those labels correspond to different RADH87 cell lines stably overexpressing UBE2D3 protein. The validation of UBE2D3 expression using Western blotting will be included in FigS3B of the revised version of the manuscript (data are already available).

      Review#1 point 8: In Figure S1D: The expression levels of the truncated IRE1 variant should be shown.

      Response 8. The expression on truncated IRE1 variant (designated as Q780*) is shown in Fig.S1D, following the expression on wild-type (WT) IRE1. This point will be indicated in the revised version of the supplemental figure legends.

      ======================================================================

      Reviewer #2

      In the current study, the authors generate evidence supporting a novel pathway downstream of IRE1α/XBP1s in GBM cells involving the activation of an E2-ubiquitin ligase, UBE2D3. In order to do this, they use a combination of patient derived and established cell lines engineered to overexpress IRE1 mutants, XBP1s or UBE2D3. They claim that UBE2D3 is upregulated downstream of XBP1s in GBM cells, and functions to activate NF-kB through the degradation of IkB, thus promoting CXCL2/IL-6/IL-8 production and the subsequent recruitment of monocytes and polymorphonuclear (PN) cells to the tumor microenvironment. However, the article has major shortcomings that need to be addressed before considering its publication

      We thank this reviewer for his/her constructive comments on our work.

      Review#2 point 1: Fig. 1: Classification of immune cells infiltrating GBM. The characterization of immune infiltrate in GBM is too simplistic. Monocytes, monocyte-derived macrophages and microglia are treated as equivalents along the text (IBA1+), making the story hard to follow. At least in mice, these populations can be easily distinguished based on CD45/CD11b/Ly6C expression (see for example Zhihong Chen et al., Cancer Research, 2017). Can the authors further analyze which of those population are actually affected under IRE1 deficiency and/or UBE2D3 overexpression? On the other hand, it is rather questionable that all CD11b negative cells are exclusively T cells, as suggested in Fig 1B. Can the authors provide evidence and/or references to support their gating strategies?

      Response 1: We thank the reviewer for this comment. Our objective was to test the impact of IRE1 modulation on the infiltration of myeloid cells in the tumor, and we did not plan to describe this effect on the complete and detailed infiltrating myeloid populations in GBM which could represent a full study on its own. However, to address this reviewer’s critique we propose to complete the characterization of the myeloid population in our mouse model using IHC by adding Ly6C staining for macrophages and granulocytes. We did not select flow cytometry approach to explore this point as suggested by the reviewer (Cheng, Cancer Res, 2017), but instead IHC was preferred as we thought that the localization of the infiltrated immune cells was important to evaluate (periphery vs. core of the tumor). The information about the localization of immune cells is already available and will be added to the revised manuscript. Concerning the second point raised by this reviewer, the strategy to characterize the immune population in human GBM specimen was to combine CD45 and CD11b markers as previously described by Hussain et al. Neuro-Oncol 2006 and Parney et al. J Neurosurg 2009. Moreover, the analysis of additional markers allowed us to confirm that CD45+ CD11b+ cells were mainly monocytic cells (that also co-expressed CD14, CD168, CD64 and HLA-DR); CD45 low CD11b high cells were granulocytes (CD66B, CD15 and CD16); and CD45 high CD11b low cells were mainly CD3+ T cells. These data are already available and will be added to the revised manuscript.

      Review#2 point 2: Fig. 1: RADH IRE1 Q780\ model - Can the authors further validate the IRE1 deficiency of their model cell line RADH87 IRE1Q780*? It appears to have severely reduced IRE1 levels when compared to the RAD87-IRE1WT cell line (figS1D). Furthermore, the WT and not the truncated form seems to be predominantly expressed. Intriguingly, XBP1 is still being spliced after tunicamycin treatment in this mutant line. All these results differ significantly from the U87-Q780* cell line originally published by Lhomond et al., 2018. Can the authors comment on these differences? Was there a mixture in cell lines? *

      Response 2: We agree with the reviewer that the level of IRE1Q780* expression on RADH87 cells is lower than the IRE1WT expression (Fig.S1D). As observed by this reviewer, XBP1 was still spliced in Q780* cells but XBP1s expression was reduced as shown in Figure S1D. This is mostly due to the ratio between the expression endogenous IRE1 and that of Q780*, which as previously shown (Lhomond et al; 2018) acts as a dominant negative and preempts endogenous IRE1 signaling. The differences observed are also probably due to the methods used, indeed we measured XBP1 and XBP1s mRNA expression in U87 cells (Lhomond et al. 2018), whereas XBP1s protein expression was used with RADH87 cells (introducing the RNA translation parameter that was not monitored in U87 cells). Differences could be also linked to the cell lines as we used the U87 immortalized and RADH87 primary cell lines.

      Review#2 point 3: Fig. 1: Impact of IRE1 inhibition on recruitment of myeloid cells to the TME. The experiment in figure 1E-F, which is the only in vivo evidence supporting a role of IRE1 signaling on myeloid cell recruitment, is very hard to interpret. The authors show no evidence that IRE1 is being inhibited under the treatment and if so, up to which extent. Furthermore, what are the cells targeted by MKC in this setting? The differences in the infiltration of PN cells seem very slight, nothing is mentioned regarding the number of mice per group, or the statistical analysis performed. I would suggest performing a simpler experiment to demonstrate an intrinsic effect of IRE1 signaling in GBM cells, comparing the recruitment of myeloid cells in tumors generated by GL261 cells expressing WT vs deficient forms of IRE1.

      Response 3: The mouse model used in the paper is fully described in (Le Reste BioRxiv 2020 - doi: https://doi.org/10.1101/841296) and all the details about the procedures can be found in this manuscript. This model was developed to recapitulate in mice the standard of care for GBM patient including surgical resection. In addition, drug delivery in brain tumors is often an issue due to the blood-brain barrier. Therefore, the IRE1 inhibitor was delivered locally after resection of the tumor, exposing both tumor and stromal cells. To quantify the myeloid cell recruitment in Fig1E-F, at least thirty random fields from tumor tissue and at least thirty random fields from tumor periphery were quantified for control (PLUG) and MKC-treated group (2 mice/group). The number of positive cells in tumor tissue and tumor periphery were then pulled together for statistical analyses. The significance of the differences in myeloid cells recruitment between control (PLUG) and MKC-treated group was estimated using unpaired student t-test. At least 8 tumors of each group were analyzed comprising 2 to 3 sections of each and 10 fields per section. In addition, we have also performed the experiments using GL261 cells knockout for IRE1, the data are already available and could be possibly added to the revised manuscript.

      Review#2 point 4: Fig. 2: Correlation between IRE1 signature and cytokine/chemokine signature. In the IRE1 signature as determined in the EMBO Mol Med paper (and to which the authors continuously refer) 6 out of 38 (15%) of the genes correspond to cytokines and/or chemokines (Il6, Il1b, Cxcl2, Cxcl5 and Ccl20) (Lhomond et al., 2018). Besides the fact that it is very unclear how this signature was obtained in the first place, it is rather surprising that in the current paper the authors correlate this "IRE1 activity" signature with the same or other cytokines/chemokines mRNA levels and come to the conclusion that there is a high correlation (fig 2A). Isn't this to be expected? Can the authors clearly explain how the IRE1 signature was determined and prove that their "IRE1 signature" is, in fact, representing IRE1 activity? For instance, it is important to cross validate their results by using an independent signature of IRE1 activity (e.g. ChipSeq XBP1s targets, Chen et al., 2014)?

      Response 4: We thank this reviewer for asking for precisions about the procedure. The IRE1 signature was fully described in Lhomond et al. 2018 and was obtained from transcriptome datasets obtained from U87 modified for IRE1 activity (Pluquet et al., 2013). IRE1 was validated on GBM patients and appeared as an important tool to evaluate IRE1 activity in tumor specimen not only in GBM but also in other tumor types (Rubio-Patiño C, Cell Metab 2018). Furthermore, IRE1 activity was also directly linked to the pro-inflammatory tumor cell secretome in various studies such as Logue et al. 2018. As indicated by this, some cytokines/chemokines studied in this work were indeed part of the IRE1 signature and correlation between this signature and their expression was indeed expected. However the other main cytokines/chemokines studied here were not present in the IRE1 signature indicating that IRE1 could have been involved in the regulation of their expression. As proposed by reviewer#2, we will include in the revised version of the manuscript the analysis of cytokines/chemokines from the dataset ChipSeq XBP1s targets (Chen et al. 2014), although this study was performed on breast tumors.

      Review#2 point 5: Fig 2: XBP1s controlling cytokines/chemokines expression in GBM cells - As suggested by the data on fig1C-D and fig2E, IRE1 appears to be constitutively active in GBM cells, as IRE1 deficiency is sufficient to cause a defect in chemokine production. However, as shown in fig S1D, XBP1s protein was not detected under basal conditions, suggesting that the deficiency in chemokine production in IRE1-deficient cell lines is XBP1s-independent. Can the authors further discuss these results?

      Response 5: We thank the reviewer for commenting this point. We think that indeed IRE1 is constitutively active in GBM cells. As we have tested XBP1s protein expression in untreated and tunicamycin-treated RAD87 cells (FigS1D), and we will also provide real time qPCR data to show the presence of basal XBP1s mRNA levels (data already available). We agree that the way we presented the results are misleading and undermine the basal expression of XBP1s. This will be fixed in the revised manuscript.

      Review#2 point 6: Fig 3: IRE1/XBP1s/UBE2D3/NF-kB axis - Authors must show the activation status of NF-kB in parental U87 cells (Fig3A), as this is a critical evidence to support that IRE1a-deficient U87-DN cells are defective in chemokine production due to an impairment in NF-kB signaling. In addition, even when tunicamycin treatment induce XBP1s and UBE2D3 (figS2D) it does not induce IkB degradation nor NF-kB phosphorylation in parental U87 and RADH87 cells (figS3C) as one should expect if IRE1/XBP1s/UBE2D3/NF-kB pathway is operating in these cells. How can this be explained? Only after XBP1s or UBE2D3 overexpression, NF-kB signaling appears to be affected.

      Response 6: As shown in Fig3A, U87 cells deficient for IRE1 signaling (DN) exhibit decreased NFkB signaling as exemplified by decreased phospho-NFkB and phospho-IkB compared to control U87 cells proficient for IRE1 signaling. In our manuscript, we mainly focused on the activation of the IRE1/XBP1s/UBE2D3/NFkB signaling axis under basal condition. One could speculate that tunicamycin treatment leads to a strong stress response that others mechanisms are activated that overwhelm the IRE1/XBP1s/UBE2D3 pathway we are describing herein. For instance, it has been demonstrated that the IRE1/JNK signaling was linked to NFkB activation upon acute ER stress (Tam et al. PLoS One. 2012;7(10):e45078; Schmitz et al. Biomedicines. 2018 Jun; 6(2): 58.) and furthermore PERK activation upon thapsigargin or tunicamycin treatment was also found to promote NFkB activation (Deng et al. DOI: 10.1128/MCB.24.23.10161-10168.2004; Fan et al. Cell Death Discov. 2018 Feb 12;4:15). We believe that the pathway we describe here might be linked to constitutive activation of IRE1 signaling (proper to tumor cells) rather than acute activation of this pathway and be compatible with sustained proliferation. To further document this point, we have already generated data about the phosphorylation status of NFKB in GL261 cells KO for IRE1 compared to the parental cells (data will be provided in the revised version of the manuscript). In addition we are currently investigating the correlation between IRE1 activity signature and that of NFkB as defined previously (Jin et al. Cancer Res. 2014 May 15; 74(10): 2763–2772.), results should be available shortly and will be added in the revised manuscript.

      Review#2 point 7: Fig 4: UBE2D3 and MIB1 – The authors should discuss better what is the possible interaction between UBE2D3 and MIB1. As shown in fig4G, silencing of MIB1 cause a severe increase in UBE2D3 protein levels but this is not commented in the text.

      Response 7: We thank the reviewer for this comment. We believe that MIB1 might also controls the expression of UBE2D3. The data are already available and will be included in the revised version of the manuscript.

      Review#2 point 8: Fig 6: Chemokines driving recruitment of myeloid cells to UBE2D3 overexpressing tumors. A formal demonstration that GL261-UBE2D3 tumors recruit higher numbers of MM and PNs through an enhanced production of CXCL2, IL-6 and/or IL-8 is lacking. For instance, they could compare the infiltration of myeloid cells in GL261-UBE2D3 vs GL261-UBE2D3-CXCL2KO tumors.

      Response 8: To address this point, we propose to test the expression of these cytokines/chemokines in the GL261 tumors after resection using ELISA. These experiments could be carried out in IRE1 KO tumors, in UBE2D3 overexpressing tumors and performed for instance using perfusion of CXCL2, IL6 or IL8 neutralizing antibodies or cells KO for these chemokines. These experiments could be performed but might lead to inconclusive results (not statistically significant) if there is redundancy between the roles of those chemokines. As such, we think that we could provide in vitro information about the respective roles of these chemokines in recruiting MM and PNs but that at present stage the in vivo demonstration is to premature.

      Review#2 point 9: Authors must provide replicates of the blots to sustain their claims: FigS1D, Fig3A, Fig3I, Fig4G.

      Response 9: Replicates and quantifications are already available and will be provided in the revised version of the manuscript.

      Review#2 point 10: The authors should include a better description of the methods regarding bioinformatic analysis. For instance, which genes where used for MM/PN/T cell signatures in fig1A/S1A?.

      Response 10: We thank the reviewer#2. This information is available and a complete description will be included in the revised version of the manuscript.

      Review#2 point 11: Missing statistical significance on fig 2C and fig 6A to support their claims.

      Response 11: Statistical values will be included in the revised manuscript.

      Review#2 point 12: Fig2F is presented in the text as mRNA levels but in the figure as protein levels.

      Response 12: This point will be fixed in the revised version of the manuscript.

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      Reply to the reviewers

      Reviewer #1 (Evidence, reproducibility and clarity (Required)):

      In this report, van Schaik et al., modified an established CUT and RUN method and combined it with previously used DamID to identify Lamin Associated Domains (LADs) with better temporal resolution. Previous DamID experiments labeled locations where lamin proteins were present within a 5-25 hour window while the new technique, pA-DamID, labels DNA within a 30 minute window providing better temporal resolution. The authors used this technique to identify LADs at multiple stages of the cell cycle and applied this protocol to different cell types. The authors FIND differences when comparing data sets between cell cycle time points and cell lines.

      We thank the reviewer for the helpful comments.

      **Major points:**

      1) The data sets generated and displayed in this manuscript seem incomplete. In Figure 1G, the authors compare lamin B2 vs. lamin B1 generated LADs in HAP-1 cells and lamin A/C vs lamin B2 LADs in hTERT-RPE cells. In figure S4, panel C compares lamin B1 and lamin B2 in K562 cells and lamin B2 and lamin A/C in hTERT-RPE cells. It would have been informative to have a complete dataset for lamin B1, lamin B2, and lamin A/C identified LADs in all cell lines analyzed. The information provided from these datasets would be useful to the scientific community.

      We did not think it was necessary to generate every lamin pA-DamID data set in every cell line, given that previous DamID studies indicated that lamins A, B1 and B2 give the same genome-wide pattern {Meuleman, 2013, 23124521; Kind, 2014; 24717229}. However, we agree with the reviewer that the missing data sets lead to a sense of incompleteness and might distract the reader from the main message of the manuscript. We suggest to generate Lamin B1 pA-DamID in hTERT-RPE cells– provided that the current Corona virus shutdown will not prevent us from doing this experiment. Doing so, we 1) have a complete lamin data set in hTERT-RPE cells, which we study in most detail in this manuscript 2) can compare all lamins within the same cell type 3) can compare all Lamin B1 DamID data to the corresponding Lamin B1 pA-DamID data.

      2) The authors discovered that LADs reposition during progression through the cell cycle. It would have been interesting to know whether these changes have transcriptional consequences? One could perform RNA-SEQ experiments to discover if LAD occupancy results in transcriptional changes and choose a few genes to confirm the findings with RT-PCR. Is this the same for lamin B1, lamin B2, and lamin A/C occupied LADs? Analyze if there are any genomic features such as CTCF or transcription factor binding sites that correlate with the loss of LADs.

      In the first part of this point, the reviewer suggests to look at transcriptional consequences of changes in NL interactions. To address this point, we require some measure of nascenttranscription during the cell cycle, which is not available in any of the studied cell lines. A potential experiment would be to map polymerase occupancy with pA-DamID / CUT&RUN or run-on transcription with any other method at the synchronized time points. However, this experiment is not trivial and we feel that this goes beyond the scope of this manuscript, which focuses on the development of pA-DamID and the m6A-Tracer with a proof-of-principle example of NL binding dynamics during the cell cycle.

      In the second part of this point, the reviewer asks whether changes in NL binding correlate with genomic features such as CTCF binding sites or transcription factor binding sites. In the manuscript, we already include correlations with various active features (active gene density / replication timing) (Fig. 3E-G, 4C-E), that generally correlate well with transcription factor binding. We have added CTCF peaks as comparison (Fig. S7F).

      3) The authors state that using H3K27me3/H3K9me3 in pa-DamID showed no enrichment. This is surprising considering that both modifications are enriched in heterochromatin and at the nuclear periphery. It appears that the peripheral enrichment is masked by the larger overall internal pool. The authors should discuss this observation and comment on the sensitivity of the method to detect local enrichment versus the global levels of a protein or modification in pa-DamID.

      We believe that H3K27me3 and H3K9me3 histone modifications show the expected pattern in their distribution in the nucleus. However, due to the peripheral mask slightly extending beyond the cell boundaries, the calculated peripheral enrichment is underestimated. This has been better described in the figure legend.There is a small enrichment at the nuclear periphery compared to diffuse Dam and untargeted pA-Dam (Fig. 1B/1C/1F). To further support the pA-DamID data quality of these histone modifications, we have added a comparison with ENCODE ChIP-seq data tracks in K562 cells (Fig. S3C).

      **Minor points:**

      Figure 1: Change colors for Figure 1F and Figure 2D. The colors are hard to discern.

      Figure 2B: Please mark which antibody was used for this analysis.

      Figure 2C: Please also overlay data from pA-DamID lamin A/C experiments.

      Figure 4: Please mention which antibody was used for the pA-DamID experiments used to generate this dataset.

      Figure 5: Please mention which antibody was used for the pA-DamID experiments used to generate this dataset.

      Figure S5 C and D: Please mention which antibody was used for the pA-DamID experiments.

      We have made edits to address the minor comments above. However, we do not have Lamin A/C data in HAP-1 and K562 cells to add to Fig. 2C.

      Reviewer #1 (Significance (Required)):

      The major contribution of this manuscript is the description of an improved method to map LADs. This is a valuable contribution. By using this new method, the findings of this paper provide some new insight in LAD dynamics throughout the cell cycle although the experiments are largely phenomenological. This is a technically sound study.

      Reviewer #2 (Evidence, reproducibility and clarity (Required)):

      The paper describes a new method for detecting Lamin associated DNA domains, which allows better time resolution than classical DamId. It is a good idea and its functionality is demonstrated in tissue culture cells. There are minor insights but it is important that we advance the field with new and better technologies, thus this version amply suffices to give evidence of that.

      We thank the reviewer for the positive feedback.

      Reviewer #2 (Significance (Required)):

      The audience is all persons working on chromatin organization in the nucleus, which is a large audience. The data are clear as they basically are proof of principle for a new technique. There is nothing major to request as revision. They might cite papers on damID in worms and tissue specific applications of this in living organisms, as this is likely to be the situation that is most interesting in the long run. The resolution (in bp) would be interesting to know and validate.

      We have extended the discussion on new applications of pA-DamID.

      We now compare data quality and resolution between DamID and pA-DamID, focusing on the mapping of NL interactions (Fig. S4D-E).These plots indicate similar data quality and resolution between the two methods.

      I have no other major revisions to request.

      Reviewer #3(Evidence, reproducibility and clarity (Required)):

      In the manuscript by Schaik et al (Van Steensel laboratory) the authors describe a very clever approach to identifying Lamina Associated Domains (LADs) using the principles of the 'cut-n-run' strategy. Specifically, they engineer the Dam methyltransferase used in canonical DamID in frame with a protein A moiety capable of interacting with an antibody (in this case lamins--B1, B2 and A/C). After permeabilization, cells are incubated with antibodies, then pA-Dam purified protein--for a brief time window--is added to mark associated DNA with GmATC. This technique is a valuable contribution to the field, particularly since, as the authors point out,an advantage of pA-DamID is that the labeled DNA can also be visualized in situ using the m6A-Tracer, before this DNA is sequenced. This allows for validation of findings and is highly amenable to cell sorting technologies. In addition, this technology allows for a time-resolved measure of LADs not currently available by standard DamID. The authors apply this technology to four different cell types. They noted that the 'maps' generated by the is technology differed from canonical DamID at very specific regions (small LADs in very localized regions) . They then embark on a series of experiments to show that these differences arise from cell cycle -related differences that are differentially picked up by the methods--with the pA-DamID allowing for dissection of more discrete cell cycle stages/configurations. In general they find an initial preference for sub-telomeric LADs to associate with the nuclear lamina fist, then more centromeric. There is some data suggesting loss/gain of LADs in specific regions/with specific features. The manuscript is well written and the data well presented. However, there are some points that need to be addressed . Overall, there is some oversimplification or omission of previous data in the field, a lack of clarity in how some of the data was interpreted, and some areas where clarification and/or additional analyses would be helpful. I sincerely hope the authors find the following critiques to be useful. Thank you for the opportunity to review your very nice work.

      We thank the reviewer for the constructive and very detailed comments, these have been extremely helpful in improving the manuscript.

      **Introduction:**

      **Microscopy studies found that telomeres are enriched near the NL in early G1 phase, leading to the hypothesis that telomeres may assist in NL reassembly onto chromatin [13].**

      ● There have been numerous studies identifying the timing and disposition of INM proteins and Lamins at m the end of mitosis (during NE reformation). Why are you citing just this one? (e.g. ​Thomas Dechat et al., 2004; T. Dechat et al., 2000; Ellenberg et al., 1997; Haraguchi et al., 2001​)

      We have expanded the introduction to better cover previous work on the reforming NL (paragraph 2) and initial genomic interactions with the NL (paragraph 3).

      **Furthermore, during S-phase B-type lamins have been found to transiently overlap with replication foci in the nuclear interior, at least in some cell types [20]**

      ● While, technically, this has indeed been reported, this study is from 1994 and has not been repeated. The cells used in this study (3T3 fibroblasts) are widely used and others have not noted this phenomenon. Soften this.

      **Other studies have indicated that lamins are important for DNA replication [reviewed in 21].**

      ● Likewise, direct roles for lamins in replication are controversial (acknowledged in the small section of the cited review on the role of lamins in replication).

      ● Perhaps combine the two sentences above to soften the implication that this is a "known" role of B-type lamins. e.g. "A handful of studies have implicated a role for B-type lamins in replication, but the direct role of the lamina in this process remains unclear. Nonetheless, ......"

      This is a very good suggestion by the reviewer. We agree that literature has been controversial and should be approached with care. We have followed the advice and changed this.

      **Results:**

      **So far, the cell cycle dynamics of genome - NL interactions have primarily been studied by microscopy. While these studies have been highly informative, they were often limited to a ​few selected loci.​**

      ● Please cite your own study (Kind et al.) and other recent papers (Luperchio et al.-​https://www.biorxiv.org/content/10.1101/481598v1​;; Zhang et al., Nature-​https://www.nature.com/articles/s41586-019-1778-y​;) in which they were either 1) not limited to a few selected loci and/or 2) not microscopy-directed studies? There is an argument to be made here for the resolution (time and b.p.) you have achieved through your studies that these studies did not.

      To our knowledge, there have been no high-throughput microscopy studies of many individual loci performed studying this. Microscopy has been performed of collective sequences (i.e. all LADs (Kind, 2013 and indeed Luperchio, 2018)), which provide additional insights but lack sequence information in the images. We have expanded the introduction to better acknowledge these microscopy studies that are not limited to single loci.We feel that observations on LAD domain clustering (Luperchio) and B compartment formation (Zhang) are better suited for the Discussion, given that these observations are not directly related to genome – NL contact dynamics. We already discussed B compartment formation in the discussion, but now also include the observed LAD domain clustering. Also, we have discussed data resolution in more detail in the results (see reviewer #2).

      How does this data correlate with TSA-seq, another antibody-based method developed by the Belmont lab, but collaboratively developed for use in identifying LADs (ie Dam alternative) with the Van Steensel group?​ I can imagine there are numerous advantages to this approach (radius of "labeling" being one).

      TSA-seq provides a different perspective on genome – NL interactions, given its distance dependence rather than contact. We have added a comparison with TSA-seq to the Discussion.

      **When Dam-Lamin B1 is expressed in vivo for 5-25 hours during interphase, LADs that interact with the NL become progressively labeled, eventually resulting in a layer of labeled chromatin of up to ~1 μm thick [8]. This is because LADs are in dynamic contact with the NL. We expected that in pA-DamID this layer would be thinner, because the NL-tethered Dam is only activated for 30 minutes. In addition, permeabilization depletes small molecules including ATP and thus prevents active DNA remodeling in the nucleus [26]. Indeed, pA-DamID yields a m6A layer that is ~2.5 fold thinner than the layer in cells that express Dam-Lamin B1 in vivo (Fig. S2A-C). This is not an artifact due to collapse of chromatin onto the NL caused by the permeabilization, because permeabilization of cells expressing Dam-Lamin B1 in vivo did not significantly reduce the thickness of the m6A layer compared to directly fixed cells (Fig. S2C). The thin layer of labeled DNA obtained by pA-DamID points to an improved temporal resolution of pA-DamID compared to conventional DamID.**

      ● I think this requires a bit more care. Your previous work clearly demonstrates LADs are dynamic. Others in the field have shown that these domains are also constrained within the larger sub-chromosomal compartment (self-interaction) of LADs (e.g. Luperchio 2018) within a chromosome. So, this is truly a temporal "snapshot" that may miss some regions of LADs that are less directly (or more dynamically) associated with the lamina, but still compartmentalized into the larger LAD sub-chromosomal compartment. It is unclear if the treatment used for this study perturbs these LAD-lamina​ dynamic​ interactions--one can imagine that the LADs are much less mobile generally under the protocol described in your supplemental information. In other words, ​LADs don't collapse, nor do they behave in the same way they would after permeabilization​. The technique has compromised some of that --which is actually fine for most of the purposes in this manuscript, but this needs to be discussed.

      As the reviewer points out, there are fundamental differences between DamID and pA-DamID in their m6A deposition that should be clear from the text. We elaborated on this in the comparison between pA-DamID and DamID.

      ● In addtion, imaging data showing dam-LaminB1/2 plus m6A-tracer is missing (figure S2). This should be included. Is the intensity of the "tracer" similar between conditions? If so, were the exposures kept constant in all images? This is important since the decay rate is highly related to intensity of signal.

      We are afraid that this figure has been misinterpreted. We have changed the figure labels and legend to explain it better. The HT1080 Dam-Lamin B1 clonal cells (new clone kindly supplied by Jop Kind) still showed significant variation in m6A-Tracer intensity per cell, suggesting different expression levels of Dam-Lamin B1. To create optimal images for halfway decay estimation, laser settings were changed between images. This has now been mentioned more clearly in the methods.

      **In some cell types, especially in​ HCT116 and ​hTERT-RPE​ cells, we noted local discrepancies between the two methods (Fig. 2A,bottom panel). These differences involve mostly regions with low signals in DamID that have higher signals in pA-DamID. However, such differences are not obvious in HAP-1 and K562 cells.**

      ● Only HCT116 data is shown in the indicated figure. hTERT-RPE cells are shown in the accompanying supplemental figure and use a different antibody (lamin B2) as the target for the pA-Dam.

      We have changed the pointer to include the supplementary figure.

      (See reviewer #1 for a similar comment.)We agree that the comparison between Lamin B1 DamID and Lamin B2 pA-DamID in hTERT-RPE cells leads to sense of incompleteness and confusion. We suggest to generate Lamin B1 pA-DamID data in hTERT-RPE cells to solve this – provided that the current Corona virus shutdown will not prevent us from doing this experiment.

      This brings up another point: the data (log2 ratio schema) shown in figure 2 is for HCT116 lamin B1 pA-Dam. Yet, the subsequent studies for transient/building interactions during G1 and into S (Figure 3) are done in hTERT-RPE cells using lamin B2. To be consistent, data from lamin B2 should be used in both figures (it seems lamin B2 data is available for all cell types). The comparison of Dam-Lamin B1 can be addressed in the Venn overlays (as they are now) and in the supplements. The hTERT-RPE data should be in Figure 2 since it is followed up on in the subsequent figure (ie it fails to meet the definition of being relegated to 'supplemental' data).

      As written in the response above, we suggest to generate Lamin B1 pA-DamID data in hTERT-RPE data.This will allow us to make a more consistent story and address these comments.

      **suggesting that the separation of LADs and inter-LADs becomes progressively more pronounced after mitosis. Nevertheless....**

      ● This is overstated, especially given the previously mentioned work (Luperchio, Zhang). More accurate to say LADs ​association with the nuclear lamina becomes more pronounced​. LADs (predominantly B-compartment) and inter-LADs (predominantly A-compartment) show much earlier separation from each other. This may be distinct from association with the lamina. This is an important distinction as it may lead to different hypotheses regarding mechanisms of LAD targeting/association with the lamina.

      We agree that this is an overinterpretation of our data. We have changed the phrasing to make it more accurate.

      **Progression from prometaphase to late telophase in HeLa cells takes about 1 hour [33], suggesting that this timepoint captures the initial interactions with the reforming NL. Remarkably, the majority of these interactions is shared with later time points, indicating that most LADs can interact with the NL throughout interphase and are defined (and positioned at the NL) very soon after mitosis.**

      ● There is wide variability in this number, some cells rapidly exit, others take significantly longer. This number is an average (and, for what it's worth, based on a very compromised cancer cell line). The "interactions' mapped are likely reflecting the ensembe measurements of the many cells that have transited into G1. Also, this statement seemingly directly contradicts the premise of many of your following data/interpretations of a sort of step-wise wave or prefered interactions from telomere proximal toward centromeric regions. This also disagrees with your previous work (Kind et al) and more recent work regarding positioning to the NL very soon after mitosis. Again, this is BULK (many cells of a continuum of configurations) versus single cell observations. This is overstated.

      We felt that there was a need to explain why we interpret the 1h time point as the initial interactions with the NL and included this reference, but the reviewer is correct that this number can vary greatly between cell types and conditions. We have removed the reference and now include FACS and imaging data supporting this claim directly.

      We have changed the phrasing of these results to make our interpretation clearer.

      **We next looked into characteristics of the dynamic LADs. At early time points, LADs with decreasing interactions do not have lower pA-DamID scores than stable LADs, suggesting that their ​detachment from the NL is not simply due to weak initial ​binding**

      ● The methods used here are dynamic proximity measures. Words like "binding" and "attachment" should be avoided (use interacting, associated, etc )

      Good point. We have replaced all occurrences of these words.

      **LAD dynamics are linked to telomere distance and LAD size in multiple cell types**

      ● Perhaps I am missing something, but I find relatively little data showing centromere-proximal LADs across cell cycle stages (referring here to Log2 ratio plots similar to what is shown for telomere-proximal LADs, Supplemental figure 6 is the only place where this is obvious.).

      To better illustrate the inverse dynamics of telomeres and centromeres in hTERT-RPE cells, we have changed Fig. 3B to a full chromosome overview.

      ● In addtion, it seems to me that you are arguing in this and the preceding section for the following parameters: intensity of the LAD region. ie small, telomere-proximal, more euchromatic, AND less "intensely" associated.

      ● What is a "small" LAD? 100 kb or less? In Figure 2 (HCT1016, log 2 ratios), the original observation that leads into a discovery of changing NL associations through the cell cycle, the LAD that changes appears to be at least average size. Perhaps a "small" LAD adjacent to an "average" LAD. Nor do the signals appear to be all that low. There are regions within this sub-chromosomal plot that do appear to be "small" "low intensity" LADs. I am uncertain what parameters are defining these attributes. Are the cut-offs the same between cell types (ie is there a rule here?).

      We do not set any cut-offs for any features that we compare with. We took the strategy to define stable and dynamics LADs (Fig. 3C) and ask whether there are differences in feature distributions, including LAD size, replication timing and other features. As you can see in Fig. 3E, LADs with decreasing NL interaction are smaller than stable or increasing LADs. This strategy is consistent between cell lines. To assist the reader in following our reasoning, we have added LAD domains and their differential status to Fig. 3B.

      ● The rules outlined above seem to break down across the different cell types. In particular, the number of active genes per Mb seems to have very little correlation overall with LADs that change. In addition, it is very unclear if "LAD size" is really a readout of both size AND intensity of interactions (understanding that this is not necessarily a direct quantitative measure of interactions).

      This comment reflects our reasoning why we added a comparison between cell types in Fig. 4. Indeed, we find no general trend that active gene density correlates with LADs with decreasing NL interactions in every cell type. In contrast, LADs with decreasing NL interactions are consistently close to telomeres and smaller in size than stable or increasing LADs. We made it clearer that LAD size solely reflects the genomic size in basepairs.

      **Correlation of pA- determined LADs that change into G1/S with B-compartment sub-types**

      ● There is certainly Hi-C data on most (all?) of the cell types analyzed in this manuscript. It would be very useful for the authors to parse out how the gain/loss LADs correlate with the B1, B2. A1, A2 (etc) compartment classifications. This may help to address the point above.

      We have now included a comparison with Hi-C sub-compartments (Fig. 4F).

      **Nucleosomal pattern of pA-DamID digestion/amplification (figure S3)**

      ● Onset of apoptosis needs to be ruled out. The nucleosomal (laddering) pattern could be due to DNA getting cleaved through programmed cell death pathways after permeabilization. These fragments could easily be amplified by the subsequent DamID protocol.

      Amplification of apoptotic fragments, if present, is visible in DamID assays using the negative controls. Every library preparation, we include one or more negative controls in which we omit DpnI. If apoptotic fragments are present in this negative control, these can ligate to the DamID adapter and result in amplification, which we consistently do notsee. We have added a supplementary figure that shows this (Fig. S3A).

      **Definition of 'bulk' assays**

      ● All of the assays were done in bulk. Some were synchronized, some were not. This is important since the implication is that anything not 'bulk' is single-cell. Throughout the manuscript and in the figures, please refer to the conditions as 'synchronized' versus 'unsynchronized'

      The reviewer is correct that our terminology is wrong. We changed all occurrences of “bulk” to “unsynchronized”.

      **Much of supplemental Figure 6 should be in a main figure**

      ● It is puzzling why the first (and most easily seen/interpreted) description of LAD organization relative to telomeres/centromeres after exit from mitosis is relegated to supplemental figures. It is a foundational experiment(s) for the paper.

      We have changed the zoomed-in Fig. 3B with a chromosome overview that better captures this main observation. We see the remainder of Fig. S6 as technical controls and details of the experiment that are useful to include but not necessary as main figure.

      **pA-Dam is possibly influenced by cell-cycle related chromatin accessibility (particularly at mitotic exit)**

      ● During the transition from mitosis to early G1, there are dynamic changes to chromatin state that are directly coupled to the cell cycle. A recent report, for instance, highlights that interactions of antibodies (or other proteins) with H3K9me2/3 modifications is likely influenced by phosphorylation of histone tails. The dynamics of histone modification/chromatin state possibly occluding or interfering with the interpretation of the results must be discussed.

      Similar to DamID, pA-DamID utilizes a Dam-control to measure DNA accessibility and control for this. We show that a change in pA-DamID score is due to changes in NL reads, while the Dam reads do not change (Fig. S6F). In other words, we find no evidence that a change in chromatin state impacts the accessibility as measured by our Dam-control and thereby influences the results. We now repeat this observation in the discussion.

      Reviewer #3 (Significance (Required)):

      N/A

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      Referee #3

      Evidence, reproducibility and clarity

      In the manuscript by Schaik et al (Van Steensel laboratory) the authors describe a very clever approach to identifying Lamina Associated Domains (LADs) using the principles of the 'cut-n-run' strategy. Specifically, they engineer the Dam methyltransferase used in canonical DamID in frame with a protein A moiety capable of interacting with an antibody (in this case lamins--B1, B2 and A/C). After permeabilization, cells are incubated with antibodies, then pA-Dam purified protein--for a brief time window--is added to mark associated DNA with GmATC. This technique is a valuable contribution to the field, particularly since, as the authors point out,an advantage of pA-DamID is that the labeled DNA can also be visualized in situ using the m6A-Tracer, before this DNA is sequenced. This allows for validation of findings and is highly amenable to cell sorting technologies. In addition, this technology allows for a time-resolved measure of LADs not currently available by standard DamID. The authors apply this technology to four different cell types. They noted that the 'maps' generated by the is technology differed from canonical DamID at very specific regions (small LADs in very localized regions) . They then embark on a series of experiments to show that these differences arise from cell cycle -related differences that are differentially picked up by the methods--with the pA-DamID allowing for dissection of more discrete cell cycle stages/configurations. In general they find an initial preference for sub-telomeric LADs to associate with the nuclear lamina fist, then more centromeric. There is some data suggesting loss/gain of LADs in specific regions/with specific features. The manuscript is well written and the data well presented. However, there are some points that need to be addressed . Overall, there is some oversimplification or omission of previous data in the field, a lack of clarity in how some of the data was interpreted, and some areas where clarification and/or additional analyses would be helpful. I sincerely hope the authors find the following critiques to be useful. Thank you for the opportunity to review your very nice work.

      Introduction:

      Microscopy studies found that telomeres are enriched near the NL in early G1 phase, leading to the hypothesis that telomeres may assist in NL reassembly onto chromatin [13].

      ● There have been numerous studies identifying the timing and disposition of INM proteins and Lamins at m the end of mitosis (during NE reformation). Why are you citing just this one? (e.g. ​Thomas Dechat et al., 2004; T. Dechat et al., 2000; Ellenberg et al., 1997; Haraguchi et al., 2001​)

      Furthermore, during S-phase B-type lamins have been found to transiently overlap with replication foci in the nuclear interior, at least in some cell types [20]

      ● While, technically, this has indeed been reported, this study is from 1994 and has not been repeated. The cells used in this study (3T3 fibroblasts) are widely used and others have not noted this phenomenon. Soften this.

      Other studies have indicated that lamins are important for DNA replication [reviewed in 21].

      ● Likewise, direct roles for lamins in replication are controversial (acknowledged in the small section of the cited review on the role of lamins in replication).

      ● Perhaps combine the two sentences above to soften the implication that this is a "known" role of B-type lamins. e.g. "A handful of studies have implicated a role for B-type lamins in replication, but the direct role of the lamina in this process remains unclear. Nonetheless, ......"

      Results:

      So far, the cell cycle dynamics of genome - NL interactions have primarily been studied by microscopy. While these studies have been highly informative, they were often limited to a ​few selected loci.​

      ● Please cite your own study (Kind et al.) and other recent papers (Luperchio et al.-​https://www.biorxiv.org/content/10.1101/481598v1&#x200B;; Zhang et al., Nature-​https://www.nature.com/articles/s41586-019-1778-y&#x200B;) in which they were either 1) not limited to a few selected loci and/or 2) not microscopy-directed studies? There is an argument to be made here for the resolution (time and b.p.) you have achieved through your studies that these studies did not.

      ● How does this data correlate with TSA-seq, another antibody-based method developed by the Belmont lab, but collaboratively developed for use in identifying LADs (ie Dam alternative) with the Van Steensel group?​ I can imagine there are numerous advantages to this approach (radius of "labeling" being one).

      When Dam-Lamin B1 is expressed in vivo for 5-25 hours during interphase, LADs that interact with the NL become progressively labeled, eventually resulting in a layer of labeled chromatin of up to ~1 μm thick [8]. This is because LADs are in dynamic contact with the NL. We expected that in pA-DamID this layer would be thinner, because the NL-tethered Dam is only activated for 30 minutes. In addition, permeabilization depletes small molecules including ATP and thus prevents active DNA remodeling in the nucleus [26]. Indeed, pA-DamID yields a m6A layer that is ~2.5 fold thinner than the layer in cells that express Dam-Lamin B1 in vivo (Fig. S2A-C). This is not an artifact due to collapse of chromatin onto the NL caused by the permeabilization, because permeabilization of cells expressing Dam-Lamin B1 in vivo did not significantly reduce the thickness of the m6A layer compared to directly fixed cells (Fig. S2C). The thin layer of labeled DNA obtained by pA-DamID points to an improved temporal resolution of pA-DamID compared to conventional DamID.

      ● I think this requires a bit more care. Your previous work clearly demonstrates LADs are dynamic. Others in the field have shown that these domains are also constrained within the larger sub-chromosomal compartment (self-interaction) of LADs (e.g. Luperchio 2018) within a chromosome. So, this is truly a temporal "snapshot" that may miss some regions of LADs that are less directly (or more dynamically) associated with the lamina, but still compartmentalized into the larger LAD sub-chromosomal compartment. It is unclear if the treatment used for this study perturbs these LAD-lamina​ dynamic​ interactions--one can imagine that the LADs are much less mobile generally under the protocol described in your supplemental information. In other words, ​LADs don't collapse, nor do they behave in the same way they would after permeabilization​. The technique has compromised some of that --which is actually fine for most of the purposes in this manuscript, but this needs to be discussed.

      ● In addtion, imaging data showing dam-LaminB1/2 plus m6A-tracer is missing (figure S2). This should be included. Is the intensity of the "tracer" similar between conditions? If so, were the exposures kept constant in all images? This is important since the decay rate is highly related to intensity of signal.

      In some cell types, especially in​ HCT116 and ​hTERT-RPE​ cells, we noted local discrepancies between the two methods (Fig. 2A,bottom panel). These differences involve mostly regions with low signals in DamID that have higher signals in pA-DamID. However, such differences are not obvious in HAP-1 and K562 cells.

      ● Only HCT116 data is shown in the indicated figure. hTERT-RPE cells are shown in the accompanying supplemental figure and use a different antibody (lamin B2) as the target for the pA-Dam.

      ● This brings up another point: the data (log2 ratio schema) shown in figure 2 is for HCT116 lamin B1 pA-Dam. Yet, the subsequent studies for transient/building interactions during G1 and into S (Figure 3) are done in hTERT-RPE cells using lamin B2. To be consistent, data from lamin B2 should be used in both figures (it seems lamin B2 data is available for all cell types). The comparison of Dam-Lamin B1 can be addressed in the Venn overlays (as they are now) and in the supplements. The hTERT-RPE data should be in Figure 2 since it is followed up on in the subsequent figure (ie it fails to meet the definition of being relegated to 'supplemental' data).

      suggesting that the separation of LADs and inter-LADs becomes progressively more pronounced after mitosis. Nevertheless....

      ● This is overstated, especially given the previously mentioned work (Luperchio, Zhang). More accurate to say LADs ​association with the nuclear lamina becomes more pronounced​. LADs (predominantly B-compartment) and inter-LADs (predominantly A-compartment) show much earlier separation from each other. This may be distinct from association with the lamina. This is an important distinction as it may lead to different hypotheses regarding mechanisms of LAD targeting/association with the lamina.

      Progression from prometaphase to late telophase in HeLa cells takes about 1 hour [33], suggesting that this timepoint captures the initial interactions with the reforming NL. Remarkably, the majority of these interactions is shared with later time points, indicating that most LADs can interact with the NL throughout interphase and are defined (and positioned at the NL) very soon after mitosis.

      ● There is wide variability in this number, some cells rapidly exit, others take significantly longer. This number is an average (and, for what it's worth, based on a very compromised cancer cell line). The "interactions' mapped are likely reflecting the ensembe measurements of the many cells that have transited into G1. Also, this statement seemingly directly contradicts the premise of many of your following data/interpretations of a sort of step-wise wave or prefered interactions from telomere proximal toward centromeric regions. This also disagrees with your previous work (Kind et al) and more recent work regarding positioning to the NL very soon after mitosis. Again, this is BULK (many cells of a continuum of configurations) versus single cell observations. This is overstated.

      We next looked into characteristics of the dynamic LADs. At early time points, LADs with decreasing interactions do not have lower pA-DamID scores than stable LADs, suggesting that their ​detachment from the NL is not simply due to weak initial ​binding

      ● The methods used here are dynamic proximity measures. Words like "binding" and "attachment" should be avoided (use interacting, associated, etc )

      LAD dynamics are linked to telomere distance and LAD size in multiple cell types

      ● Perhaps I am missing something, but I find relatively little data showing centromere-proximal LADs across cell cycle stages (referring here to Log2 ratio plots similar to what is shown for telomere-proximal LADs, Supplemental figure 6 is the only place where this is obvious.).

      ● In addtion, it seems to me that you are arguing in this and the preceding section for the following parameters: intensity of the LAD region. ie small, telomere-proximal, more euchromatic, AND less "intensely" associated.

      ● What is a "small" LAD? 100 kb or less? In Figure 2 (HCT1016, log 2 ratios), the original observation that leads into a discovery of changing NL associations through the cell cycle, the LAD that changes appears to be at least average size. Perhaps a "small" LAD adjacent to an "average" LAD. Nor do the signals appear to be all that low. There are regions within this sub-chromosomal plot that do appear to be "small" "low intensity" LADs. I am uncertain what parameters are defining these attributes. Are the cut-offs the same between cell types (ie is there a rule here?).

      ● The rules outlined above seem to break down across the different cell types. In particular, the number of active genes per Mb seems to have very little correlation overall with LADs that change. In addition, it is very unclear if "LAD size" is really a readout of both size AND intensity of interactions (understanding that this is not necessarily a direct quantitative measure of interactions).

      Correlation of pA- determined LADs that change into G1/S with B-compartment sub-types

      ● There is certainly Hi-C data on most (all?) of the cell types analyzed in this manuscript. It would be very useful for the authors to parse out how the gain/loss LADs correlate with the B1, B2. A1, A2 (etc) compartment classifications. This may help to address the point above.

      Nucleosomal pattern of pA-DamID digestion/amplification (figure S3)

      ● Onset of apoptosis needs to be ruled out. The nucleosomal (laddering) pattern could be due to DNA getting cleaved through programmed cell death pathways after permeabilization. These fragments could easily be amplified by the subsequent DamID protocol.

      Definition of 'bulk' assays

      ● All of the assays were done in bulk. Some were synchronized, some were not. This is important since the implication is that anything not 'bulk' is single-cell. Throughout the manuscript and in the figures, please refer to the conditions as 'synchronized' versus 'unsynchronized'

      Much of supplemental Figure 6 should be in a main figure

      ● It is puzzling why the first (and most easily seen/interpreted) description of LAD organization relative to telomeres/centromeres after exit from mitosis is relegated to supplemental figures. It is a foundational experiment(s) for the paper.

      pA-Dam is possibly influenced by cell-cycle related chromatin accessibility (particularly at mitotic exit)

      ● During the transition from mitosis to early G1, there are dynamic changes to chromatin state that are directly coupled to the cell cycle. A recent report, for instance, highlights that interactions of antibodies (or other proteins) with H3K9me2/3 modifications is likely influenced by phosphorylation of histone tails. The dynamics of histone modification/chromatin state possibly occluding or interfering with the interpretation of the results must be discussed.

      Significance

      N/A

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      Reply to the reviewers

      Reviewer #1 (Evidence, reproducibility and clarity (Required)): **Summary:** The work reports finding a molecular genetic basis for individual differences in behavior in different strains of outbred mice, even including individual behavioral differences between mice of the same inbred genetically isogenic strain. The authors were able to measure copy numbers for the tandemly repeated intronic snoRNA clusters SNORD115 and SNORD116 and found correlation with measures of anxiety in open-field test and elevated plus maze. Expression data for previously proposed targets of these snoRNAs are also provided.

      We note that this description represents only part of the achievements in our paper. The key of our paper is that we have not only used "different strains of outbred mice", but in addition one very different species of mouse (Apodemus) and a Guinea pig species. We believe that the test in very different species with very different genetic backgrounds is the crucial proof for the specificity of the effect.

      **Major comments:** 1.The techniques to measure copy numbers are challenging and the authors' conclusion that ddPCR was their method of choice is convincing. They were able to obtain limited optical mapping (Bionano zephyr) data, only for SNORD116 and only in mouse, but these data are useful to corroborate those obtained with ddPCR. 2.Figure 3 reports single copy numbers for individuals that are presumably heterozygous. Do we have to assume that the numbers reported represent the larger alleles since the ddPCR method does not allow to distinguish two different size alleles, as was shown for optical mapping?

      The numbers are derived from the whole genome DNA, i.e. represent the cumulative copy number of both alleles. We have updated the text to make this clear.

      3.The analyses reported do not take into account the specific parental origin of the alleles used in the regression analyses. Since PWSCR-specific SNORDs are only expressed from the paternal chromosomes, this generates some uncertainty about the whole dataset.

      We had explained why it is not possible to distinguish the two alleles with the current technology. Hence, it is evidently also not possible to determine which allele comes from the paternal side. In fact, given that we showed that copy numbers can change every generation, even the knowledge of which chromosome is the paternal one would not predict its copy number. Accordingly, it lies in the nature of the whole phenomenon that this uncertainty is given. It is therefore just the more surprising that we still can observe correlations that are much stronger than has been shown for natural alleles of any genetic locus implicated in behavioral traits so far.

      4.Lines 353-365: The ankrd11 exon-specific RNAseq data are confusing and too preliminary. More work needs to be done to resolve the splice variants in this region and their relationship to SNORD116 copy numbers. Alternatively lines 356-361 should be deleted.

      We have included the data to show that the mechanism must be different from the one that is seen for Htr2c. This difference is clearly documented and we should therefore like to retain it. What is missing is to show the actual mechanism by which SNORD116 causes the alternative splicing. This will require more biochemical approaches that go beyond the current study.

      5.In all tested rodents, higher SNORD copy number was correlated with higher relative anxiety score. In the human samples, however, higher anxiety scores were associated with lower copy numbers. These apparently contradictory results are not mentioned in the abstract, and are not satisfactory explained in the text.

      We have decided to leave the human data out from the current manuscript. First, the behavioral tests for the rodents are indeed not directly comparable with the questionnaire scores in humans. Second, in human genetics one usually asks the results to be confirmed in an independent study. hence, we plan to extend the human data, but to present them eventually in a follow-up paper.

      6.Extension to other species would be desirable but was limited by availability of genomic data: Results are presented for wood mouse only for SNORD115 and for the guinea pig for SNORD116.

      Given that we show a strong correlation between SNORD115 and SNORD116 copy numbers for those species where the information is available for both loci, we do not think that this is a major limitation of our study.

      **Minor comments:** 1.The authors present skull shape data related to SNORD116 copy numbers, but fail to consider how these data are relevant to the craniofacial abnormalities reported in an ankrd11 mutation. Barbaric et al (2008) reported a dominant ENU- induced mutation caused shortened snouts, wider skull, deformed nasal bones, reduced BMD, reduced osteoblast activity and reduced leptin levels. This phenotype was traced to a heterozygous missense mutation (conserved glutamate to lysine change) in an HDAC binding site. They postulated that the mutation fails to recruit HDACs to a transcription complex and to inhibit hormone-receptor activated gene transcription. What is the postulated link between this mechanism and the here reported skull shape data correlated with SNORD copy number variation?

      The described missense mutation is located in the differentially spliced exon, i.e. a direct functional link is given. We have added this information to the text and compared the direct phenotypic effects from their study and our study.

      2.The observed co-variation of copy numbers between the two SNORD clusters could indicate a duplication involving the entire region, This could be tested by determining the dosage of IPW, UBE3a and Snrpn genes.

      While this is a theoretical possibility, it was not described in the literature before. Also, in our systematic survey of copy number variation in mouse populations (Pezer et al. 2015) we did not find a deviation of these genes from expected diploid copy number in any of the populations analysed.

      3.Line 129 "the RNA coding region" and Line 148 "snoRNA coding parts" (and elsewhere) does seems correct, as by definition, this is non-coding RNA. The region they are referring to could be called the "processed C/D box snoRNA". The mechanism that generates these C/D box snoRNAs is well established: the "genes" are located in introns of host genes - and after transcription - the spliced out introns are exonucleolytically trimmed to the functional sizes. Both SNORD115 and 116 clusters are within a large transcript that originates from the SNRPN promoter of the paternal allele.

      We adjusted the wording to make clear that we refer to the mature RNAs.

      4.Figure 2 does not show data on skull shape as claimed in the legend.

      We apologize - this was a carry-over from an older version of this figure. The skull shape analysis had been moved to a new figure in the current version of the manuscript.

      5.S1 Figure: Snprn should be Snrpn

      Thank you for spotting the error - we have corrected this

      Reviewer #1 (Significance (Required)): This provocative work proposes the regulation of behavioral variance by dosage changes of a regulatory RNA. The dosage changes are apparently caused by dynamic and frequent alteration in copy number. This is a novel concept and worthy of publicizing. Extensive data documentation is provided for others to analyze and possibly replicate. The data potentially throw light on the function of the tandemly repeated imprinted snoRNA clusters in the PWS critical region. Novel aspects of this work include the discovery of copy number variation of these snoRNAs; and validation of a target of SNORD116: Ankrd11 is one of many potential targets of SNORD116 that was previously computationally predicted, this paper reports experimental evidence for this interaction. The work would be of interest to researchers in behavioral evolution, non-coding RNA function, epigenetics and overall genome evolution. Define your field of expertise with a few keyword: Molecular genetic disorders, Prader-Willi syndrome, mouse models

      Reviewer #2 (Evidence, reproducibility and clarity (Required)): **Summary** Maryam Keshavarz et al. aimed at seeking the molecular basis underlying individual behavioral variance within populations. Focusing on the Prader-Willi Syndrome (PWS) gene complex, which has been well recognized being associated with neurodevelopmental disorders, anxiety and metabolic issues, the authors found that the levels of PWS region's small nucleolar RNAs SNORD 115/116 of individual animals correlated with these individuals' behavioral test scores. The variations in transcript processing of some anxiety-associated target genes also revealed correlation with SNORD 115/116 copy numbers. Authors implicated that the copy numbers of SNORD 115/116 within PWS plausibly influenced behavioral variances among individuals. • Authors first validated that the droplet digital PCR (ddPCR) was suitable for quantifying variations in copy numbers of genomic clusters. Their ddPCR data showed confident correspondence with reads calculation of whole-genome-seq dataset. Also, ddPCR showed good replicability and congruent tissue-to-tissue similarity. • Authors found the ddPCR-measured SNORD copy numbers from several mice populations showed significant regression with SNORD RNA levels, respectively. Also, the anxiety profiling using Open Field Test and Elevated Plus Maze test indicated a significant regression between SNORD copy numbers and anxiety profiling scores, namely individual mouse with higher copy numbers received higher relative anxiety scores. Some other representative genes outside PWS, such as Sfi1 and Cwc22, failed to show such copy number-anxiety score regression. • Authors applied RNA-seq of individual mice with different SNORD 115/116 copy numbers and analyzed potential target gene regions. They found the level of alternative splice-resulted exon Vb of gene Htr2c, a serotonin receptor, was positively correlated with SNORD 115 copy number. Also, an alternative splicing product of gene Ankrd11, a chromatin receptor regulating GABA receptor, was found to positively correlated with SNORD 116 copy number. Positive correlation to SNORD copy numbers also occurred to some Htr2c and Ankrd downstream genes. • Authors used a landmark-based analysis to score mice craniofacial features and found the scores were in relationship with SNORD 116 copy numbers. • Authors also found significant regression between SNORD copy numbers and behavioral evaluations in other rodents. In humans, the Tridimensional Personality Questionnaire (TPQ) based evaluation also showed correlation with SNORD 115 and 116 copy numbers. **Major comments** The study mainly revealed important correlations between copy numbers of 2 small nucleolar RNAs and cognitive behavioral variance of different individual animal. Although very useful and important findings, the study did not provide any evidence about the causality between SNORD 115/116 and the observed behavioral phenotypes. For instance, • #1: the behavioral observations (i.e. anxiety) may not be merely regulated by the PWS gene complex.

      It is already well understood that the respective behavioral observations have a polygenic basis. But our data show that the SNORD copy numbers act as major modulators of the behavior.

      • #2: the paper did not show if manipulations on mouse SNORD 115/116 could affect target genes as well as the consequential behavioral phenotypes.

      A direct interaction between SNORD115 and its target gene HTr2c has previously been shown in cell culture experiments. Further, we show that the commonly used inbred mouse strain C57Bl6 carries already different copy number alleles that would not be different from artificial manipulation of the copy number. There is a long tradition in mouse genetics to accept also spontaneous alleles as genetic proof, not only the alleles that were created by artificial intervention.

      Further, as also pointed out in response to reviewer 1, in the absence of the possibility to do a direct genetic manipulation in a given genetic background, we use the comparative analysis between different genetic backgrounds to prove causality.

      Reviewer #2 (Significance (Required)): Authors provided a potential molecular basis regulating the PWS region, which is a genomic imprinted gene complex and related to many neurodevelopmental diseases in mammals, including humans. Considerably cost-saving than whole-genome deep-seq, the application of droplet digital PCR on copy number (esp. in stretching regions) measurement can overcome some technical difficulties, for example, qPCR has limit in resolution when differentiating subtle variance in copy numbers; the Nanopore seq and current mapping algorithm show difficulties when placing the internal repeats also. Authors proposed SNORD copy number as a potential explanation to the individual-to-individual variance within the same species or even the same population.

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      Reply to the reviewers

      Reviewer #1:

      (Evidence, reproducibility and clarity (Required)):

      This study aims to develop tools for yeast researchers to automatically segment and classify yeast colonies. The machine learning method enables rapid classification compared to manual counting.

      \*MAJOR CONCERNS:***

      Please include additional details about the types of images that must be captured for segmentation and categorization. It is important to provide details of what level of magnification might be needed during image capture. We anticipate that providing clear protocols for altering thresholds to classify colonies might be one way to overcome this challenge

      That’s correct. Details on image acquisition, such as the level of magnification, are important to obtain accurate results.

      To address this, we provide a detailed protocol in our companion article on ProtocolExchange: https://protocolexchange.researchsquare.com/article/nprot-7305/v1

      We have updated the manuscript to include this link.

      While the program crops colonies and segments them accurately, there is no spatial information of where these colonies are located in the image. This loss of spatial information limits the ability to use this platform to identify colonies of interest following experiments such as a genetic screen.

      In principle it would be possible to retain the location of each cropped colony in the form of (x,y) coordinates in pixels. This could be included in a future release. However, we doubt the utility of such information for a genetic screen, unless identification of a positive hit could be linked to robotic picking of the identified colony (which would certainly go beyond the scope of this work). In reality, researchers will pick positive hits manually anyways, making our pipeline superfluous for such an application. We emphasize that we have developed our pipeline for large-scale quantification of red/white color assays. Here the pipeline makes a huge difference, as compared to manual counting.

      The inability to accurately recognize sectored colonies as sectored (rather than red) is a significant limitation to the usage of this program for quantitative assays. While differentiating between red and white colonies is useful, the conclusion by the authors about its value for quantitative assays is limited unless variegation can be accurately defined. The authors should either soften this conclusion or qualify what quantitative measurements might mean given the limitations of their classification program. This somewhat diminished our overall enthusiasm.

      The reviewer correctly points out that our algorithm shows lower accuracy when differentiating between red and variegating colonies than when differentiating between white and non-white colonies (including red, variegating and pink). Given this observation, we initially focused on predicting white vs. non-white colonies with our tool. However, the output of our pipeline also includes more granular predictions of numbers of white, red, pink and variegating colonies. We therefore leave it up to the user to decide which level of granularity is more appropriate, taking into account the tradeoff between granularity and accuracy. In particular, we note that for the colonies we tested, splitting the non-white category of predictions into red, variegating and pink resulted in a decrease of sensitivity from 0.98 for the non-white category to 0.86-0.88 for the individual categories, while the corresponding specificity showed a smaller reduction from 1.0 to 0.97-0.98. Considering that a lack of any predictions for the red, pink, and variegating categories effectively prohibits the researcher from detecting them at all, even a reduced sensitivity may be better than nothing and therefore acceptable in this case. In order to make this clearer in the text, we provide a more detailed comparison of performance metrics between levels of prediction, which may help to guide the user’s decision.

      This program must be benchmarked with other colony classifiers. Cell Profiler is an example of a popular yeast colony segmentation program. How does this machine learning based tool compare with other colony segmentation and categorization programs. One possibility is to include an additional figure that compares their program with clear benchmarks. The outcome of effort based on benchmarking is not as important since we believe it is useful to have many alternatives for yeast segmentation and categorization. We think this revision would be essential to the manuscript and would add significant value.

      We have used other approaches and were not satisfied with the outcomes. Hence, we developed our own pipeline, specifically designed to accurately distinguish red from white colonies and quantify such assays at a large scale.

      When using CellProfiler we could not reliably distinguish variegating, pink, and red colonies. White colonies show up in the Red, Blue and Green channel, Red colonies mainly in the Red Channel. Therefore, variegating, pink and red colonies can be distinguished from white by reduced Blue and Green values, which is indirect and caused several issues. One of the problems was reflection of the flash during image acquisition, giving two reflective white patches on each colony that differed in pixel size depending on the magnification and colony size. We tried to prevent reflection with a ‘tent’, which reduced but could not eliminate the reflection. Therefore, the MaxIntensity of the Green/Blue channel was always the same of each colony, impeding classification. Furthermore, most red/pink colonies had a slim white rim, which was sometimes bigger/smaller and the relative area of rim to colony depends on the colony size, which made it impossible to tell a bit variegating from red by the output values from CellProfiler.

      If deemed useful by the editors, we will be happy to mention this in the manuscript. A systematic comparison with other classifiers seems to be a bit of an overkill though. As stated by this reviewer, the outcome of such comparison would not matter much. It is important that the community has several approaches to choose from, so that the best solution can be found for each specific application.

      \*MINOR CONCERN***

      The program currently saved cropped images of each segmented colony. This takes up a lot of storage space. It might be useful to provide an option to save or not save these cropped images. This flexibility will be valuable for users but does not detract from the major conclusions of the manuscript.

      While we appreciate that the need to save individual images of cropped colonies may be a drawback for some users, in the current implementation it is not possible to avoid this step. One could imagine a scenario in which all cropped images were stored in RAM prior to classification rather than written to a computer’s disk; however, we believe that most users would have more limitations on the availability of RAM than on disk storage, therefore making this option also not feasible.

      The authors have provided excellent examples of colonies they believe are red, white or sectored. More accurately defining a pink colony would be valuable for users of this program. How much of red is classified as pink by this program?

      As the reviewer points out, it is difficult to give an objective definition of a pink colony. In this case, we relied exclusively on subjective expert annotations to define which colonies were pink (as well as for all other categories).

      We acknowledge that this may introduce some error into the model, as there may be some overlap between red and pink colonies or between pink and variegating colonies; however, this problem also exists in the case of manual annotation. As shown in Figure 1d, for the colonies we tested, 4 out of a total of 55 colonies annotated as red by an expert were predicted as pink by our algorithm. We would like to emphasize that our pipeline alleviates biases between different researchers who would be annotating colony color manually, therefore improving reproducibility. Such biases could be subjective or objective, such as different monitors used to inspect the images.

      Providing an example data set with the protocol would be helpful for users with limited Python experience. In combination with their protocol on Protocol exchange, this would serve as a valuable resource for novices in programming.

      We agree with the reviewer’s suggestion and will be happy to provide an example dataset used in the manuscript. We will defer to the journal’s guidelines as to the best way to share these raw images.

      One technical issue of the program is that the program tries to open all files in the specified folder even if they aren't jpg. This causes problems if there are additional or hidden files in the folder and the program cannot process the additional files.

      We appreciate the reviewer pointing out this issue and have fixed it in a new version of the code.

      Reviewer #1 (Significance (Required)):

      This manuscript describes a machine learning approach to segment and categorize yeast colonies based on a red/white selection assay. The approach has been implemented using Python which makes this widely accessible to many researchers. Their detailed protocol on Protocol Exchange is a valuable resource which made it possible for us to evaluate its performance. The program meets its goals of reducing user time via manual counting. It is also reasonably accurate in discriminating between red and white colonies based on our initial tests. However, there are several important concerns that the authors will need to address before this manuscript can become a valuable resource for the yeast community. It is important to note that our framework is one where we have a great interest in quantitative yeast genetics but cannot evaluate the strengths and weakness of the computational approach. So much of the review is focussed on what would be needed to make this tool more user appropriate.

      Reviewer #2

      (Evidence, reproducibility and clarity (Required)):

      \*Summary:***

      Carl et al present an application of a deep learning-based image analysis able to segment and classify individual yeast colonies by their phenotype in a special plate. They evaluated the method and show that it provides the accuracy similar to the one achieved by experts' manual classification.

      \*Major comments:***

      The key conclusions are convincing. The evaluation is performed on 3 datasets showing different properties (strong presence of phenotype, almost lack of the phenotype, gradual change of the phenotype).

      The claims are carefully formulated. The deep learning methodology (training, validation, using modern technologies such as transfer learning, Unet, augmentation) is carefully designed and carried out. The evaluation is sound. The limitations are discussed.

      For a short paper as it's formulated currently, no additional experiments are necessary.

      The methods are implemented and are available on GitHub.

      However, I'd strongly recommend to share also the data used in the paper, to make possible the reproduction of the results as well as to be used as examples for future users.

      As stated above, we agree with the reviewer’s suggestion and will be happy to provide an example dataset used in the manuscript. We will defer to the journal’s guidelines as to the best way to share these raw images.

      No replicates are provided unfortunately. The manuscript would benefit from showing results from replicates, especially because they should be easily obtainable.

      It is not clear to us to which experiment the reviewer is referring. All of the results presented in Figure 2 did include replicates, as detailed in the figure legend.

      \*Minor comments:***

      I'm not familiar with the state of the art to judge on whether prior studies are referenced.

      The text and fitures are very clear and well formulated.

      Reviewer #2 (Significance (Required)):

      Despite the conceptual innovation is average, the method is well-developed and seems to be very useful for yeast analysis.

      I'm not an expert in the application area to judge the state of the art. The carried out deep learning methodology is top notch.

      The manuscript can be interesting and useful for experts using the described assay for yeast.

      My expertise is in omics, image analysis, and machine learning.

    1. Super. Clearly defined research focus, although a massive topic in terms of scope and scale. Some thoughts.

      (1) Might not be a bad idea to revisit some of the traditional development economic works on rural to urban migration, although the push / pull factors you are identifying are much more regional or global in nature. Two good ones to look at are Todaro:

      https://www-jstor-org.proxy.wm.edu/stable/1807860?seq=1#metadata_info_tab_contents

      and Sir Arthur Lewis' Economic Development with Unlimited Supplies of Labour. Please let me know if you can't find a copy of his 1954 essay that earned him the Nobel Prize.

      (2) I'm wondering a bit about which data science methodology is best to investigate further with regard to your research topic. Argument could be made for thinking about the application of a hierarchical bayesian model with the survey data you have identified. There are also arguments in favor or gravity type models with the CDR data you are considering.

      (3) Migration patterns are such a big topic that it can be difficult to know where to start in terms of geospatial data, data science / machine learning methods / models etc…A common starting point for thinking about migration is in terms of push factors and/or pull factors, more traditionally within the context of rural to urban migration. The scope and scale of what you are investigating is of a much greater magnitude and complexity than these traditionally considered foundations from development economics. Additionally, thinking of migration of this magnitude in terms of only human movement and behavior, likely over simplifies the problem. For example, we could use CDR data to infer the origins and destinations of where people are coming from and where they wish to go as part of a transportation system model (even if it is by foot or mixed mode). I think you need something that seeks to accomplish far more than simply transport behavior and movement, because you also wish to understand the motivations behind why people choose to leave (push factors) as well as the reasons they are attracted to some location (pull factors). I think looking into agent based models as a data science method for describing, analyzing could also be a promising path forward. A simulation system, such as an ABM, may incorporate different model that address factors at the origin as well as at the destination.

      Will be very interested to find out more about the direction you have selected moving forwards! Just keep going!

    1. Really good stuff. Your research focus is very clearly defined. Some thoughts.

      (1) Migration patterns are such a big topic that it can be difficult to know where to start in terms of geospatial data, data science / machine learning methods / models etc...A traditional starting point for thinking about migration is in terms of push factors and/or pull factors, more traditionally within the context of rural to urban migration. The scope and scale of what you are investigating is of a much greater magnitude and complexity than these traditionally considered foundations from development economics. Additionally, thinking of migration of this magnitude in terms of only human movement and behavior, likely over simplifies the problem. For example, we could perhaps use CDR data to infer the origins and destinations of where people are coming from and where they wish to go as part of a transportation system model (even if it is by foot or mixed mode). I think you need something that seeks to accomplish far more than simply transport behavior and movement, because you also wish to understand the motivations behind why people choose to leave (push factors) as well as the reasons they are attracted to some location (pull factors). I think looking into agent based models as a data science method for describing, analyzing and potentially predicting migration throughout North America might be a good place to start. A simulation system, such as an ABM, may incorporate different model that address factors at the origin (crime as a push factor) as well as at the destination (income differentials, as discussed by Todaro). There is simply a lot to think about here.

      (2) It might be helpful to reflect back on some of the historical references. Todaro is an often cited source.

      https://www-jstor-org.proxy.wm.edu/stable/1807860?seq=1#metadata_info_tab_contents

      Another good one to consider is Sir Arthur Lewis and his Economic Development with Unlimited Supplies of Labour. Please let me know if you can't find a copy of his 1954 essay that earned him the Nobel Prize.

      Another good source, is WIEGO.

      https://www.wiego.org

      Just keep going!

    1. et still find that our students of color struggle and fai

      This may be a pet peeve, but I feel many in the the discipline fail to think sociologically about these kinds of questions, and focus far too much on what is right in front of their noses - language, assessment, feedback, etc. Consider, for example, Ira Kaznelson's When Affirmative Action Was White, and Rothstein's The Color of Law. They describe how the huge, transformative public policy programs of the New Deal and Fair Deal, as well as areas like housing policy, lending, zoning, etc. gave enormous, staggering, transformative boosts to white Americans, while systematically excluding people of color. The largest middle class the world has known was created through a slew of programs that people of color were excluded from, and a whole set of policies, esp, housing and lending and zoning and penal, made sure to engrave racism in our schools, housing, urban geographies, prisons, and in wealth accumulation (most family wealth is generated via the transfer of housing value from one generation to the next). Obviously many other forms of racism were important, but this huge framework of laws, regulations, and policies both reflected racism, and were absolutely crucial in reproducing it, with affects the have an enormous impact today.

      Yes racism is overdetermined. I'm just skeptical that how teachers assess or give feedback to students in first year university writing classes matters much. I say that (I hope) not defensively or out of a sense of guilt or to deflect blame. I just think we could change everything about how we give feedback and assess writing (we could even follow Inoue's "labor based" model of assessment) and this would change...nothing.

      Furthermore, from my perspective, the great strains and stresses on universities that I see doing the most to undermine education come largely from the neoliberalization of higher ed - the relentless defunding of public ed and shifting of costs to students, in order to pay for tax cuts for the wealthiest; casualization of labor, deprofessionalization, adjunctification, the explosion of class sizes to save costs, etc etc. Those things drive pedagogy in many ways. Most of our writing teachers teach 5 sections of 30 students, and sometimes other sections at community college. They work 70-80 hours a week. Their feedback and assessment practices are driven largely by these material considerations. In such situations poor students, first gen students, students of color, may suffer most as they don't have all the support and familiarity with university that other students do. These kinds of issues seem largely ignored by Inoue, although I imagine he must be aware of them.

    2. I know you are good people. And because I love you, I will be honest with you, and it may hurt. But I promise you, it hurts not because I’ve done something wrong, but because I’m exposing your racial wounds.

      This is a really interesting set of sentences. It echoes the intro to George Yancey's Backlash: What Happens When We Talk Honestly About Racism in America. Yancey's book emerged from his "Dear White America" columns for the NYT, and the ferocious, staggering racism with which these columns were received by some white Americans. The book asks white readers to think honestly and deeply about impulses to deny, ignore, or get angry when racism is discussed. But I have several reactions to Inoue's framing. 1) It could be a way of asking white readers to do the kind of careful introspection Yancey describes.2) it could be a way of framing things that constructs in advance objections or challenges as "hurt" and resentment at hearing Inoue's "honesty," his revealing of racial wounds. It also frames things in terms of love/hate, honesty/dishonesty, wounding/healing. In this scenario, if I disagree with Inoue am I necessarily accusing him of doing something "wrong," or refusing racial healing? This second framing seems to move things away from the space of argument and debate and into some other space. Perhaps that space is valuable in some ways. But this framing carried into the later online discussions of Inoue's text, and was used by some to dismiss critique as refusal to acknowledge racial injury or refusal to acknowledge the reality if racism in the U.S.

    1. Quarterly Journal of Speech Vol. 95, No. 1, February 2009, pp. 43􏰀65 Bordering the Civic Imaginary: Alienization, Fence Logic, and the Minuteman Civil Defense Corps D. Robert DeChaine

      Current figurations of the ‘‘immigration problem’’ in the United States challenge our understanding of the rhetoricity of contemporary bordering practices. The public discourse of the Minuteman Civil Defense Corps serves to chart the alienization of undocumented migrants and the enactment of alien abjection on the U.S.􏰀Mexico border. Alienization promises an antidote to majoritarian anxieties regarding national disunity in the form of a shoring-up of cultural boundaries that border-crossing subjects render troublesome. Ultimately, the fence logic engendered by groups such as the Minutemen reveals how struggles over the boundaries of citizenship both enable and limit an affect-charged civic imaginary.

      Keywords: Borders; Alienization; Minuteman Civil Defense Corps; Citizenship; Social Imaginaries

      The specter of the border haunts the language of social relations. At present, some 12 million undocumented migrants reside temporarily or permanently in the United States.1 Many of them entered the country across the border with Mexico, a frontier spanning nearly 2,000 miles from the Californian Baja through Texas and to Tamaulipas. These border-crossing individuals, the majority of whom venture to the United States in search of work and enhanced economic opportunities, are not recognized by the U.S. government as citizens, defined in legal terms as native or naturalized rights bearers. Nor, for the most part, are they considered legitimate in the broader sense of what Charles Taylor terms ‘‘recognition’’*that is, they are not viewed as valued and respected members of the national community.2 Indeed, upon D. Robert DeChaine is Associate Professor of Communication Studies and Liberal Studies at California State University, Los Angeles. An earlier version of this essay was presented at the 2007 National Communication Association convention in Chicago. The author would like to thank Editor John Louis Lucaites, Mike Willard, Michelle Ladd, Scott Rodriguez, and two anonymous reviewers, each of whom made decisive contributions to the quality of the essay. Correspondence to: Department of Communication Studies, California State University, Los Angeles, 5151 State University Drive, Los Angeles, CA 90032-8111, USA. Email: ddechai@calstatela.edu ISSN 0033-5630 (print)/ISSN 1479-5779 (online) # 2009 National Communication Association DOI: 10.1080/00335630802621078

      44 D. R. DeChaine close examination, the cultural politics of recognition reveals a telling link between state legitimacy and public morality. As Kent Ono and John Sloop note in their analysis of the role of media rhetoric in the passage of California’s Proposition 187 in the 1990s, [T]he contemporary citizenship narrative casts immigration in moral terms: Those who abide by U.S. laws and procedures for how to become U.S. citizens are cast as good and moral citizens; those who do anything but systematically follow expectations of U.S. government officials and their supporters are seen as bad and immoral ‘‘illegals.’’3 The transgressive act of unauthorized border crossing thus produces a double exclusion: it renders migrant persons both legally and morally abject. Given the subordinate status of undocumented migrants in the United States today, what is particularly interesting regarding the ‘‘immigration problem,’’ as it is typically invoked in public discussion, is that it is not framed predominantly in terms of the migrants themselves. After all, the United States is a nation composed of immigrants, so the oft-repeated narrative goes, and its promise as a land of opportunity for hard-working, law-abiding citizens continues to be exalted as among its preeminent gifts. Rather, the problem is most often cast in terms of a lack of border integrity: its leakiness; the ease with which undocumented migrants are able to slip across it unnoticed; and the inability or unwillingness by the U.S. and Mexican governments to seal, secure, and protect the national frontier and its stark line of demarcation. Notwithstanding the nativism and xenophobia that have historically shaped and continue to shape popular attitudes toward undocumented migrants in the United States, the problem of immigration, it seems, lies not with the migrant, but with the border.4 The prevalent characterization of the immigration problem as primarily a concern about the border gestures toward a number of longstanding assumptions regarding what a border is and how it functions. According to the traditional geography-based logic, a border exists as a given entity whose contours can be cleanly and clearly recognized, measured, and mapped.5 A border’s givenness and mappability implies its stability as a resource for delineating spatial territory. Despite its tenacity, the traditional border logic has increasingly been subjected to critique. This scrutiny is owing in part to the emergence and development of postmodern theories of the social character of space beginning in the 1960s, and to a subsequent proliferation of spatial metaphors endemic to human social life in a globalized world.6 Against the traditional view of borders as given and stable entities, the countervailing claim is that whatever form they may assume, all borders are socially motivated constructs.7 They are bounding, ordering apparatuses, whose primary function is to designate, produce, and/or regulate the space of difference. Thus conceived, borders simulta- neously shore up insides and mark off outsides while establishing the terms of their relationality. They perform both division and containment functions, differentiating the self from others, one culture from another, desirable elements from undesirable ones, and, often enough, ‘‘us’’ from ‘‘them.’’ Moreover, according to the revisionist

      account, a border’s contingent configuration is often masked by certain political and economic discourses that labor diligently, sometimes feverishly, to maintain the semblance of stability, integrity, and reality. Recent scholarship in communication studies, chiefly in the area of immigration discourse, has begun to attend to the subject of borders and their cultural significance. A majority of this work aligns with critical scholarship that ascribes a truth function to the border, focusing on its constructedness and on the ambivalent identities of border(ed) subjects.8 Thus considered, a border operates as an inducement to action, deployed by agents in specific contexts to warrant claims to both unity and division.9 Circulating as a robust spatial metaphor, the figure of the border functions as a prevalent organizing doxa in a group’s collective vocabulary. Such assumptions, theorizations, and criticism regarding the constitution and function of the border beckon a considera- tion of two interrelated questions, each of which holds epistemological and ontological significance. What kinds of operations are at play in the construction of borders? And once entwined in the cultural fabric of a political community, how do constructed borders shape human values, attitudes, and actions? The aim here is to focus on the rhetoricity of contemporary bordering practices by charting the ‘‘alienization’’ of undocumented migrants in the United States, and to explicate the operations by which alienizing discourse is enacted on and around the U.S.􏰀Mexico border.10 Alienization, I contend, is a bordering project that draws force from a variety of common linguistic and nonlinguistic resources to render individuals and groups abject and unassimilable*irredeemable others whose putative exclusion from the national body is virtually absolute. Alienization materializes as a reactionary rhetoric goaded by a profound anxiety of incompleteness, an anxiety akin to what Arjun Appadurai refers to as a ‘‘fear of small numbers’’ engendered by a minority population whose alterity is perceived by a majoritarian national ethnos as a threat to its unity.11 Thus conceived, alienization promises an antidote to disunity and incompleteness in the form of a shoring-up of cultural boundaries that border-crossing migrant subjects reveal to be troublesome. Moreover, as a form of world making, alienization shapes a population’s collective attitudes toward and practices of citizenship. I share Robert Asen’s view of citizenship as a discursive mode of public action that is ‘‘always conditioned by social status, relations of power, institutional factors, and material constraints’’; as such, citizenship enactment necessarily involves hegemonic struggles over the very meaning of the term ‘‘citizen’’ in a multipublic sphere.12 Evoking the concept of the ‘‘social imaginary,’’ I examine how struggles over the boundaries of citizenship both enable and limit an affect-charged ‘‘civic imaginary.’’ An analysis of alienization and its bearing on the constitution of the civic imaginary requires a shift in focus away from the consideration of borders as physical, geographical entities to consideration of their instrumentality as performative, sociocultural productions. It places emphasis on the process, practice, and affect of bordering, and on the effects, both material and discursive, of border rhetorics on particular social collectivities. My chief ambition, then, is to throw light on the relationship between physical and social bordering practices, and to identify the very human implications of those practices for the shaping of civic culture. In order to lend historical specificity to the Bordering the Civic Imaginary 45

      46 D. R. DeChaine discussion, I focus attention on the crafting of public appeals in support of the Border Fence Project, an initiative launched by the Minuteman Civil Defense Corps (MCDC) in 2006. Originally created in 2005 by former elementary school teacher, military veteran, and California resident Chris Simcox in response to a perceived ineffective- ness of U.S. governmental action, MCDC proclaims itself to be ‘‘the country’s largest volunteer grassroots border security advocacy group,’’ boasting more than 350,000 constituents.13 The professed aim of the MCDC’s Border Fence Project is ‘‘to secure America’s sovereign territory against incursion, invasion, and terrorism’’ through the construction of a steel security fence along the U.S.􏰀Mexico border.14 The MCDC and its Border Fence Project has garnered considerable attention from popular media, and support from prominent members of the U.S. government. Drawing on a range of textual evidence including websites, speeches, visual and print media, an online discussion forum, and interviews with Simcox and other MCDC members, I show how the group’s public advocacy of the border fence reflects an articulation of economic, racist, and nationalist narratives that together produce a border rhetoric in which the alienized subject becomes both figurally and literally fenced out of the sacrosanct space of U.S. citizenship. The MCDC’s Border Fence Project presents a timely example of a symbolic enactment of alienization that fuels public sentiment and influences policy regarding undocumented migrants in the United States. Rather than dismiss the organization as extremist, as popular U.S. media often do, or consider its views to be peripheral to dominant attitudes toward immigration, I argue that a close examination of the MCDC identifies its reliance on symbolic strategies and commonly shared values that are anything but radical. In the analysis, I hope to illustrate the ease with which physical and geopolitical borders map onto cultural and ideological borders. Against the popular argument that the immigration problem is preeminently about the integrity of the border rather than about the qualities of the migrant as a human being, I contend that their relationship is in fact profoundly fraught. ‘‘Suspect bodies,’’ asserts Lisa Flores, ‘‘carry the border on them.’’15 As a transgressive, racialized subject, the alienized migrant literally embodies the border, rendering problematic any tidy relationship between physical and social space. Crafting the Abject: The Border(ing) Project of Alienization An examination of the rhetorical dimensions of the border requires a shift in focus from borders to bordering, from a consideration of static entities to analysis of a dynamic practice. As a social ordering practice, bordering produces and enforces spaces of identity and difference, defining terms of identification and exclusion. As such, it influences a community’s ways of seeing and experiencing itself, its members, and those deemed to be outside or unworthy of membership. In this section, I outline a project of cultural and political abjection, whose symbolic form operates according to an affective logic of alienization. My intention is to identify the characteristic features of alienization, to explain its motivations and functions, and to suggest some of its implications for its bordered subjects. My discussion is informed by the

      complementary projects of critical rhetoric and cultural studies, each of which claims a stake in uncovering operations of power that shape human experience.16 While I would propose a certain degree of applicability to non-U.S. contexts, my focus is on the specific formation of alienization in post-9/11 U.S. society. Alienization in the United States did not materialize full-blown in the present social-political moment. Indeed, its current manifestation attests to a historical record of laws, policies, and practices reflecting longstanding attitudes about immigration and migrants. As Flores contends, ‘‘Contemporary images of immi- grants, such as that of the illegal alien, do not emerge in a vacuum. Instead, they are part of our nation’s history of immigration, race, and nation; they bring with them varied meanings, reflecting their origins and uses.’’17 This history has been steeped in nativism, an ideology based on a systematic exclusion of designated others, that can be traced back to at least the nineteenth century. Driven by the twin discourses of racism and capitalism, nativism has contributed to the construction of narratives that cast the migrant as culturally different or inferior, a necessary source of labor, and a drain on U.S. government resources.18 Its early manifestations are evidenced in the Chinese Exclusion Act of 1882, which denied migrants citizenship on the basis of national origin, and the Immigration Acts of 1921 and 1924, which placed numerical limits on immigration while entrenching for decades ‘‘a global racial and national hierarchy that favored some immigrants over others.’’19 During the Great Depression, as demand for labor diminished and job competition increased, nativism was reinvigorated in the form of voluntary repatriation campaigns that resulted in the deportation of a half a million Mexican undocumented migrants, U.S. citizens of Mexican descent, and permanent legal residents. During World War II, the U.S. government, presuming that all U.S. citizens of Japanese origin were racially inclined to disloyalty, incarcerated 120,000 persons, effectively nullifying their citizenship status. After the war, with the increased demand for labor, the government instituted the Bracero program, a contract labor policy for Mexican migrants that Mai Ngai describes as ‘‘America’s largest experiment with a ‘guest worker program’’’ to date.20 Arcing into the present, nativism has endured in actions such as 1954’s Operation Wetback, which led to the forcible deportation of more than a million Mexican migrant laborers, efforts in the 1980s to militarize the U.S.􏰀Mexico border, California’s Operation Gatekeeper, Propositions 187 and 209 in the 1990s, state laws mandating English-only policies, and recurring efforts to pass an English Language Amendment to the U.S. Constitution. As such historical patterns of response to migrants underscore, an account of the present conjunctural formation necessarily includes the history of nativist-born attitudes and practices that have designated immigration as a problem and the migrant subject as an undesirable outsider to the American civic community. In its most elemental sense, alienization is a form of otheringa way of seeing and not seeing, of experiencing and not experiencingthat conditions modes of human subjectivity and action. Rather than considering alienization primarily as a condition, or as a terminal state, or in terms of a culmination of effectsalthough it is certainly experienced on each of these registersI propose that it is most usefully understood Bordering the Civic Imaginary 47

      48 D. R. DeChaine as an unstable hegemonic process that must constantly adjust the character of its form if it is to win the consent of a national community guided by properly American liberal democratic values. As a hegemonic process, alienization operates as a fundamentally rhetorical mode of action. Invoking Kenneth Burke’s formulation, it functions terministically to direct and reflect the experiential reality of symbol users.21 As a shaper of collective attitudes, it provides a national community with a repertoire of symbolic resources for naming and thus bringing into being its valuative structure. These resources include an array of metaphors ascribed to alienized persons, as well as linguistic and visual figures that serve both to condense and to amplify public values. As I elaborate below, for example, the metaphoric construction of an alien invasion, and inverted visual images such as an upside-down American flag or a photograph of a breached border fence, can provide powerful symbolic grist for mobilizing public sentiment against those perceived as threatening to the sanctity of American values and national security. Moreover, alienizing rhetoric operates according to an essentialist logic based on negative linguistic difference. As Mark Lawrence McPhail explains, ‘‘The socio-political realities of negative difference are products of a language defined in essentialist terms as much as they are a reflection of self-evident realities that exist independent of the languages we use to construct them.’’22 Viewed in this light, alienization operates as a moralizing discourse that tells the truth about undocumented migrants and the essential nature of their otherness. The terministic and ideological functions of language and imagery are integral to the development of a hegemonic border logic that negatively posits absolute identities onto other(ed) migrant individuals and groups. Alienization signifies a context-specific conjuncture of discourses that interanimate and mutually reinforce one another. In its present configuration, it is enacted as an expression of racial, ethnic, and national identity that categorizes and differentiates Americans from non-Americans*or, more accurately, un-Americans. It demon- strates the intimate social and political linkage of race and nation in directing racism as an expression of xenophobic attitudes toward inferior and undesirable constituents of the national body.23 As a bordering practice, alienization shares much in common with what Howard Winant describes as ‘‘racialization,’’ a process involving attempts to fix identities provisionally in accordance with particular social, historical, and political attitudes about race.24 Furthermore, racializing rhetoric is readily linked with economic arguments that reinforce cultural and political attitudes toward migrants. In their analysis of the rhetoric of California’s Proposition 187, for example, Ono and Sloop describe the racialization of the migrant as the discursive production of an ambivalent subject who is both desired and reviled, necessary for both capitalist production and the object of ‘‘loathing of the laborer who does anything other than work specific jobs associated with facilitating the interests of efficient capital processes.’’25 Arguments about the migrant’s relative economic value are tethered to ideologically charged prescriptions of racial and ethnic identity. The undocu- mented migrant becomes both integral to and separated from proper capitalist relations of production; as such, alienization bears more than a passing resemblance to Karl Marx’s conception of ‘‘alienation.’’26

      Alienization functions discursively to shape public understandings of social and political identities. However, to gauge its influence only in terms of discourse misses a great deal about what drives it as an expression of national communal values. At the motivational level, alienization operates according to an affective logic similar to that which Appadurai describes in terms of an ‘‘anxiety of incompleteness.’’27 Appadurai accounts for what he takes to be an increasing propensity towards ethnic-based violence committed by national majorities (including liberal democracies) against their minority populations, a tendency he attributes in large part to the anxieties of social life in a globalized world: [W]here the lines between us and them may have always, in human history, been blurred at the boundaries and unclear across large spaces and big numbers, globalization exacerbates these uncertainties and produces new incentives for cultural purification as more nations lose the illusion of national economic sovereignty or well-being.... [T]he tip-over into ethnonationalism and even ethnocide in democratic polities has much to do with the strange inner reciprocity of the categories of ‘‘majority’’ and ‘‘minority’’ in liberal social thought, which produces what I call the anxiety of incompleteness. Numerical majorities can become predatory and ethnocidal with regard to small numbers precisely when some minorities (and their small numbers) remind these majorities of the small gap which lies between their condition as majorities and the horizon of an unsullied national whole, a pure and untainted national ethnos . . . . The anxiety of incompleteness (always latent in the project of complete national purity) and the sense of social uncertainty about large-scale ethnoracial categories can produce a runaway form of mutual stimulation, which is the road to genocide.28 According to Appadurai, it is the national ethnic minority populations who bear the brunt of the uncertaintiesand the violencespurred by globalization and its disjunctive modes of organization. Majorities, goaded by the anxiety of incomplete- ness and a fear of small numbers, find a scapegoat in the figure of the minority, whose presence is both necessary and unwelcome: necessary for dirty work such as fighting wars and shouldering menial labor, but unwelcome for the threats their racial, financial, linguistic, and cultural border-blurrings pose. Above all, Appadurai claims, national ethnic minorities ‘‘blur the boundaries of national peoplehood,’’ a cardinal transgression for which they are not to be forgiven.29 They are the embodiment of the anxiety of incompleteness. Minorities problematize the meanings of ‘‘us’’ and ‘‘them’’*and for that, they must pay. Appadurai’s argument regarding the majoritarian fear of small numbers lends to an understanding of the affective logic of alienization in the United States today. In a globalized world, where both ‘‘vertebrate’’ and ‘‘cellular’’ tendencies of hypercapit- alism move across national boundaries with swiftness and ease, it is the figure of the border-crossing migrant who increasingly signifies the social (dis)ease of the U.S. border problem.30 To be sure, arguments casting undocumented migrants as criminals and threats to national security have long figured in U.S. popular, political, and legal discourse. However, public expressions of anxiety regarding the status of migrants have once again become pronounced in the wake of global free trade policies such as those engendered in the General Agreement on Trade and Tariffs Bordering the Civic Imaginary 49

      50 D. R. DeChaine (GATT) and the North American Free Trade Agreement (NAFTA). This has partly to do with the U.S.-led effort to relax economic borders under the auspices of creating an integrated North American market while at the same time resisting an equivalent integration of cross-border labor.31 However, underlying this contradictory policy, I contend, is an affective impulse motivated by fear of losing control. As economic borders loosen, sociocultural borders tighten; as the U.S. economy becomes ever more subject to the disjunctive flows of a global cultural economy, its majoritarian reaction is to allay its anxieties by maintaining control where it canon cultural terrain. In a post-9/11 climate stoked by an omnipresent affect of terrorthe threat of a cellular enemy who is both outside and potentially inside our national borders it is perhaps unsurprising that the population’s fears and uncertainties, as well as its search for enemies, turn inward. Undocumented migrants and their rapidly growing small numbers serve as constant reminders of the incompleteness of a unified, pure American ethnos. Their geographical and cultural border crossings are perceived as challenges, indeed threats, to national sovereignty. Alienization promises an antidote to border anxieties: the present U.S. cultural climate precipitates uneasiness and a compulsion to engage in shoring-up operations, to effect a return to an imagined wholeness, unity, authenticity, and knowability. Thus conceived, alienization is the panicked reaction to the anxiety of incom- pleteness. Coursing through the national body, it casts alienized subjects as abject, inassimilable outsiders to the American community. The abject migrant is not absent from social experienceabjection is not synonymous with absence, as non- recognition is not the antonym of recognition. Rather, because she is both necessary and unwelcome, she is both visible and invisible, both acknowledged and ignored.32 Indeed it is this ambiguous positionality, the troublesome both/and of migrant subjectivity, that compels a national community to search for a means of reconciliation. However, reconciliation entails symbolic violence. In its materializa- tion as a border rhetoric, alienization operates through victimage. Blamed for the ills of a society that proclaims root values of tolerance and pluralism but longs for wholeness, the undocumented migrant fulfills the role of a ‘‘perfect enemy,’’ a vessel for that which the American reviles, disavows, and fears.33 Rhetorical victimage relies on reductive categories and stereotypical modes of representation in its rendering of the subject-scapegoat it designates. Marouf Hasian Jr. and Fernando Delgado note this reductive tendency in their analysis of California’s Proposition 187: ‘‘The typology of illegal immigrant becomes a signifier meaning Mexican (collapsing distinctions among Mexicans, Mexican Americans, Latinos) in a stereotypical manner similar to the social construction of welfare mother as African American female.’’34 Through victimage, the reductive figure of the border-crossing, border-blurring migrant serves as a vessel for containing the threat of an un-American presence from within. As an ideological production that is woven thickly into the fabric of society, alienization is infused in the language and images of everyday communication practices. A reactionary expression of the nativism that has historically shaped the character of U.S. political culture, alienization assumes a more rather than less

      ordinary rhetorical form. Nowhere is its manifestation more apparent than in the invocation of ‘‘illegal immigration,’’ a key figure that is rapidly subsuming the normative rhetoric of immigration in U.S. political culture. Its ubiquity in popular media, town hall meetings, public policy analysis, congressional hearings, and political campaign platforms attests to its ascendance as an orienting devil term. And the potency of illegal immigration is nowhere more concentrated than in the signifier for its idealized subject, the ‘‘illegal alien.’’ The illegal alien is the ideal embodiment of illegal immigration precisely because she is always already doubly stigmatized. Through a process of repetition and sedimentation in public culture, the articulation of the designations ‘‘illegal’’ and ‘‘alien’’ gives them the appearance of a singularity, of being inextricably interlocked. More and more, in fact, ‘‘illegal’’ is becoming an implicit modifier, the already- present threat that marks the alienized subject. To invoke the term ‘‘alien’’ is now nearly all that is required. Her illegality is given. Through the alien’s consolidation as the implicit subject-signifier of illegal immigration, alienization is enacted linguis- tically and performatively as a self-evident expression of the abject other. Moreover, through her tacit link with illegal immigration, the alien is made available for identification with a variety of other(ing) signifiers. These have historically included constructions of the alien as ‘‘criminal,’’ ‘‘communist,’’ ‘‘animal,’’ ‘‘dirt(y),’’ and ‘‘disease(d).’’35 Such signifiers call attention to the ultimately unknowable quality of the alien, an ascription that is threatening to the national community since it belongs to the one who seeps through the cracks, gets in and out, and troubles the very discreteness of the national inside/outside binary. Across all of its identifications and significations, it has become clear, ‘‘Whether invoked directly or indirectly, the figure of the ‘illegal alien’ is hauntingly consistent.’’36 Indeed, the totalizing ascription of ‘‘illegal alien’’ to the alienized subject performs an essentializing function that literally denies the possibility of a positive referent. Given her overwhelmingly negative social-cultural status, redemption of the alienized subject is unlikely in the United States today. In order to be refigured as a potential member of the American community, she would need to undergo a radical transformation, entailing both avowed and ascribed identity reformation. In effect, the alien would have to become an altogether different person. It is true that there are legal pathways to U.S. citizenship, although in rapidly restricting forms.37 However, the fact that alienization involves concomitant racialization makes the prospect of redemption difficult, if not doubtful. Not only do border guards check papers, but since the alienized subject carries the border on her back, she is constantly subject to surveillance and search. She may or may not be what she seems; although she is among the community, although she may be naturalized, she is not naturally of the community. As Bhikhu Parekh astutely notes in his discussion of multicultural citizenship, ‘‘Although equal citizenship is essential to fostering a common sense of belonging, it is not enough. Citizenship is about status and rights; belonging is about acceptance, feeling welcome, a sense of identification. The two do not necessarily coincide.’’38 Bordering the Civic Imaginary 51

      52 D. R. DeChaine Alienization, I have suggested, is a border(ing) rhetoric par excellence. It operates in U.S. society as a hegemonic project concerned with the forging and maintaining of dominant American civic values. At the structural level, it signifies an alliance of racist, capitalist, and nationalist discourses that materializes rhetorically. At the motivational level, it is goaded by majoritarian anxieties of national incompleteness and a longing for civic communion. Although reactionary in tone, it reflects widely shared values and attitudes. Dogged by the threat of an ambiguous enemy within, the primary labor of alienizing rhetoric is the production of the abject subject, the illegal alien, the embodiment of that which is both necessary and unwelcome in the space of U.S. citizenship. Reviled for her border-crossing and border-blurring behavior, the alien is constructed through symbolic acts of reduction and victimage. With her identity tethered to the negative, racialized figure of illegal immigration, the alien’s prospects for redemption are acutely diminished. Above all, the rhetorical production of the alien illustrates the collective desire for purity, perfection, and order*a compulsion, as Burke insists, that is a hallmark of the human condition.39 Put differently, although alienization appears primarily to be about the imputed qualities of the undocumented migrant, it is also substantially about the fraught logic of the border and the symbolic power that it wields. Pledging ‘‘Eternal Vigilance’’: Rhetorical Alienization in the Minuteman Civil Defense Corps Since its inception under the leadership of its founder Chris Simcox, the Minuteman Civil Defense Corps has been effective in shaping public attitudes about the immigration problem in the United States. Proclaiming itself as ‘‘one of the most important, socially responsible, and peaceful movements for justice since the civil rights movement of the 1960s,’’ Simcox and the MCDC have garnered support from a number of legislators, including Republican representatives Duncan Hunter of California and Tom Tancredo of Colorado.40 Like other contemporary social movement groups, the MCDC is acutely attuned to the politics of the public screen and its power to mobilize popular attitudes.41 In order to promote its mission and its border operations, the organization relies heavily on hypermedia for the dissemina- tion of information, public statements, speeches, fundraising campaigns, and volunteer musters. Its official and unofficial discourse proliferates in the blogosphere, and postings of speeches by Simcox as well as protests against him can be found on popular video sites such as YouTube. Its continued media presence and its often infamous appearances on college campuses have helped the MCDC to maintain a significant profile in the public conversation on immigration reform, while contributing to its ethos as a controversial activist group. A substantial amount of the MCDC’s public advocacy has focused on its effort to secure funding for the construction of approximately 70 miles of fencing along portions of the U.S.􏰀Mexico border in southeastern Arizona. The MCDC has dubbed the initiative the Minuteman Border Fence Project, a campaign that serves as an umbrella for a number of regional fundraising efforts.42 Professing to be ‘‘doing the

      job the [U.S.] President and the Senate refuse to do,’’ MCDC-aligned volunteers began constructing bits of the fence themselves in October 2006.43 Referring to the proposed border fence as ‘‘America’s ultimate protection against foreign invasion and terrorism,’’ the MCDC capitalizes on the historical symbolism of the Minutemen of the American Revolution, describing its undertaking as not a solution but an example of what committed AmericansWe the Peoplemust do in order to safeguard the border and the citizenry.44 Through its efforts to rally public support for a security fence along the U.S.􏰀Mexico border, particularly in its crafting of appeals that draw on affect-charged language and images of formative American values, the border(ing) rhetoric of the MCDC demonstrates how public under- standings of national identity, community, citizenship, and ‘‘the other’’ are mutually constructed. Reflecting commonly shared beliefs about migrants and immigration in U.S. political culture, its appeals bespeak a ‘‘fence logic’’ that effectively displaces claims about alien subjectivity and refigures them as claims about conditions said to result from nonsecure U.S. borders. In this way, the group enacts a project of alienization that avoids overt racism by framing the immigration problem as a concern about geographical borders rather than about the alien per se. In all, the MCDC contributes to the production of an anxiety-ridden narrative of citizenship of what it means to be a member of the broadly imagined American Community. Ultimately, its fence logic both shapes and reflects broadly held attitudes about culture and difference in contemporary U.S. society. The MCDC demonstrates its similarity to other social-political movement groups insofar as its public appeals, if they are to be acted on, must successfully construct an orientation toward a situationa compelling exigency, a plausible accounting of involved actors, and a fitting and attainable mode of redress. What most distinguishes the MCDC from other activist groups is its particular crafting of a vision of national community, an understanding of civic identity that seeks resonance with a dominant regime of true American values. In constructing appeals that articulate values of social unity, national security, rule of law, and civic responsibility, the MCDC draws from a common vocabulary of U.S. political culture to advance an alienizing logic that shores up communal boundaries while simultaneously defining terms of exclusion. The MCDC’s casting of the rhetorical situation turns on its characterizations of the duty-bound Minuteman volunteer and the nature of the national security nightmare at the U.S.􏰀Mexico border that impels him to action.45 The United States, the group warns, is a nation in peril. The threat strikes at the heart of the American democratic ethos: the Border Fence website asserts that the country ‘‘is under siege by forces and interests that have the capacity, over time, to destroy our great experiment of responsible self-government.’’46 The sovereignty of the nation and the unity of the American people have become jeopardized as a result of those whose unauthorized movements impinge on them. The danger, the MCDC insists, issues from the current state of unsecured U.S. borders, giving rise to a ‘‘human tsunami’’ of illegal border crossers and the threats they pose to citizen safety, economic stability, and cherished American values.47 Bordering the Civic Imaginary 53

      54 D. R. DeChaine The forces and interests that are claimed to undermine American unity and sovereignty are both internal and external. In an address entitled ‘‘A Letter to My Fellow Americans,’’ Jim Wood, Executive Director of the Border Fence Project, cautions, ‘‘[T]he national language, character and culture of our sovereign American people are under attack by ‘multi-culturalism’ run rampant. It’s dangerous and it’s destructive.’’48 Employing violent metaphors and reductive categories to consolidate the gravity of the alien threat, constituents of the MCDC make repeated reference to the imputed ‘‘illegal invasion’’ now said to be underway.49 Such an evocation points to the danger to the American ethnos of an abstract, massified enemy already among us, ‘‘the illegal aliens residing in our midst who have been allowed by a feckless government to violate our laws and mock our sovereignty as a nation.’’50 The fear of increasing numbers of alien noncitizens in the United States and their ability to undermine American unity is further evidenced on the Border Fence Project’s website, which cites California as an example of the disunity produced by an ‘‘increased flux of illegals, who displace legal citizens whose numbers are decreasing there.’’ Moving unchecked across the U.S. border, the alien-enemy is now within, infecting the body politic. It corrupts the government, disunifies its citizenry, dilutes its culture, and precipitates a virulent strain of anti-Americanism: as Simcox stated, in response to May 1, 2007 demonstrations in which ‘‘anti-American extremist militants’’ flew upside-down U.S. flags and flew Mexican flags above the U.S. flag, I think it exposes an anti-American, anti-European sentiment. And we see that on college campuses across the country. The vitriol, the hate. The hate groups that exist on college campuses and then take to the streets are calling for a reconquista of the United States.51 Flags and other visual imagery are routinely employed by the MCDC to dramatize the anti-Americanism fueled by the alien invasion. For example, an image on the Border Fence Project website titled ‘‘Tear for My Country’’ features a close-up photograph of a portion of a human face, eye cast downward, with a tear rolling down its cheek. In the foreground flies a Mexican flag, positioned on its mast above an upside-down American flag. Superimposed at the bottom of the image is an iconic rendering of Uncle Sam (unaccompanied by its familiar ‘‘I Want You!’’ caption) pointing at the viewer, as if to admonish her to take up the pro-American resistance. In public appearances, Simcox and other MCDC members also often display photographs of anti-American migrant protesters, offering them as evidence of an enemy in our midst whose force, like that of a virulent disease or inexorable flood, must be contained. The destructive forces that threaten American unity, sovereignty, and democracy are the byproduct of more ominous threats to national security and public safety that an unsecured U.S. border represents. The MCDC invokes national security as a caution to Americans about the dangers of terrorism, criminality, and violence posed by an illegal invasion across broken borders. The alien-terrorist menace is real. ‘‘We’re being attacked by people from a foreign country,’’ exclaims Simcox; ‘‘Is that not

      Bordering the Civic Imaginary 55 terrorism in its own right?’’52 Those who threaten national security, whether wittingly or unwittingly, are culpable: ‘‘If you’re breaking into this country when this country is at war, then you’re a potential enemy of this country, and you should be treated accordingly.’’53 In MCDC discourse, a link between illegal immigration and ‘‘terrorism,’’ another prominent devil term in post-9/11 U.S. society, is forged through an association of the border crosser with crime and violence. Simcox, emphasizing this association, makes reference to ‘‘the violence caused daily by illegal entrants and often violent alien intruders,’’ and advocates ‘‘the feasibility and efficacy of fencing to secure America’s borders from illegal incursion by aliens and international criminal cartels.’’54 And the alien-borne violence is growing.55 As the number of unauthorized border crossings increases, ‘‘More gang violence erupts in our cities by murderous thugs such as MS-13, the ruthless gang of self-professed friends of al Qaeda.’’56 The deadly admixture of crime, violence, and terrorism at the hands of undocumented border crossers is perhaps most dramatically asserted by the Border Fence Project: ‘‘Because illegal aliens murder 5,000 innocent Americans every year and we take a trillion dollar hit overall to our economy, illegal immigration rewards us with a 9/11 or worse every year.’’57 Close-up photographs of decrepit and hole- ridden fencing along the U.S.􏰀Mexico border, and images of aliens individually and collectively breaching the fence, are offered by the MCDC as evidence of the national security nightmare that is claimed to exist. A Minuteman Border Cam with multiple views of the border fence is featured on the MCDC website, so that viewers may witness for themselves the always-looming threat to national security in real time. By playing on national anxieties regarding a terrorist threat both beyond and within U.S. borders, the MCDC collapses categorical distinctions between terrorism, criminality, and alien subjectivity, drawing an abstract figure of the dangerous, already suspect un-American other. The MCDC readily identifies the U.S. government as the primary agency responsible for the immigration problem. Although it is the alien who crosses the border into the United States, she is rarely directly named as the villain. Rather, the group insists, it is the U.S. government (and to a lesser extent, the Mexican government) and its lax attitude toward border enforcement that allows aliens to cross the border. The government’s negligence in upholding its commitment to safeguard the border has ominous consequences. As Simcox fervently warns, The existing border crisis is a dereliction of duty by those entrusted with American security and sovereignty, leaving America vulnerable to terrorist infiltration and an unprecedented crime wave caused by drug smugglers, rapists, thieves, human traffickers and murderers who currently cross our borders at will.58 In its attribution of the problem, the MCDC fuses the values of national security and the rule of law, imputing a causal relationship between lax border enforcement, a permissive welfare state, and the economic and social burdens shouldered by law- abiding U.S. citizens. Placing emphasis on this causal relationship, Simcox calls on fence logic to offer a remedy:

      56 D. R. DeChaine When you secure the borders, cut off welfare and social services to illegal aliens at the expense of the American taxpayer, and strictly enforce the laws against hiring illegally, the problem of what to do with the estimated 12-20 million illegal aliens already in this country will drastically shrink in size and scope.59 Political, economic, and social reform such as that proposed by Simcox and the MCDC requires a strong, morally committed U.S. government. Simcox warns, In a time of war, the future of the American republic hangs in the balance . . . . The confidence of we the people can only be restored by those elected officials who will truly act in defense of Americaher liberty, sovereignty, security and prosperity in the face of all foreign threats.60 Taken at face value, the MCDC’s assignation of blame to the U.S. government would seem to belie the main thrust of alienization, the chief operation of which is the rendering of the abject alien subject. However, the displacement of the alien and her deflection away from being the primary agent of blame is key to the crafting of the group’s persuasive appeals. For all its drama and hyperbole, the MCDC’s alienizing, nativist rhetoric appears on its surface to be relatively devoid of overt racism and open hostility toward migrants as persons. Notwithstanding the xenophobic tenor of its appeals, one is hard pressed to locate explicit epithets directed against migrants in its official discourse. It is tempting to attribute this lack of directness to what Stuart Hall terms ‘‘inferential racism,’’ those ‘‘apparently naturalised representations of events and situations related to race, whether ‘factual’ or ‘fictional’, which have racist premises and propositions inscribed in them as a set of unquestioned assumptions.’’61 As its narrative makes clear, inferential racism infuses the group’s language and visual imagery. Be that as it may, the indirectness of the MCDC’s alienizing project is also reflective of its formal construction, which effectively displaces an alien-centered threat and refigures illegal immigration as a border-centered threat. Alienizing rhetoric does not so much actively ascribe an identity quality to the alien subject as to passively describe that which is said to result from the condition of unsecured borders. As an effect of this formal refiguration, ‘‘invader’’ is reinscribed as ‘‘invasion’’; rather than an agent-centered claim that aliens are invading our nation, it is more common in MCDC discourse to encounter condition-centered statements such as ‘‘Our borders are under attack’’ or ‘‘Our borders are being invaded.’’ Likewise, agent-centered references to aliens as terrorists or criminals are more often refigured as statements referring to the threat of terrorism and criminal activity*both claimed to be effects of the condition of unsecured borders. Functioning as the rhetorical form of inferential racism, alienization depersonalizes (and dehumanizes) the alien subject while providing the rhetor, whether an individual or an organization, with an alibi. By establishing a socially acceptable position for anti-migrant sentiment, one that places blame not on a person or an ethnic group but on an impersonal condition, the formal construction of alienization provides an inoculation against charges of racism and scapegoating. Thus conceived, rhetorical alienization allows the MCDC and other alienizers to proclaim unself-

      consciously that broken borders, not aliens, are the source of the immigration problem. As the MCDC’s account of the exigency works to underscore, the dire situation on the U.S.􏰀Mexican border demands immediate and decisive action. Against the corrupting forces of an alien invasion and a complicit U.S. government, the group calls on true Americans ‘‘to safeguard our beloved nation from violence, from sedition, from wholesale attack by those who do not love America and wish to see her destroyed or irremediably altered from a nation of liberty, equality, and justice, and a bastion of western civilization.’’62 Such an intervention requires selfless action, honor, respect for tradition, and love of country: moral qualities embodied in the figure of the MCDC citizen volunteer. Deriving ethos from the venerable narrative of the American Revolutionary Minuteman, the MCDC citizen volunteer exemplifies the values of a national polity based on the rule of law and civic responsibility. As Article Four of the Minuteman Pledge attests, ‘‘[a] Minuteman believes in a strong, safe and secure America that begins with borders open only to those who have a legal right to enter, and who have met all the lawful criteria to cross into our territory established by the sovereign American people.’’63 In its appeal to the U.S. citizenry to secure its borders, the MCDC extols individual and collective responsibility, cementing together American virtues of patriotism and duty in taking action against present and future threats of an alien invasion. The U.S. government’s dereliction of duty must be met by the force of duty-bound American citizens*patriotic Minutemen who ‘‘will not sit idly by while we are colonized by another country.’’64 As the Minuteman Border Fence Project proclaims, ‘‘The politicians will talk; the liberal media and the ‘Hate America’ crowd will offer AMNESTY to law breakers. The Minutemen will ACT in defense of America!’’65 The appeal to American responsibility is future oriented: in the fight to secure U.S. borders against the alien invasion, the ‘‘future security and public tranquility for generations of our American posterity’’ hangs in the balance.66 Looking toward the future well-being of the country, the MCDC citizen pledges his commitment to responsibility as an individual, consecrating it in a vow before God and his fellow Americans. Invoking a phrase popularized by Wendell Phillips in a speech to American abolitionists, the Minuteman declares, ‘‘‘Eternal vigilance is the price of liberty . . . ’ And so I will stand watch on America’s borders and in her sovereign interest until relieved from duty by my fellow countrymen.’’67 As movement-oriented action, this commitment to responsibility is not simply individual; it is also collective. The MCDC pledges eternal vigilance, promising to ‘‘continue to stand watch at the border and report illegal activity, build border fencing, urge local and federal officials to enforce the law and push for the enforcement of our laws to keep our country and your families and children safe.’’68 Several Border Fence Project websites feature photographs of groups of MCDC citizen-patriots building, repairing, and guarding the border fence, offering a vivid demonstration of the organization’s collective commitment to civic duty and responsibility. The theme of eternal vigilance recurs Bordering the Civic Imaginary 57

      58 D. R. DeChaine regularly in MCDC discourse and serves to codify its sacred pledge to the American people. On the California border, an MCDC-run outpost named Camp Vigilance serves as a base for the group’s semi-annual musters, from whence fence-building teams are organized and deployed. Additionally, the MCDC offers an opportunity to enact eternal vigilance virtually. On joining its ‘‘Become a Cyber Minuteman!’’ campaign, those owning property on the U.S.􏰀Mexico border are provided with motion-sensing surveillance cameras that down-stream video directly to MCDC representatives, local police departments, and U.S. Border Patrol agents.69 What is apparent from this brief examination of the MCDC’s rhetoric is that its impassioned call for greater U.S. border security bespeaks an intense border insecurity. Relying on common tropes of American identity in the crafting of its appeals, the group’s rhetoric reveals a stock of motivations consonant with broadly shared majoritarian anxieties. Through hyperbolic appeals to the oblique yet powerful values of social unity, national security, rule of law, and civic responsibility, the group crafts a vivid human drama in which modern Minutemen take up their historical calling to pledge eternal vigilance, dutifully safeguarding American borders from the threat that lies beyondand, increasingly, within. The current corruption of the U.S. government, its political process, its economy, and its cultural character provides proof that the danger posed by millions of necessary but unwelcome border- crossing aliens is real. The alien invasion strikes at the very heart of national civic identity, threatening its purity. Harboring terror both known and unknown, the invasion must be stopped; the future safety of American citizens and the sanctity of American values are at stake. The fear of small but increasing numbers of illegal aliens combines with anxieties of national disunity and a federal government complicit in a hegemonic project of colonization. Only a counter-hegemonic movement led by a unified corps of committed American patriots can hope to stop the invasion, fence the border, protect national sovereignty, and illuminate a path toward civic restoration. As its appeals make clear, the MCDC relies on a fence logic that underwrites its construction of the immigration problem. In characterizing the threat of an alien invasion as preeminently an issue of border security, the MCDC contributes to an essentializing epistemology of the border. In its public discourse, national borders are static and given; their realness is never in question. The implications of such an epistemology and the worldview it constructs are not only political but profoundly ethical. Fence logic works to (re)inscribe the naturalness of geographical borders, positing the alien as a subject who is naturally out of place. Border security is thus framed as being about legal enforcement, not about wielding power and control. As Joseph Nevins explains, [T]he ‘‘illegal’’ is someone who is officially out of placein a space where he does not belong. Thus, the official relationship of the ‘‘illegal alien’’ to the particular national space in which he finds himself defines his status. The practice of territorialitythe effort to exert influence over people and/or other phenomena by asserting control over a defined geographic areareinforces the designation of the ‘‘illegal.’’ Territoriality helps to obfuscate social relations between controlled and

      Bordering the Civic Imaginary 59 controller by ascribing these relations to territory, and thus away from human agency.70 It is not only geographical borders that are naturalized. As a territorializing apparatus, the MCDC’s fence logic also works to (re)inscribe the social-cultural borders as natural. Defining clear and self-evident lines between American citizen and alien invader, as the MCDC does, reveals the ease with which psychic territories and their cultural investments map onto and reinforce physical territories and their geopolitical investments. More importantly, the us/them binary that such bordering practices work to cement into place all but guarantees an irredeemable non-place for the racialized, alienized, border-crossing migrant in the United States today. Ultimately, as the MCDC’s rhetoric illustrates, the naturalization of borders reflects an affect-charged, ideological operation. It provides an anxiety-ridden American ‘‘we’’ with terms for ordera common-sense orientation for gauging the truth about people, places, social statuses, and communal allegiances. Bordering the Civic Imaginary I have argued for the centrality of the figure of the border in contemporary rhetorical culture. In so doing, I have advanced an account of political and cultural abjection that prompts a scholarly reorientation away from a static concept of borders toward a dynamic conception of bordering practices. Proposing alienization as the name for a racializing project that produces an abject migrant subject, I have focused attention on the articulation of discourse and affect and its labor across geopolitical and sociocultural space to subjugate the border-crossing migrant. In particular, I have aimed to convey a sense of the panicked character of alienizing rhetoric, and, through the example of the Minuteman Civil Defense Corps, to illustrate the enduring tenacity of fence logic in a globalized world. Although reactionary in tone, the symbolic strategies that the MCDC uses to craft its appeals, and the attitudes it expresses, are far from radical. The liberal democratic values propounded by the groupunity, national security, the rule of law, patriotism, and responsibilityand the ordinariness of the language and images it mobilizes to advance its cause demonstrate the group’s close affinities with the tradition of U.S. social justice movements. As such, its narrative offers a cautionary tale about the all too easy slide from communal appeals based on majoritarian values to violent ethnonationalist exhortations. Writ large, the fears and anxieties that spur the MCDC’s alienizing practices signal the increasing friction between the denationalizing tendencies of economic and cultural globalization and a redoubling of desire for a singular national community, purity, and civic identitya condition betraying a long history of racial, ethnic, socioeconomic, and gender exclusions in the United States. The alienization enacted by the MCDC and other adherents of fence logic is goaded by an overriding fear of losing control: this fear itself is based on a fictional premise*that the American community (and its putative boundaries of identity and citizenship) was ever in control, secure in a state of order, sovereignty, and wholeness. ‘‘Borders,’’ as Anne

      60 D. R. DeChaine Demo reminds, ‘‘function as an index of sovereignty because their very presence (real or imagined) symbolizes claims of authority over a territorial entity.’’71 In this sense, border(ing) rhetorics serve as crucial sites for investigating intersections of community, territory, identity, and power in contemporary society. As the analysis illustrates, rhetorical renderings of the border are instrumental in the crafting of American civic community. In his discussion of borders and power relations, David Newman argues, [T]he stronger the barrier function of the border, the more powerful the imagined, the more abstract the narrative of what is perceived as lying on the other side. Perceptions of borders usually focus on what exists on the other ‘‘invisible’’ side of the line of separation. Borders exist in our mind by virtue of the fear we have of the unknown of the ‘‘there’’ and which, in turn, causes us to stay on our side of the border in the ‘‘here.’’72 Newman’s emphasis on the correlation between the fear of the unknown, the perceived integrity of borders, and the mystification of the other speaks to the current hold that fear has on U.S. civic culture. The oxymoronic alien subjectshe who is both known and unknown, simultaneously inhabiting the ‘‘here’’ and ‘‘there’’ of the national bodyincites a redoubling of political and moral boundaries in an era of an omnipresent war on terror. Burke’s dictum that ‘‘[a] way of seeing is always a way of not seeing’’ aptly characterizes the situation of the always already suspect border- crossing migrant.73 The intensifying compulsion to deal with the immigration problem, a defining exigency of post-9/11 America, underscores the terministic power of alienizing rhetoric to direct and reflect the social reality of the abject alien noncitizen. Put another way: as alienization is enacted, citizenship is enacted. Significantly, the fears and anxieties foregrounded in this discussion also attest to the force of the imagination as an integral agency in human social life.74 The construction of modern social imaginaries, according to Taylor, involves ‘‘the ways people imagine their social existence, how they fit together with others, how things go on between them and their fellows, the expectations that are normally met, and the deeper normative notions and images that underlie these expectations.’’75 Extending Taylor’s definition, my analysis of the excision of the alienized subject from the space of U.S. citizenship lends credence to an equally powerful notion of the civic imaginary as a social horizon for defining the constituents and limits of civic culture. A contested space for adjudicating conditions of citizen identity and inclusion, the civic imaginary is a contentious production with material consequences for those who deliberately or incidentally occupy it. Both real and imagined, performative and affective, it is a consummately rhetorical space where culture and politics converge, identity is shaped, and power is wielded. An adjunct of the public sphere, it proscribes conditions for citizenship enactment and the voices that are to be included in and excluded from deliberation.76 More than anything, the space of the civic imaginary serves as an ethical horizon for the articulation of the citizen as an embodiment of personhood. For alienizers like the MCDC, civic personhood reduces the border-crossing migrant to a racialized juridical subject. Crucially lacking in such a formulation is consideration of the border-crossing migrant as a human being

      worthy of recognition, respect, and dignity. For this alone, the present construction of the American civic imaginary warrants concerted critical attention. Alienization continues apace in the United States today, with real implications for those alienized. In 2006, approximately 222,000 undocumented migrants were removed from the United States, roughly a 20 percent increase from the previous year.77 Moreover, as Ngai notes, ‘‘In the aftermath of the terrorist attacks on the United States on September 11, 2001, the Department of Justice used immigration laws to arrest and detain over 1,100 aliens, many without charge and in secret.’’78 Despite resistance in Congress and protests by numerous city mayors and property owners, plans to complete a security fence along the U.S.􏰀Mexico border have been recently stepped up by way of a presidential mandate granting Michael Chertoff, Secretary of the U.S. Department of Homeland Security, supra-constitutional authority to build it. Current U.S. immigration policies and the social attitudes that shape them do more than affect individuals’ legal status; they tear apart families, deny basic services to those in need, and create conditions whereby growing numbers of the U.S. population are demonized as necessary but unwelcome nonpersons. Such attitudes and conditions, as Dana Cloud has convincingly argued, are not new. Indeed, they are entirely congruent with the naturalizing border epistemology of Samuel Huntington’s ‘‘clash of civilizations’’ thesis and its ascendant hegemony.79 What is novel, I have claimed, is the discursive-affective alliance that shapes its present rhetorical form. The ordinariness of alienization as a mode of citizenship enactment beckons scholars to attend closely to the ways in which alienizing rhetoric works its way into the public vernacular. As I have hoped to show, its conception of the American citizen reveals a profoundly immoral discourse that excludes, racializes, and otherizes individuals and groups*a discourse all too readily conscripted for the cause of national unity in troubled times. A vibrant civic imaginary requires an understanding of personhood that moves beyond questions of legal status to include recognition and respect as formative constituents of citizenship. In the name of such a counter- hegemonic project, it is the debunking of alienizing practices that is urgently needed today. This does not mean substituting border logic for a logic premised on a ‘‘world without borders,’’ as some have suggested; to unborder, after all, is still a bordering practice.80 Rather, it entails a collective effort to recognize, make public, and work against alienization and its material and symbolic violence. Such a project, I have insisted, begins by taking borders seriously. In his meditation on the ‘‘unreflective nationalism’’ that an uncritical view of borders is currently fomenting in U.S. civic culture, Robert Chang observes, ‘‘Although the border is everywhere, your perspective may render it invisible. It is through this invisibility that the border gains much of its power.’’81 Working to unmask this invisibility will be vital to a project that aspires to broaden rather than border the horizon of the civic imaginary. Bordering the Civic Imaginary 61

      62 D. R. DeChaine Notes [1] Jeffrey S. Passel, ‘‘The Size and Characteristics of the Unauthorized Migrant Population in the U.S.,’’ Pew Hispanic Center, March 7, 2006, http://pewhispanic.org/files/reports/61.pdf/. It must be acknowledged that this is a contested statistic. My choice of ‘‘migrant’’ to characterize undocumented persons in the United States reflects my intention to leave open the variety of statuses, motivations, and modes of mobility of border-crossing individuals. For a sympathetic discussion of the term, see Mae M. Ngai, Impossible Subjects: Illegal Aliens and the Making of Modern America (Princeton: Princeton University Press, 2004), xix􏰀xx. [2] Charles Taylor, ‘‘The Politics of Recognition,’’ in Multiculturalism and ‘‘The Politics of Recognition,’’ ed. Amy Gutmann (Princeton: Princeton University Press, 1992), 25􏰀73. [3] Kent A. Ono and John M. Sloop, Shifting Borders: Rhetoric, Immigration, and California’s Proposition 187 (Philadelphia: Temple University Press, 2002), 26. [4] Ronald Walter Greene, ‘‘Malthusian World(s): Globalization, Race, and the American Imaginary in the Immigration Debates of the Twentieth Century,’’ in Argumentation and Values: Proceedings of the Ninth SCA/AFA Conference on Argumentation, ed. Sally Jackson (Annandale, VA: Speech Communication Association, 1995), 191􏰀95. See also Marouf Hasian Jr. and Fernando Delgado, ‘‘The Trials and Tribulations of Racialized Critical Rhetorical Theory: Understanding the Rhetorical Ambiguities of Proposition 187,’’ Communication Theory 8 (1998): 245􏰀70; Justin Akers Chaco ́n and Mike Davis, No One Is Illegal: Fighting Violence and State Repression on the U.S.􏰀Mexico Border (Chicago: Haymarket Books, 2006). [5] Michael Curry, ‘‘On Space and Spatial Practice in Contemporary Geography,’’ in Concepts in Human Geography, ed. Carville Earle, Kent Mathewson, and Martin Kenzer (Lanham, MD: Rowman and Littlefield, 1995), 3􏰀32; Henk Van Houtum, ‘‘The Geopolitics of Borders and Boundaries,’’ Geopolitics 10 (2005): 672􏰀79. [6] D. Robert DeChaine, ‘‘Imagined Immunities: Border Rhetorics and the Ethos of Sans Frontie`risme,’’ in Interdisciplinarity and Social Justice: Revisioning Academic Accountability, ed. Ranu Samantrai, Joe Parker, and Mary Romero (New York: State University of New York Press, in press). [7] Phil Hubbard, Rob Kitchin, Brendan Bartley, and Duncan Fuller, Thinking Geographically: Space, Theory and Contemporary Human Geography (New York: Continuum, 2002), 33; Joe Moran, Interdisciplinarity (New York: Routledge, 2002), 165. [8] On the rhetorical constructedness of borders, see Lisa A. Flores, ‘‘Constructing Rhetorical Borders: Peons, Illegal Aliens, and Competing Narratives of Immigration,’’ Critical Studies in Media Communication 20 (2003): 362􏰀87; D. Robert DeChaine, Global Humanitarianism: NGOs and the Crafting of Community (Lanham, MD: Lexington Books, 2005). On the ambivalent character of border(ed) subjects, see Gloria Anzaldu ́ a, Borderlands/La Frontera: The New Mestiza (San Francisco: Aunt Lute, 1987); Pablo Vila, Crossing Borders, Reinforcing Borders: Social Categories, Metaphors, and Narrative Identities on the U.S.􏰀Mexico Frontier (Austin: University of Texas Press, 2000). [9] Aimee Carrillo Rowe, ‘‘Whose ‘America’? The Politics of Rhetoric and Space in the Formation of U.S. Nationalism,’’ Radical History Review 89 (2004): 119􏰀20. [10] I borrow the term ‘‘alienization’’ from Joseph Nevins, Operation Gatekeeper: The Rise of the ‘‘Illegal Alien’’ and the Making of the U.S.􏰀Mexico Boundary (New York: Routledge, 2002), 143. [11] Arjun Appadurai, Fear of Small Numbers: An Essay on the Geography of Anger (Durham, NC: Duke University Press, 2006), 51􏰀52. [12] Robert Asen, ‘‘A Discourse Theory of Citizenship,’’ Quarterly Journal of Speech 90 (2004): 204. [13] Minuteman Civil Defense Corps, ‘‘Minuteman Civil Defense Corps Replaces Volunteer Administrators,’’ http://minutemanhq.com/hq/print.php?sid􏰁342/.

      Bordering the Civic Imaginary 63 [14] Minuteman PAC, http://www.minutemanpac.com/about.php/. [15] Flores, ‘‘Constructing Rhetorical Borders,’’ 381. See also Robert S. Chang and Keith Aoki, ‘‘Centering the Immigrant in the Inter/National Imagination,’’ California Law Review 85 (1997): 1397; Linda Bosniak, The Citizen and the Alien: Dilemmas of Contemporary Membership (Princeton: Princeton University Press, 2006). [16] On the complementarity of critical rhetoric and cultural studies, see Maurice Charland, ‘‘Rehabilitating Rhetoric: Confronting Blindspots in Discourse and Social Theory,’’ Communication 11 (1990): 253􏰀64; Thomas Rosteck, ed., At the Intersection: Cultural Studies and Rhetorical Studies, Revisioning Rhetoric: A Guilford Series (New York: Guilford Press, 1999), 1􏰀23. [17] Flores, ‘‘Constructing Rhetorical Borders,’’ 363. [18] Pierrette Hondagneu-Sotelo, ‘‘Unpacking 187: Targeting Mejicanas,’’ in Immigration and Ethnic Communities: A Focus on Latinos, ed. Refugio I. Roch ́ın (East Lansing: Julian Samora Research Institute, Michigan State University, 1996), 93. [19] Ngai, Impossible Subjects, 3. [20] Ngai, Impossible Subjects, 129. [21] Kenneth Burke, Language as Symbolic Action: Essays on Life, Literature, and Method (Berkeley: University of California Press, 1966), 44􏰀62. [22] Mark Lawrence McPhail, The Rhetoric of Racism Revisited: Reparations or Separation? (Lanham, MD: Rowman and Littlefield, 2002), 66. [23] Hemant Shah, ‘‘Race, Nation, and Citizenship: Asian Indians and the Idea of Whiteness in the U.S. Press, 1906􏰀1923,’’ Howard Journal of Communications 10 (1999): 251. [24] Howard Winant, Racial Conditions: Politics, Theory, Comparisons (Minneapolis: University of Minnesota Press, 1994), 59. [25] Ono and Sloop, Shifting Borders, 27. [26] See, for example, Marx’s discussion of alienation in ‘‘Economic and Philosophical Manuscripts,’’ in Karl Marx: Early Writings, trans. Rodney Livingstone and Gregor Benton (London: Penguin Books, 1975), 279􏰀400. [27] Appadurai, Fear of Small Numbers, 8􏰀10. [28] Appadurai, Fear of Small Numbers, 7􏰀9. [29] Appadurai, Fear of Small Numbers, 45. [30] Appadurai, Fear of Small Numbers, 21􏰀31. [31] Douglas S. Massey, ‘‘Understanding America’s Immigration ‘Crisis’,’’ Proceedings of the American Philosophical Society 151 (2007): 309􏰀27. [32] It is worth adding that women and men are often differently alienized. As some scholars have noted, the meaning of ‘‘alien’’ is itself gendered, shifting its connotation according to particular sociohistorical exigencies. Hondagneu-Sotelo, for example, notes, ‘‘Contemporary xenophobia targets women and children because it is they who are central to making settlement happen’’ (‘‘Unpacking 187,’’ 93). [33] Burke, Language as Symbolic Action, 18. [34] Hasian and Delgado, ‘‘Trials and Tribulations,’’ 257. [35] Otto Santa Ana, ‘‘‘Like an Animal I Was Treated’: Anti-immigrant Metaphor in U.S. Public Discourse,’’ Discourse and Society 10 (1999): 191􏰀224. See also Sarah Hill, ‘‘Purity and Danger on the U.S.􏰀Mexico Border, 1991􏰀1994,’’ South Atlantic Quarterly 105 (2006): 777􏰀99. [36] Flores, ‘‘Constructing Rhetorical Borders,’’ 363. [37] With a recent increase in applications for U.S. naturalization, and under pressure to err on the side of caution in vetting applicants for potential security threats, federal examiners have increased their rejection rates. In 2007, for example, approximately 12 percent of applications for naturalization were denied. See Julia Preston, ‘‘Perfectly Legal Immigrants, Until They Applied for Citizenship,’’ New York Times, April 12, 2008.

      64 D. R. DeChaine [38] Bhikhu Parekh, ‘‘What Is Multiculturalism?’’ Multiculturalism: A Symposium on Democracy in Culturally Diverse Societies 484 (December 1999), http://www.india-seminar.com/1999/ 484/484%20parekh.htm/. [39] Burke, Language as Symbolic Action, 3􏰀24. [40] Chris Simcox, ‘‘About Us,’’ Minuteman Civil Defense Corps, http://www.minutemanhq.com/ hq/aboutus.php/. [41] Kevin Michael DeLuca and Jennifer Peeples, ‘‘From Public Sphere to Public Screen: Democracy, Activism, and the ‘Violence’ of Seattle,’’ Critical Studies in Media Communication 19 (2002): 125􏰀51. [42] The Border Fence Project emerged on the heels of the 2006 Secure Fence Act, authored by California Republican Representative Duncan Hunter, which required the U.S. Department of Homeland Security to build 854 miles of fence along the 1950-mile U.S.􏰀Mexico border. [43] Chris Simcox, ‘‘Senate and President Promote Anarchy at Borders and in American Cities,’’ Minuteman National Blog, April 11, 2006, http://www.minutemanhq.com/b2/index.php/ national/2006/04/11/. [44] Border Fence Project, BorderFenceProject.com, http://www.borderfenceproject.com/. [45] Chris Simcox, ‘‘Minuteman Corps Expands is [sic] Efforts to Secure the Border,’’ Minuteman Border Fence, January 2, 2007, http://www.minutemanhq.com/bf/pl7.php/. [46] ‘‘About Us,’’ Minuteman Border Fence, http://www.minutemanhq.com/bf/about.php/. [47] Simcox, ‘‘Minuteman Corps.’’ [48] Jim Wood, ‘‘A Letter to My Fellow Americans,’’ BorderFenceProject.com, http://www.border fenceproject.com/letter.shtml/. [49] See, for example, Minuteman Border Fence. [50] Chris Simcox, ‘‘Open Letter to the President of the United States,’’ MCDC Forums, June 19, 2007, http://forum.minutemanhq.com/phpBB2/viewtopic.php?t􏰁11793/. [51] Chris Simcox, interview by Trish Hinojosa, NOW on the News, PBS, May 4, 2007. [52] Chris Simcox, interview by Alan Colmes, Hannity & Colmes, Fox News, April 3, 2006. [53] Susy Buchanan and David Holthouse, ‘‘Minuteman Leader has Troubled Past,’’ Southern Poverty Law Center, http://www.splcenter.org/intel/news/item.jsp?aid􏰁149&site_area􏰁 1&printable􏰁1/. [54] Simcox, ‘‘Open Letter’’; ‘‘Donate to the Minuteman Border Fence,’’ Minuteman Border Fence, https://secure.responseenterprises.com/mmfence/?a􏰁571. [55] The MCDC works to document what it perceives as an increase in criminal activity in the United States as a result of unsecured borders. For example, the online MCDC forum includes a main thread titled ‘‘Illegal Alien Crime,’’ with the subheading ‘‘Please post any articles about illegal alien crime and DUI incidents here. Having these in one place will illustrate the tragic consequences of illegal migration.’’ ‘‘MCDC Forums,’’ Minuteman Civil Defense Corps, http://forum.minutemanhq.com/phpbb2/. [56] Simcox, ‘‘Minuteman Corps.’’ [57] ‘‘DIY Border Fence,’’ PirateNews.org, http://www.piratenews.org/newswire/html/. No docu- mentation for either of these statistics is provided on the website. [58] Brian Bonner, ‘‘Minutemen to Build Arizona-Mexico Border Fence,’’ April 20, 2006, http:// bonner.wordpress.com/2006/04/20/minutemen-to-build-arizona-mexico-border-fence/. [59] Chris Simcox, ‘‘Message from Chris Simcox,’’ Minuteman Border Fence, http://www. minutemanhq.com/bf/about.php/. [60] Simcox, ‘‘Open Letter.’’ [61] Stuart Hall, ‘‘The Whites of Their Eyes: Racist Ideologies and the Media,’’ in The Media Reader, ed. Manuel Alvarado and John O. Thompson (London: BFI Publishing, 1990), 13. [62] Minuteman Civil Defense Corps, ‘‘About Us.’’ [63] Minuteman Civil Defense Corps, ‘‘The Minuteman Pledge,’’ http://www.minutemanhq.com/ hq/mmpledge.php/. [64] Wood, ‘‘Letter to My Fellow Americans.’’

      Bordering the Civic Imaginary 65 [65] ‘‘Build It Now!’’ Minuteman Border Fence, http://www.minutemanborderfence.com/. [66] Simcox, ‘‘Open Letter.’’ [67] Minuteman Civil Defense Corps, ‘‘Minuteman Pledge.’’ [68] Simcox, ‘‘Minuteman Corps.’’ [69] ‘‘Become a Cyber Minuteman!’’ BorderFenceProject.com, http://www.borderfenceproject. com/cybermm.php/. [70] Nevins, Operation Gatekeeper, 147􏰀48. [71] Anne Demo, ‘‘Sovereignty Discourse and Contemporary Immigration Politics,’’ Quarterly Journal of Speech 91 (2005): 295. [72] David Newman, ‘‘On Borders and Power: A Theoretical Framework,’’ Journal of Borderlands Studies 18 (2003): 20. [73] Kenneth Burke, Permanence and Change: An Anatomy of Purpose (New York: New Republic, 1935), 70. [74] Cornelius Castoriadis, The Imaginary Institution of Society, trans. Kathleen Blamey (Cam- bridge, MA: MIT Press, 1987). See also Arjun Appadurai, Modernity at Large: Cultural Dimensions of Globalization (Minneapolis: University of Minnesota Press, 1996); Charles Taylor, Modern Social Imaginaries (Durham, NC: Duke University Press, 2004); Benedict Anderson, Imagined Communities: Reflections on the Origin and Spread of Nationalism (London: Verso, 1983). [75] Taylor, Modern Social Imaginaries, 23. [76] See, for example, Michael Warner, Publics and Counterpublics (New York: Zone Books, 2005); Dilip Parameshwar Gaonkar and Benjamin Lee, eds., ‘‘New Imaginaries,’’ Special Issue, Public Culture 36 (2002); Robert Asen and Daniel C. Brouwer, eds., Counterpublics and the State (Albany: State University of New York Press, 2001); Craig Calhoun, ed., Habermas and the Public Sphere (Cambridge, MA: MIT Press, 1993). [77] Julia Preston, ‘‘As Pace of Deportation Rises, Illegal Families Are Digging In,’’ New York Times, May 1, 2007. I was unable to locate reliable data regarding 2007 deportation rates. [78] Ngai, Impossible Subjects, 269. [79] Dana L. Cloud, ‘‘‘To Veil the Threat of Terror’: Afghan Women and the BClash of Civilizations􏰂 in the Imagery of the U.S. War on Terrorism,’’ Quarterly Journal of Speech 90 (2004): 285􏰀306. [80] DeChaine, ‘‘Imagined Immunities.’’ [81] Robert S. Chang, ‘‘A Meditation on Borders,’’ in Immigrants Out! The New Nativism and the Anti-Immigrant Impulse in the United States, ed. Juan F. Perea (New York: New York University Press, 1997), 244, 246.

    1. April is the cruellest month, breeding Lilacs out of the dead land, mixing Memory and desire, stirring Dull roots with spring rain.

      While we may imagine spring as a great season, where life begins to bloom from the earth, who exactly would think the opposite? Why would spring be considered cruel?

    1. NBA suspends season until further notice after player tests positive for the coronavirusplayundefinedPlay VideoPlayUnmuteCurrent Time 0:00/Duration 4:29Loaded: 0.00%0:00Stream Type LIVESeek to live, currently playing liveLIVERemaining Time -4:29 SharePlayback Rate1xChaptersChaptersDescriptionsdescriptions off, selectedCaptionscaptions settings, opens captions settings dialogcaptions off, selectedClosed CaptionsAudio Trackdefault, selectedAutoplay, selectedClosed CaptionCaption SettingsSettingsFull ScreenThis is a modal window. An error occurred during video playback, possibly due to network connectivity or being in a backgrounded browser tab. Please try again. Error Code: PLAYER_ERR_TIMEOUT Session ID: 2020-03-16:f17f313a4840e53d53e1f0b0 Player Element ID: vjs_video_3 OK Close Modal DialogBeginning of dialog window. 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This modal can be closed by pressing the Escape key or activating the close button.CloseFacebookTwitterEmailCopy LinkCopy Embed LinkURLURLStart atCopyEmbed CodeCopyNBA season suspended due to coronavirus (4:29)Adrian Wojnarowki joins SVP to give the latest details about the NBA's suspending the season after Rudy Gobert tests positive for the coronavirus. (4:29)FacebookTwitterFacebook MessengerEmailMar 12, 2020ESPN News Services FacebookTwitterFacebook MessengerPinterestEmailprintThe NBA suspended its season after a Utah Jazz player tested positive Wednesday for the coronavirus."The NBA is suspending game play following the conclusion of [Wednesday's] schedule of games until further notice,'' the league said in a statement issued shortly after 9:30 p.m. ET. "The NBA will use this hiatus to determine next steps for moving forward in regard to the coronavirus pandemic.''The player is Rudy Gobert of the Jazz, sources told ESPN's Adrian Wojnarowski. That led to Utah's game at Oklahoma City being postponed just before tipoff.The NBA is expected to address next steps with teams when it conducts a call with the board of governors at 12:30 p.m. ET Thursday, sources told ESPN's Ramona Shelburne.Also Wednesday, the NBA G League announced in a statement that it has "suspended the 2019-20 season, effective after tonight's games."Mavericks owner Mark Cuban said during ESPN's broadcast of Dallas' 113-97 victory over the visiting Denver Nuggets that he was shocked by the news that the season was being suspended.Editor's PicksSources: Jazz center Gobert has coronavirusWhat we know and don't know about the NBA's suspension of playNBA players react to league suspending season due to coronavirus2 RelatedHe said his initial reaction was, "This is crazy. This can't be true."He continued: "I mean, it's not within the realm of possibility. It seemed more like out of a movie than reality."Cuban then added that he isn't an expert and defers to NBA commissioner Adam Silver on these matters."I trust Adam [Silver]. You know what? It's really not about basketball or money," Cuban said. "Literally, if this thing is exploding to the point where all of a sudden players and others have had it, you think about your family. You want to make sure you're doing this the right way. Now it's much more personal, and you've seen what's happened in other countries, but just the whole idea that it's come this close and potentially a couple players have it, just, 'stunning' isn't the right word. Just crazy."After the game, Cuban said teams have been told they can continue to practice, and he added that players have been told they should not have visitors from out of town.The last game to be completed Wednesday was Dallas' win over Denver, as the New Orleans Pelicans' game at the Sacramento Kings was postponed because one of the referees scheduled to work that game worked Utah's game on Monday."The game was canceled out of an abundance of caution," the NBA said.A source told ESPN's Royce Young that Gobert was never at Chesapeake Energy Arena on Wednesday, but he was in Oklahoma City and was set to play if he tested negative for the coronavirus.Players were on the floor for warm-ups when they were told to return to the locker rooms. About 30 minutes later, fans were told by the public-address announcer that the game was postponed "due to unforeseen circumstances."The Thunder players were cleared to leave the arena, but Utah's players are still there, and sources told Wojnarowski that the team would need to coordinate with Oklahoma and Salt Lake City public health organizations before it could return to Utah.Players from teams the Jazz have played within the past 10 days were told to self-quarantine, sources told ESPN's Brian Windhorst. Those teams are the Cleveland Cavaliers, New York Knicks, Boston Celtics, Detroit Pistons and Toronto Raptors."I'm sure I probably had contact with [Gobert]. But at the same time, like I said, [I'm] just taking precautions," Detroit's Langston Galloway said. "We've been washing our hands, and when the reports started coming out, everybody's kinda been on their hand sanitizer, washing their hands, just staying focused on that moment of, hey, [we have] interaction with a lot of different people and knowing that at the end of the day, you might've touched the ball, you might've interacted with a fan, and just being [cautious] with that going forward."Charlotte Hornets coach James Borrego, speaking before his team played Wednesday at Miami -- where news of the shutdown broke during the fourth quarter -- said, "These are scary times.''There are 259 games, roughly 21% of the schedule, left to play this season -- and no one knows if, or when, things will resume."We believe in the leadership of the league,'' Philadelphia 76ers general manager Elton Brand said.Information from The Associated Press was used in this report. 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    1. Note: This rebuttal was posted by the corresponding author to Review Commons. Content has not been altered except for formatting.

      Learn more at Review Commons


      Reply to the reviewers

      We are grateful to all three reviewers for their careful analysis of the manuscript, and for their constructive comments. Two common critiques were:

      (1) that assaying origin firing via an independent method would strengthen the conclusions, and (2) that additional analysis of ribonucleotide incorporation to exclude the retention of lagging-strand primers would allow us to definitively determine whether Pol ɛ plays a role in lagging-strand synthesis.

      We will include experiments to address both critiques in a revised manuscript. To independently verify changes in origin efficiency, we will sequence nascent BrdU-containing DNA across a time course from cells released into S-phase: we will also use the last timepoint of our Okazaki sequencing analysis to control for potential cell-cycle differences. To further test the contribution of Pol ɛ and ascertain whether lagging-strand primers are retained, we will analyze ribonucleotide incorporation in both wild-type and pol2-M644L (Pol ɛ ribonucleotide hypo-incorporating) strains. We address individual specific comments and our planned revisions in more detail below.

      Reviewer #1 (Evidence, reproducibility and clarity (Required)):

      This study examined the consequences of limiting levels of DNA polymerase d (Pol d) in yeast. The authors first reported multiple genome instability consequences following lowered Pol d level, including defect in S phase progression, growth defect, elevated spontaneous DNA damage, accumulation of ssDNA and activation of replication and DNA damage checkpoint. These observations are solid but not unexpected. By genome wide analysis using the Okazaki fragment (OF) mapping and ribonucleotide mapping (for polymerase usage), the authors claim a few potentially novel and striking observations that lowered Pol d differentially impact efficiencies of early vs. late origins, and that lowered Pol d results in Pol e participating in lagging strand synthesis around late origins. However, I remained unconvinced based on the data presented. These observations need to be further substantiated and alternative interpretations should be considered.

      \*Main concerns:** *

      One of major conclusions the authors tried to make is that the early vs. late origins are differentially affected by low level of Pol d. First, they used OF mapping data to examine origin efficiency. Asynchronous "Cultures were treated with IAA for 2h before the addition of rapamycin for 1h to deplete DNA ligase I (Cdc9) from the nucleus via anchor-away". IAA concentrations used were of 0, 0.2 mM, 0.6 mM, and 1 mM. The problem is that Figure S1 clearly showed that treating asynchronous cultures with >0.1 mM of IAA for as short as 30 min significantly alters the cell cycle profiles, mainly resulting in accumulation of S phase cells, to different extent. Presumably Okazaki fragments accumulated from these cultures suffering from the synchronizing effect may not be representative of the real change in global replication profile. For instance, it is not difficult to predict that the Okazaki fragments enrichment may be skewed towards late origins if more cells are accumulated in mid S phase following Pol d depletion. For this reason, I don't believe the result is conclusive. The experiment may be re-designed for samples at different time points following release from G1.

      We agree that altered cell-cycle profiles might affect the number of Okazaki fragments sequenced in late vs early replicating regions of the genome. As noted by reviewer 3 in cross-commenting, these differences should not affect origin efficiency calculations as these are based on the ratio of reads on each strand (and therefore normalized). To more directly address this question, we will calculate origin firing efficiencies from the final timepoint of the arrest-release experiments shown in Figure 4 as suggested by the reviewer. We will also analyze origin efficiency using BrdU over a time course.

      This concern also should preclude the authors from drawing conclusion about Pol e usage on lagging strands based on comparison between HydEn-seq data and OF mapping data shown in Figure 6. In fact, the rNMP incorporation change is very similar between early and late origins. The only evidence that the author rely on is the discrepancy in OF data between the two groups origins, which makes the reliability if origin efficiency measurement the central piece of data in this study. Thus, alternative approaches should also be considered to map origin efficiencies.

      As noted above, we agree that an independent method of tracking origin firing efficiencies would be helpful to strengthen our conclusions. To this end, we will analyze time courses of BrdU incorporation from cultures released into S-phase.

      Even if Pol e strand bias is lowered at late origins, as the authors tend to believe, there are still alternative models other than Pol e being used for lagging strand synthesis. For instance, if TLS polymerases are used on lagging strands, it could result in more ribonucleotide incorporation on the lagging strand, as they are lower-fidelity polymerases. Alternatively, if Pol d scarcity leads to more Pol e synthesis or lower RNA primer processing, it might also contribute to more apparent ribonucleotide incorporation on the lagging strands.

      We feel that the widespread use of TLS polymerases is unlikely, especially given the data in figure 6A that show no growth or viability change upon deletion of all three TLS polymerases in the Pol ∂ depletion strain, even at very low levels of Pol ∂. We agree with the reviewer that our data do not conclusively rule out increased retention of lagging-strand primers – as we state in the text. We aim to test this possibility by analyzing ribonucleotide strand bias in a pol2-M644L strain that incorporates fewer ribonucleotides than the wild-type Pol ɛ. In this case, increased lagging-strand primer retention would lead to a lagging-strand bias of ribonucleotides upon Pol ∂ depletion, while increased Pol ɛ usage would not. An analogous experiment with wild-type POL2 is potentially harder to interpret because the wild-type polymerase is the predominant source of ribonucleotides in a wild-type strain (Nick McElhinny et al, 2010 - PMID:20729855), but we now have the data for this strain in hand and ready to analyze.

      In Figure S5, the two HydEn-seq replicates are very different, where replicate1 shows very low strand bias. I suspect perhaps the strain used for replicate 1 does not contain pol2-M644G or rnh202 deletion.

      The change in ribonucleotide incorporation is indeed substantially stronger in one replicate than the other. We have additional time-course data from a Pol ∂ depletion showing that ribonucleotide strand bias decreases over time as Pol ∂ is depleted, and will include this in a revised manuscript.

      Reviewer #1 (Significance (Required)):

      Given that different aspects of Pol d deficiency have been implicated in various human diseases and cancer, this type of analysis is of interest to the field.

      Reviewer #2 (Evidence, reproducibility and clarity (Required)):

      \*Summary** *

      In this manuscript the authors explore the basis of the deleterious effects of reduced levels of the replicative Polymerase delta. This polymerase plays several important roles in the replication process: it synthesizes most of the lagging strand, but also extends the first primer during the synthesis of the lagging strand, and it contributes to the removal of the RNA and most of the DNA synthesized by primase/Polymerase alpha during Okazaki fragment maturation. In this study, the authors systematically analyze the impact of Pol delta depletion in S. cerevisiae. They use a degron-tagged allele to modulate the levels of the polymeraes and apply mainly NGS methods and classical genetics to explore the consequences for survival, checkpoint signalling and replication features such as fork speed, origin firing efficiency and Okazaki fragment length and distribution. They report that Pol delta depletion leads to a checkpoint activation via the Rad9-dependent damage signalling pathway (but not the Mrc1-dependent replisome-associated signalling) and an accumulation of single-stranded DNA. Phosphorylation of histone H2A is taken as a marker of DNA double-strand breaks, and from the observation that deletion of recombination factors, but not end-joining factors aggravate the fitness of Pol delta-depleted cells they conclude that homologous recombination is responsible for the repair of these breaks. Analysis of replication by Okazaki fragment sequencing indicates a slight decrease in the firing efficiency of early/efficient origins, but an increase in the firing efficiency of late origins. They also observe a reduction in fork speed, although they are not able to attribute this to either a globally slower fork movement or an increase in the stalling of individual forks. They find that Pol delta depletion does not change the size of Okazaki fragments, but causes defects in the nick translation during Okazaki fragment maturation. Finally, they use NGS technology to show that the leading strand Polymerase epsilon engages in lagging strand replication particularly at late origins when Pol delta is depleted. From their observations, the authors develop a model where depletion of Pol delta primarily affects late replicating regions. They explain this by invoking a stable association of Pol delta with early replisomes, which sequesters the enzyme, thus causing an under-supply at replisomes that assemble later during S phase. This then leads to the involvement of Pol epsilon on the lagging strand. Based on the observation that fork speed and Okazaki fragment maturation are both affected, they propose that these two reactions normally compete for Pol delta, suggesting that optimal replication would require two molecules of the polymerase per fork.

      \*Major comments** *

      The experiments shown here are largely clean and well controlled, and the manuscript is written nicely and well-structured.

      Compared to the Okazaki fragment analysis, the treatment of double-strand breaks appears somewhat cursory and remains inconclusive. Phosphorylation of H2A seems insufficient evidence for double-strand breaks, as other structures could also give rise to that signal. These lesions should be detected in a more direct manner, e.g. pulsed-field gel electrophoresis. The authors also don't provide a mechanism by which such breaks would emerge. Related to the minor effect of the ku mutant, I am wondering about the altered appearance of the colonies in Figure 2F (concerning both ku70 and rad51) - what is different about these, and could their „denser" appearance explain the slight suppression effect observed?

      We agree that our treatment of double-strand breaks is limited: consistent with comments from all three reviewers about which aspects of this work are most novel, we intend to focus as much as possible on replication enzymology here. We will tone down the language around double-strand breaks in the manuscript.

      Concerning the damage signalling: it is surprising to see a damage signal at concentrations of IAA that do not lead to a significant depletion of Pol delta yet (0.05 mM). At this point, it is hard to imagine DSBs to form. Could the authors explain this discrepancy?

      We note that, as observed in Figure 1A and to a slightly lesser extent in Fig. 2E, Pol ∂ levels are already substantially reduced in 0.05 mM IAA. This reduction appears sufficient to induce damage

      The notion that late origin firing is enhanced despite checkpoint activation is counterintuitive. Do the authors think that this effect overcomes the suppression of late origins that is normally associated with checkpoint activation? It would be helpful to test whether abolishing that phenomenon (e.g. by a mec1-100 mutant) would enhance the effect and render late origins even more active.

      We thank the reviewer for this excellent suggestion: we will test the effects of a mec1-100 mutant and include the results in a revised manuscript.

      It would be important to characterise the fork speed defect better, using alternative methods rather than just relying on Okazaki fragments. A differentiation between slower fork progression and more frequent fork stalling would be relevant and might help to evaluate the contribution of Pol epsilon. This might be accomplished by DNA fibre analysis. Alternatively, BrdU incorporation could serve to observe replication over the entire genome rather than only in the vicinity of replication origins. It would also be important to differentiate fork speeds in early versus late replicating regions - according to the authors' model, the defects should be most obvious in the late regions (Fig. 4 concerns only early origins).

      As noted above in our response to reviewer 1, we will use BrdU incorporation to independently verify changes in fork speed and origin firing. Analysis of fork speed in late-replicating regions is challenging regardless of the methodology used, due to contributions from converging forks, but we will try to do this

      Figure S3: Considering the differences in cell cycle progression, it would make more sense to compare equivalent stages of the cell cycle / S phase rather than identical time points.

      We can include this analysis, although the changes in cell cycle progression and origin firing efficiency make such comparisons somewhat subjective

      Considering that the Okazaki fragment analysis was done with non-synchronised cultures, is it possible that the skew in the cell cycle profile could influence the Okazaki fragment pattern?

      (copy-pasted from our response to a similar query by reviewer 1)… We agree that altered cell-cycle profiles might affect the number of Okazaki fragments sequenced in late vs early replicating regions of the genome. As noted by reviewer 3 in cross-commenting, these differences should not affect origin efficiency calculations as these are based on the ratio of reads on each strand (and therefore normalized). To more directly address this question, we will calculate origin firing efficiencies from the final timepoint of the arrest-release experiments shown in Figure 4 as suggested by the reviewer.

      Would it be possible to monitor not only total Pol delta levels, but also the level of Pol delta bound to the chromatin? It is shown that the level of Pol delta is depleted in the whole cell extracts, but it would be interesting to see how severe the depletion is on the chromatin.

      We agree that the relative fraction of chromatin-bound vs free Pol ∂ is an interesting question, and will attempt this experiment. However, we note that extensive depletion of Pol3 makes it hard to detect by Western blot, so the results are likely to be most informative at modest depletion levels. Regardless, these data should give us an idea of the size of the ‘free’ Pol ∂ pool in cells with normal or mildly reduced Pol ∂.

      Figure 6 is confusing and should be clarified: - Figure 6B: assigning the Watson and Crick strands in the schematic would make that figure easier to understand; - Figure 6B-C: the axes are labeled as 'Fraction of rNMP on Watson strand', but would it not make more sense if they were labeled 'Fraction of rNMP in Crick strand'? - Figure 6D-E: the right side scale is labelled as 'increase in rNMP on Crick strand' while in the figure legend is says it is 'change in the fraction of ribonucleotides mapping to the Watson strand. That description should be clarified; - Figure 6D: using 'Change in Okazaki fragments strand bias' to label the black line (description in the box above the figure) instead 'Change in Okazaki strand bias' would be more appropriate; - Figure 6F: the authors seem to have subtracted strand bias measured for Okazaki fragments from the strand bias measured for rNMP. It is valid to subtract these biases from each other, considering that the two structures arise independently and with different frequencies?

      We can make changes to figure 6 as suggested. Regarding the validity of subtracting strand biases, we think this is sufficient to give at least a semi-quantitative view of Pol ɛ usage since both of our sequencing approaches produce quantitative readouts that directly report on replication direction or polymerase usage, respectively.

      \*Minor comments:** *

      Can the authors conclude that Pol delta deficiency/ incompleteness of lagging strand synthesis affects the nucleosome deposition onto DNA? (Figure 5-A)

      We cannot rule out that this is occurring, and we agree that this is an interesting question for future studies. But the changes that we observe Okazaki fragment terminus location are very similar to our previously published observations from cells lacking Rad27 function, consistent with decreased nick translation.

      Why did the authors use rnh202Δ and not a mutant in the catalytic subunit of RNase H2?

      Deletion of any subunit of the heterotrimeric RNase H2 complex completely abolishes its function in yeast, so RNH202 was a somewhat arbitrary choice

      An extra control might be useful: comparing POL3-AID rnh202Δ with the POL3-AID pol2M644G rnh202Δ triple mutant could further confirm that the observed effect is Pol epsilon-dependent.

      We agree (see also our response to reviewer 1). In addition to the wild-type, we will analyze pol2-M644L – a mutant in which Pol ɛ incorporates fewer than normal ribonucleotides. An increase in ribonucleotide density on the lagging strand in pol2-M644L would support increased primer retention on the lagging strand.

      Figure 2H: It would be good to see the cell cycle distribution corresponding to the western blot images.

      We can include this

      Various spelling, grammar or precision of expression issues: - Pg. 4, line 4: endonucleolytically instead of nucleolytically. - Pg. 6, line 10: Remove 'was'. - Pg. 6, line 12: Remove 'in vivo' from the subtitle. - Pg. 6, line 14: 'an C-terminal' should be 'a C-terminal' - Pg 16, line 13: Phrasing implies that the synthesis of both leading and lagging strands by Pol delta in regions in the vicinity of replication origins is essential - please quote any paper testing its essentiality. - Please follow standard yeast genotype nomenclature, remove ';' when listing the yeast genotypes (e.g. POL3-AID mec1Δ sml1Δ instead of POL3-AID;mec1Δ;sml1Δ- example from Figure 2-B). - Concentrations of IAA are missing in few places (e.g. legend of Figure 1-C, page 24). - Figure 1A: add the label 'IAA (mM)' - Figure 2G: pleae provide a shorter exposure of the H4 blot in addition to the one shown. - Figure 6: adding a schematic presenting the events at actively and passively replicating late origins (and the predictions about leading and lagging strand bias) would help to understand the figure. - The format of the references is inconsistent. - 'On Watson/Crick strand' should be replaced with 'in Watson/Crick strand' We will fix typos, etc

      Reviewer #2 (Significance (Required)):

      This is a nice piece of work that provides in vivo confirmation of several observations that have been made in purified recombinant systems. In that sense, the overall novelty is limited, but this type of study is still important to do, as biochemical assays do not always reflect what is happening in cells, and this study gives insight into basic activities of the replisome. The participation of Pol epsilon in lagging strand synthesis is an interesting observation. Overall, the study will be of interest for the DNA replication field. My own expertise is in replication, predominantly in yeast. I have experience in NGS analysis of replication as well as in genetic analysis of the DNA damage response. I therefore feel competent to evaluate all aspects of the manuscript.

      Reviewer #3 (Evidence, reproducibility and clarity (Required)):

      This manuscript describes the consequences of reducing the cellular concentration of a pol-delta subunit in S. cerevisiae. Pol-delta plays multiple cellular roles at both the replication fork (it is one of two DNA polymerases responsible for lagging strand synthesis) and during repair synthesis after DNA damage. The authors combine genetic and genomic methodologies to characterise how reduction in pol-delta concentration impacts on cellular fitness and specifically lagging strand synthesis. Overall it is technically a well executed study that is clearly presented and the data are predominantly appropriately interpreted.

      \*I have a number of major comments:** *

      1) The authors apply OK-seq (a methodology first developed by the senior author and therefore they are clearly experts at this) to study the consequence of pol-delta depletion on genome replication. OK-seq requires isolation of Okazaki fragments and this in turn requires removal of DNA ligase (Cdc9) - the authors achieve this with the anchor away system. My concern is that in these experiments the authors are depleting two factors required for lagging strand synthesis: pol-delta and ligase; it is unclear to me how the authors can determine the relative contribution of each depletion to the observed phenotypes. Could some of the observed phenotypes (e.g. fork slowing, 5' and 3' ends of Okazaki fragments, etc) be a consequence of the double depletion, rather than just pol-delta depletion as concluded by the authors? The authors present this as a method to determine genome replication timing, but really it is an assay to look at fork direction. Given the need for an addition mutation in OK-seq (cdc9), I would encourage the authors to consider a more direct assay for replication dynamics upon pol-delta depletion, such as a copy number measure (or BrdU-ip) of DNA replication or DNA combing - these methods don't require Cdc9 depletion and could therefore ensure that observed phenotypes are a consequence of pol-delta depletion (rather than the double depletion).

      As outlined in our response to the first two reviewers, we will do BrdU-IP experiments. We agree that the double depletion may have an effect on fork speed, and that BrdU-IP will allow us to test this possibility. However, we note that our analysis of Okazaki fragment initiation/processing requires the depletion of Cdc9, so for this we are limited to looking at differences between Cdc9 depletion alone vs Cdc9 depletion + Pol3 depletion.

      2) One major conclusion reached by the authors is that pol-epsilon can contribute to lagging strand synthesis upon pol-delta depletion (at least during late replication). This conclusion comes from the authors use of HydEn-seq to measure rNTP incorporation from which the contribution of a polymerase (pol-epsilon in this case) to strand synthesis can be determined. In a manner analogous to OK-seq, this requires the introduction of additional mutations (both in the polymerase and by the removal of RNaseH activity). The authors interpretation that pol-epsilon can play a role in lagging strand synthesis is dependent upon there being no temporal change in pol-delta strand-displacement activity, despite continued pol-delta depletion through S phase. It is not clear to me that the data presented in Fig 5 & 6 has the sensitivity to conclude this (and the OK-seq data is also subject to the potential bias of the double depletion of pol-delta and Cdc9). I feel that a necessary control to support this conclusion, would be to undertake the HydEn-seq experiment in the absence of the pol-epsilon mutation (just pol-delta depletion in the absence of RNaseH activity). This would allow the authors to measure any increase is residual rNTPs (likely from pol-alpha primase) on the lagging strand as a consequence of pol-delta depletion and determine whether they are equally likely in early and late S phase.

      As discussed in our response to the first two reviewers, we will analyze analogous data from both a POL2 wild-type and a pol2-M644L strain that incorporates fewer ribonucleotides than the wild-type.

      \*The following comments are more minor:** *

      -for the experiment in Fig S1B, the growth in 1.0 mM IAA is somewhat surprising given how sick the cells appear on equivalent plates. I couldn't find in the methods a description of the experimental conditions.

      The cells grow very slowly in 1 mM IAA (doubling time doubles). We think this is quite consistent with the poor growth on plates

      -there is considerable variability in the S phase kinetics from bulk DNA analysis (flow cytometry) when comparing Fig 1C, 2D, S3. Fig 1C appears to be the exception, with all the other figures showing poor S phase progress by comparison. It would be useful for the authors to recognise these differences and comment upon them. E.g. they appear to all be identical experiments, but are there experimental differences that could explain the different kinetics?

      We see some variability in our release, but generally cells enter S-phase at around 30 minutes. The release in figure S3 was carried out at 25 ˚C rather than 30 ˚C, which accounts for the additional delay in these data

      -Fig 2F, why is the rad51-deletion less severe that rad52-deletion - should they not be identical?

      We agree that these should logically be very similar: we do not know why the two mutants behave slightly differently at some (but not all) IAA concentrations

      -Fig 2H - could the authors show the flow cytometry (in a supplemental figure) for this experiment?

      We can show this

      -Fig 3B-E: OEM is described as a measure of origin efficiency - how is possible for this to have negative values?

      OEM describes Okazaki fragment strand bias around previously identified origins. If such an origin does not fire in our strain background, a negative OEM can result.

      -pp9: "Analysis of Okazaki fragment strand bias across the genome suggested that the average direction of replication was relatively similar at most loci across all Pol3 depletion conditions". The authors data is quantitative and they should be able to quantify how similar their data are across the various conditions, rather than making a qualitative statement: "relatively similar".

      We apologize and can re-phrase this. The intention of this statement is simply to draw the reader’s attention to the fact that global distributions of Okazaki fragments are not completely altered (e.g. only 1-2 origins per chromosome) as a prelude to the more quantitative analysis that follows in figure 3.

      -pp9: "origin firing efficiency in S. cerevisiae correlates strongly with replication timing"; it would be useful for the authors to support this statement with a citation.

      We will add 1-2 citations to support this statement

      -Fig 4A: it would help the reader if the authors could show 'zoomed in' examples of the points that the authors make (in addition to the whole chromosome view): slowed fork progression, reduced early origin activity, increased late origin activity (e.g. an origin that is normally passively replicated, that upon pol-delta depletion is no longer passively replicated and therefore becomes more efficient), etc.

      We agree that this would be helpful, and can add examples in the supplement

      -pp11: "An analogous global decrease in replication-origin firing efficiency has been observed in Pol ∂-deficient human fibroblasts" - but the authors are reporting a global increase in origin firing efficiency (Fig 3B).

      We can re-phrase this.

      -the nucleosomal ladder in Fig 5A is only weakly apparent from the gel and not particularly apparent from the density trace, this makes it's disappearance upon IAA treatment hard to interpret. Is the weak nucleosomal ladder what the authors had anticipated (in the absence of IAA)?

      We do not expect a weaker nucleosomal ladder than normal in the absence of IAA. In our experience these gels just sometimes give better ladders than others, and we hope that the traces help with interpretation

      -I found the effects being described by the authors in Fig 5B & C difficult to see, particularly for the transcription factors. Furthermore, why are these data differently normalised to those in Fig 4B & C (median vs. maximum)

      In figure 4 we normalize to maximum since all DNA should eventually be replicated, and we therefore think that showing coverage relative to a maximum value of 1 is most informative. In figure 5 we compare distributions of termini around obstacles, and therefore feel that normalizing to the median is a more appropriate way to compare enrichment around a given meta-element. The shapes of the graphs would be unchanged by choosing a different normalization point. In order to make changes easier to see, we can make the lines thinner in figure 5 and/or change the y-axis scale.

      -the final sentence of the results section returns to an analysis of the OK-seq data and is essentially a temporally segregated analysis (Fig S6) otherwise equivalent to that presented in Fig 5B. Given the importance placed on these data by the authors in the interpretation of the HydEn-seq data, I feel that it would help the reader to have been presented with these data earlier in the results section.

      We can move these data up

      -p22: OK-seq methods. The authors should indicate the culture conditions for these experiments.

      We can include this

      -p22: Computational analyses: the authors should indicate which reference genome assembly they used.

      We can include this

      -Fig 6B & C: the y-axis labels are confusing - do the authors mean Crick strand here?

      Oops. Yes, we do. We thank the reviewer for catching this

      \*REFEREE CROSS COMMENTING:** *

      All three reviewers seems to be in broad agreement about this manuscript. There is one significant concern raised by the other reviewers that I'd missed: that some of the Okazaki fragment analysis was done with non-synchronised cultures. I agree with this concern, however I don't think that there is necessarily a problem with the alternative explanation suggested by reviewer #1 ('Okazaki fragments enrichment may be skewed towards late origins'). While the accumulation of S phase cells might well be expected to lead to a bias towards isolating more Okazaki fragments from around late origins, the authors calculate the fraction of reads (i.e. Okazaki fragments) mapping to each strand. The potential presence of more late S phase cells would give greater sequence coverage over late replicating regions, but alone would not alter the fraction of reads mapping to each strand. However, I agree that interpretation of this experiment is not as simple as suggested by the authors and there may well be alternative explanations along the lines suggested by reviewer #1.

      There was a subsequent Okazaki fragment experiment performed with synchronised cells (Fig 4) and it might be possible to use these data to assess any differential impact on early vs late origins.

      We agree, and will do this analysis

      Reviewer #3 (Significance (Required)):

      My expertise is in DNA replication and genome stability, particularly replication timing and replication origin function.

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      Reply to the reviewers

      __Reviewer #1 (Evidence, reproducibility and clarity (Required)):


      In this article, the authors characterize a complex formed by sec22b-stx1-Esyt2. They investigate how such interactions are involved in the modulation of dynamics of the plasma membrane in the context of neuritogenesis. They conclude that the contact sites between the ER and the plasma membrane, mediated by the afore-mentioned complex, contribute the expansion of the plasma membrane.

      **Major comments:**

      Overall, the article clearly shows that in mammalian cells there is an interaction between sec22b-stx1-Esyt2 which appears to be important for filopodia formation and possibly neuritogenesis in neurons. However, performing additional experiments to better clarify some links and assumptions made by authors could strengthen the article.

      The manuscript relies on work performed either on cell lines (HeLA, PC12) or primary neuronal cultures. Although it is clear the value of the findings obtained using cell lines, they should be seen as a complementary rather than an exclusive approach. This is particularly important as the authors often make claim about neuron-related cellular biology.

      For instance, the biochemistry-based findings on the interaction and characterization of the protein complex (Figure 1) are all derived from experiments perfomed in Hela or PC12. As the authors have the capacity to culture and manipulate primary neuronal cultures, such findings should be validates in neuronal cells. The authors could also consider performing biochemical experiments (i.e. co-ip) of the endogenous proteins in neuronal cultures or brain tissue.*

      ->Endogenous Co-IP has been tried in E18 brain tissue. One experiment using brain tissue demonstrated co-immunoprecipitation of endogenous Sec22b and E-Syt2. Unfortunately, repetitions of this experiment failed due to high background in negative control (naïve Rabbit IgG). We agree with the reviewer that this data is worth trying again. We will carry out this co-immunoprecipitation experiment from cultured neurons to answer the reviewer’s request.

      The authors do show some evidence regarding the complex in neuronal cells using PLA (proximity ligation assay, figure 2) or super resolution microscopy, however, these findings should be corroborated by stronger findings targeting interaction and not based on simple proximity.

      ->We agree with this reviewer that PLA is limited in demonstrating the occurrence of a protein complex. We would like to stress that we have used PLA complementarily to immunoprecipitation and that we already have shown STED super-resolution data (Figure 3). In order to strengthen the STED data, we will include more details in the figure, as a supplementary movie and a supplementary spreadsheet with the quantification of the distance between the E-Syt2/Sec22b clusters to the plasma membrane stained using WGA. The STED data demonstrate that 50% of the clusters are closer than 33.6nm to the plasma membrane, a distance in the range of ER-PM contact sites.

      A similar critique regards the experiments using RNA-interference of Figure 4. Performing loss-of-function experiments in neuronal cultures would strengthen and complement the results obtained via over-expression approaches shown in Figure 5.

      ->The loss-of-function experiments in neuronal cultures using siRNA were attempted unsuccessfully. The three E-Syts have largely different cDNA sequences thus three distinct siRNAs must be transfected in order to silence all three simultaneously. This is quite challenging in neuronal cultures and we were never able to get strong silencing of the three E-Syts. In the following points, we plan to carry out further experiments using expression of a fragment of Sec22b (Longin domain). We are confident that this is a better approach to demonstrate the importance of Sec22b/E-Syt interaction.


      *Given that the authors have already in place all the necessary technology for the suggested biochemical and morphological-related experiments, these could be carried out swiftly within 3-4 months.

      **Minor comments:**

      The manuscript is really technical and at times tough to follow; it could benefit from key sentences to better guide the reader, particularly if not coming from the specialist field, in the appreciation of the experiments and results.

      Authors should submit the manuscript to a severe round of proofreading. There are several inconsistencies and sometimes what looks like internal comments: i.e. in the methods "STED Missing" or the fact that "LTP" is used everywhere but not defined and considering that the targeted audience is most likely neuroscience-based could easily lead to confusion.

      *

      ->We fully agree with this reviewer and apologize for leaving behind such errors. We will carefully proofread the revised ms.

      *The experiments appear to have been repeated a sufficient number of times and the statistics seem adequate. It would be advisable to show in dot-plots the findings rather than in bar graphs all findings and not just the morphometrics-relative ones.

      *

      ->We will modify the figures according to this reviewer’s suggestion.

      Reviewer #1 (Significance (Required)):

      *This work closely follows the excellent previous work from the Galli laboratory. As such, it is mostly incremental from a technical perspective and does not present particularly novel findings. An interesting aspect would be in addressing directly the influence of the described interactions in the lipid transfer between ER and the plasma membrane but in that sense the manuscript falls short. Although it is to be appreciated the functional readouts in terms of neuritogenesis, in the present state the manuscript features findings suitable for a very specific audience.

      I believe that the appropriate audience for the present manuscript lies within the neuroscientific community interested in development, specifically neuritogenesis, and/or membrane dynamics. Additionally, it might be interesting also for researchers outside of the neuroscience community and interested in the dynamics between ER and plasma membrane.

      *

      ->We are happy to read the comments of this reviewer. Nevertheless, we would like to stress the importance of deciphering precise molecular mechanisms in any biological process. Here, we are the first to demonstrate an interaction between lipid-transfer proteins E-Syts and ER v-SNARE Sec22b. As an example, the molecular mechanism connecting synaptic SNAREs and synaptotagmin has been the topic of more than 500 publications since seminal articles in the early 1990’s. We think that the first article linking E-Syts to SNAREs cannot be considered as a mere increment from our previous work.

      The activity of E-Syts to transfer lipids in vitro has been well established __(1–3) In addition, recent work by the De Camilli lab using Origami showed that reducing the distance between liposomes enhanced the lipid transfer mediated by E-Syt2 (3). Therefore, we did not carry out experiments such as combining SNAREs and E-Syt2 in artificial membranes in vitro because we considered that there would not be much more to demonstrate than what has already been done. Furthermore, we considered the experiments in cells, particularly neurons, much more critical at this point. Demonstrating transfer of glycerophospholipid between ER and PM in cells cannot be performed like other lipids’ transfer at other membrane interfaces for the following reasons: phospholipids are very abundant (4) and they are not modified upon transfer (1)__, there are no specific dyes to detect glycerophospholipids (unlike phosphoinositides), and ER and PM are too close to distinguish if a glycerophospholipid is in one or the other membrane. Such a challenging experiment would require the ability to setup a specific biochemical assay circumventing these constraints. We think that this is out of the scope of the present study focused on the role of E-Syt/Sec22b complex.

      Nevertheless, in order to get further insights on this question, we will express WT and mutant E-Syt2, purify the PM using the protocol of Figure 4 in Saheki et al __(1)__, followed by lipidomics analysis. We hope that this approach further supports our idea that E-Syts mediate an important lipid transfer mechanism towards the PM.

      * Keywords regarding my expertise: Molecular and Cellular Neuroscience, Morphometrics, Dendrite, Neurons, Dendritogenesis, Biochemistry, Imaging, Microscopy.


      __

      Reviewer #2 (Evidence, reproducibility and clarity (Required)): *This manuscript identifies and characterises a novel interaction between E-Syts and Sec22b and demonstrates that lipid transfer between the ER and PM contributes to the development of filopodia and neuronal expansion. This interaction with E-Syt2 occurs through the Longin domain of Sec22b Sec22b association. The authors suggest a continuum with further interactions with syntaxin1, that mediates neurite outgrowth. Overall I find this study very interesting and convincing. The experimental analysis is well carried out and the claims are well aligned with their results.

      I only have minor issues:

      Figure 1. Some of the western blots have several bands and it is difficult to know which band is the relevant one. They should be indicated in the fig panel. Further panel E and F are barely readable and should be redrawn with the appropriate line and font size.*

      ->We will make the changes requested by this reviewer in Figure 1.

      • *

      Figure 2: is there a difference between the number of dots in axons and dendrites? Can the author elaborate on this aspect as it is not clear from the image presented.

      ->We could not combine PLA with further staining of MAP2 and TAU. Indeed, to perform PLA, neurons are already double labelled to detect the proteins of interest. At the stage of the neurons used in this study, both axons and dendrites are growing. Therefore, we did not invest in distinguishing between axons and dendrites. Because growth cones are known to be the major sites of membrane growth, we instead distinguished dots within neurites and in growth cones. We will make the other changes requested by this reviewer in Figure 2.

      Figure 7: statistical analysis should be indicated from the BoNT/A and BoNT/C as BoNT/A represent an appropriate control cleaving SNAP25 but not Syntaxin.

      ->We agree with this request and we will add statistical analysis as suggested, using BoNT/A as an additional control.

      On top of controlling fusion and neuronal outgrowth, syntaxin has a role in survival and its cleavage leads to neuronal death. Is this pathway mediated by E-Syts interactions?

      ->We have stated in the ms: “Since exposure to BoNT/C1 at high concentrations and for long incubation periods causes degeneration of neurons in culture __(5,6)__, various concentrations and incubation times were tested, and a 4-hour treatment of neurons with 1nM BoNTs was chosen to avoid such deleterious effects.” Accordingly, we did not see any degeneration in our experimental conditions.__ __


      Reviewer #2 (Significance (Required)): This papers identifies the molecular mechanism of neuronal outgrowth. It is highly significant. ->We are very grateful to this reviewer for pointing out the high significance of our article.


      __Reviewer #3 (Evidence, reproducibility and clarity (Required)):

      __*1. The evidence for the claim that the Sec22b/Stx1 complex and E-Syts colocalize in native cells (neurons) and bind in heterologous cells is strong (3 independent lines of evidence: co-immunoprecipitation, Proximity Ligation (PLA) and STED super-resolution microscopy) However, the current title of the paper makes a claim beyond this interaction/proximity, based on evidence that is obtained with E-Syt over-expression in wildtype cells. The physiological relevance of the effects remain elusive with over-expression in wildtype cells only.

      Furthermore, it is plausible that overexpression of membrane binding/bending C2-domains promotes neurite outgrowth and ramifications by a non-specific effect (as shown for copine C2 domains, PMID:25450385 and indirect evidence for synaptotagmins1,2,7).*

      * This issue is especially relevant in the light of the fact that loss of all 3 Extended Synaptotagmins does not affect normal mouse development and viability (PMID: 27399837)

      It would be more appropriate to choose a more descriptive title*

      • *

      ->We agree with this reviewer that the original title may be too strong and are now proposing the following, more descriptive title:

      Role of the Sec22b/E-Syt complex in neurite growth and ramification

      We are fully aware that proteins harbouring C2 domains could potentially promote non-specific effects when overexpressed. However, we do not think this is the case here because none of the morphological effects of E-Syt2 expression in neurons and HeLa cells were reproduced by mutants lacking the SMP or the membrane-anchoring domains. Based on work on Copine __(7)__, a cytosolic protein, E-Syt2 still containing 3xC2 domains but lacking the membrane-anchoring domain should have shown a morphological effect if non-specific binding to phosphoinositides was the mechanism of action. We will discuss this point in the ms.


      • The evidence for the working model that the morphological effects of E-Syt2 depends on the Sec22b/Stx1 complex is not strong. Although plausible, the positive effect on neurite outgrowth (E-Syt2 overexpression) and the negative effects (inhibition by Stx1 cleaveage, Sec22b-Longin or Sec22b extended linker expression) may in fact be independent

        The evidence could be strengthened by PLA measurements in neurons over-expressing Myc-E-Syt2 and Sec22b to assess MSC density. It is predicted that in both conditions, MCS density increases. MCS density by PLA measurements could also be performed in Sec22b-P33 and DLongin overexpressed and BoNT/C1 treated neurons. According to the model, the number of MCS should go down. This is of special interest for BoNT/C1 treatment, as it is important to show that the altered morphology is not purely caused by a pre-state of degeneration that is known to be induced by BoNT/C1. In addition, EM measurements of ER-PM distances might provide an independent line of evidence.*

      ->We agree with this reviewer that additional experiments could strengthen the description of the molecular mechanism. To this end, we will carry out the following experiments:

      1/Co-immunoprecipitation experiments of endogenous Syntaxin, Sec22b and E-Syt2 in cells expressing GFP as control or Longin-GFP to demonstrate that expression of the Longin domain perturbs the association of Sec22b with E-Syt2 and Syntaxin.

      2/PLA measuring the association between E-Syt2 and Syntaxin in cells expressing GFP as control or Longin-GFP to demonstrate that expression of the Longin domain perturbs the association between E-Syt2 and Syntaxin using a complementary approach.


      Unfortunately, membrane-associated, BoNTC1-cleaved syntaxin corresponds to a short fragment undetectable by available antibodies whereas the fragment detected by the antibody after BoNTC1 cleavage lacks the transmembrane domain (Figure 7a). Therefore, we cannot perform PLA in BoNTC1-treated neurons.


      We are confident that further exploring the mechanism of action of the Longin domain, together with the data already in the ms, will make it very clear that the morphological effects of E-Syt2 depends on the Sec22b/Stx1 complex.



        • Link between neurite outgrowth and lipid transfer is weak. The authors argue that functional E-syt/Sec22b/Stx interaction is important for neurite outgrowth by mediating lipid transfer. The only line of evidence they provide is the absence of outgrowth effects in E-syt mutants lacking SMP or membrane spanning domains. However, from the data it is unclear whether these mutants are correctly folded, expressed and/or localized. Additional ICC stainings of the mutants in neurons are necessary to drive this point home. *
      • *

      ->The mutants and siRNA have been already used and validated in Giordano et al. 2013 __(8)__, therefore we did not carry out experiments aiming at basic characterization of these reagents. To answer this request, we will show images of the subcellular localization by ICC of WT and mutant E-Syt2 in the revised Figure 6 or in a Supplementary Figure.


      In addition, the authors might make the link between neurite outgrowth and lipid transfer stronger by examining PM lipid levels and distribution in control, Myc-E-Syt2 and E-Syt2 mutant neurons.

      ->We agree with this reviewer that this question is of high relevance. In order to answer this request, we will express WT and mutant E-Syt2, purify the PM using the protocol of Figure 4 in Saheki et al __(1)__, followed by lipidomics analysis. We hope that this approach further supports our idea that E-Syts mediate an important lipid transfer mechanism towards the PM.

        • There is no clear evidence that E-syt first binds to Sec22b, after which Stx1 leaves SNAP25 and joins the interaction. This should be indicated as speculation.

        * ->We will make it clear that our model in Figure 9 is a hypothetical model.

      • An apparent discrepancy exists between the TKD E-syts effects (i.e. reduced MSC density, Fig 4) and the lack of neurite outgrowth defects in TKO E-syts. According to the proposed model, the levels of E-syt correlate with the number of MSCs and thereby neurite outgrowth. Furthermore, to knock down E-Syts, single siRNAs against the three E-syts were used in Fig4. Off-target effects are not controlled in this approach. Using multiple siRNAs and/or siRNA resistant rescues would be required for robust conclusions.

        *

      ->The mutants and siRNA have already been used and validated in Giordano et al. 2013 __(8)__, therefore we did not carry out experiments aiming at basic characterization of these reagents. In addition, we would like to stress the complexity of carrying out a rescue experiment of a triple KD of proteins.

      Statistical analysis is incomplete. It is not clear whether statistical assumptions (e.g. normal distribution) were checked before performing the tests, and whether non-parametric alternatives where used if assumptions were not met.


      ->We thank this reviewer for making this important alert. We would like to stress that we have always checked whether samples followed the normal distribution and made non-parametric tests__. We will include this comment in the methods.__

      In Fig4, a T-test is used between multiple groups. This test can only be used when comparing two groups. Number of (independent) measurements is not clear in Fig1, 2, 4

      ->In all the figure legends the number of repetitions is specified


      All figures: display all individual data points in all bar graphs (as shown in 5c)

      *

      *

      \*Minor comments:**

      1. Inconsistencies on distances in model. Syts are enlongated proteins and thought to be found in MSCs of ~20 nm (Fernandez-Busnadiego, 2015). Trans-SNARE complexes start to interact when the distance between membranes is ~8 nm (Liu, 2007). In the introduction, the authors suggest that incomplete zippering might occur between Stx and Sec22b, resulting in a distance between 10 and 20 nm, which would allow E-Syt localization. In the discussion, however, the authors suggest a model where Sec22b/Stx interaction is important to bring the membranes in ~10 nm distance to enhance LTP activity. Proof for either model is lacking. Please clarify.*

      Fig1A: Please clarify the multiple bands? for Stx3 (anti-eGFP).

      • *

      ->These additional bands are recognised by the anti-GFP antibody, the tag being N-terminal, thus they represent proteolytic fragments. We consistently observe these in our experiments.

      Fig2: There is no size marker for panels C1-C6

      • *

      ->We will make the appropriate correction.

      Fig3: Both proteins seem to show a diffuse pattern. Please specify the validity of measuring average distance. A higher magnification zoom of staining pattern in the growth cone and visualization of the calculation could benefit interpretation.

      • *

      ->We agree with this reviewer that Figure 3 was not optimal to show all the extent of our STED data. In order to strengthen this part, we will include more details in both the figure and as a supplementary movie and supplementary spreadsheet with the quantification of the distance between the E-Syt2/Sec22b clusters to the plasma membrane stained using WGA. The STED data demonstrate that 50% of the clusters are closer than 33.6nm to the plasma membrane, a distance in the range of ER-PM contact sites.

      • E-Syt2 and E-Syt3 are used interchangeably throughout the manuscript and E-Syt1 is left out completely. It would help the reader if the authors could elaborate on their interpretation on the similarities and differences in structure and functionality between the three E-Syts.
      1. Why is there a red line in Fig 7b?*

      __->We added the red line to highlight the shift of SNAP25 band in BoNTA samples. If misleading, it can be removed

      Reviewer #3 (Significance (Required)):__

      A growing body of literature recognizes the importance of close proximities between membranes, facilitating direct interaction between organelles (Scorrano et al., 2019). Membrane Contact Sites (MCSs) are shown to be important for a wide range of cellular functions, such as lipid and calcium transfer. E-Syts have been recognized as one of the key players in neuronal MCSs, mediating lipid transport (Fernández-Busnadiego et al., 2015). A study published in 2014 by the authors of the current study revealed another two proteins important for MSCs in neurons (Petkovic et al., 2014). ER protein Sec22b and PM SNARE Syntaxin1 were shown to form a non-fusogenic trans-SNARE complex, important for lipid-transfer mediated neurite outgrowth. Gallo and colleagues have now provided important new evidence that these two components (E-Syts and Stx1/Sec22b) are together and may work together at MSCs.

      ->We thank this reviewer for stressing the importance of our article and agree with the conclusion of __Fernández-Busnadiego et al. (9) on E-Syts being one of the key players in neuronal MCSs, mediating lipid transport. We think that our work is a further key piece of evidence in the demonstration of the importance of E-Syts in neuronal development.__

      Bibliography

      Saheki Y, Bian X, Schauder CM, Sawaki Y, Surma MA, Klose C, et al. Control of plasma membrane lipid homeostasis by the extended synaptotagmins. Nat Cell Biol. 2016 Apr 11;18(5):504–515. Yu H, Liu Y, Gulbranson DR, Paine A, Rathore SS, Shen J. Extended synaptotagmins are Ca2+-dependent lipid transfer proteins at membrane contact sites. Proc Natl Acad Sci USA. 2016 Apr 19;113(16):4362–4367. Bian X, Zhang Z, Xiong Q, De Camilli P, Lin C. A programmable DNA-origami platform for studying lipid transfer between bilayers. Nat Chem Biol. 2019 Jul 18;15(8):830–837. Alberts B, Johnson A, Lewis J, Raff M. The lipid bilayer. Molecular Biology of …. 2002; Osen-Sand A, Staple JK, Naldi E, Schiavo G, Rossetto O, Petitpierre S, et al. Common and distinct fusion proteins in axonal growth and transmitter release. J Comp Neurol. 1996 Apr 1;367(2):222–234. Igarashi M, Kozaki S, Terakawa S, Kawano S, Ide C, Komiya Y. Growth cone collapse and inhibition of neurite growth by Botulinum neurotoxin C1: a t-SNARE is involved in axonal growth. J Cell Biol. 1996 Jul;134(1):205–215. Park N, Yoo JC, Lee Y-S, Choi HY, Hong S-G, Hwang EM, et al. Copine1 C2 domains have a critical calcium-independent role in the neuronal differentiation of hippocampal progenitor HiB5 cells. Biochem Biophys Res Commun. 2014 Nov 7;454(1):228–233. Giordano F, Saheki Y, Idevall-Hagren O, Colombo SF. PI (4, 5) P2-dependent and Ca2+-regulated ER-PM interactions mediated by the extended synaptotagmins. Cell. 2013; Fernández-Busnadiego R, Saheki Y, De Camilli P. Three-dimensional architecture of extended synaptotagmin-mediated endoplasmic reticulum-plasma membrane contact sites. Proc Natl Acad Sci USA. 2015 Apr 21;112(16):E2004–13.

    1. Environmental: safety of where you live, quality of the programs and services available (or unavailable) to you Familial: how loving and caring your family may be, how much time and support your family offers each other Within-person: your temperament, abilities (and inabilities), skills and characteristics that make you who you are

      I think a lot of the time when we think of resilency we think of the individual person. What are they doing to impact how they approach the challenges and stretches in life? How much of it is attributed to personality?

      There are two other factors that our out of the control of the child. As educators, we can do something about these two factors. Our hands are not tied and we can be the reason that a child develops some resiliency. Obviously, connecting and collaborating between home and school is preferable. But we only have control over our classrooms and sometimes that has to be enough.

    1. Importance of Resilience

      I appreciate the way Rachel Wagner explains resiliency. I think the rubber band example is a wonderful visual to help explain how every person has different events in their life that may "stretch" them and resilience is being able to healthily cope with the those stretches.

      I also appreciate the tips that she discusses in the video. The first thing she brings up is having positive relationships. As educators, we know that building relationships is a key our and their success. I like how Rachel mentions that these positive relationships can be small moments. It does not need to be a grandiose thing but a simple interaction. Rachel also mentions the important to encourage students to figure out how to approach different challenges they may face. An important aspect of this is giving children the space to figure it out. As educators, these are important things to keep in mind.

      Rachel also brings the concept of not being able to pour from an empty cup. Before we, as adults, can help our students become more resilient we have to be in a space to give ourselves. This involves setting our own boundaries and practicing resiliency ourselves.

    1. Note: This rebuttal was posted by the corresponding author to Review Commons. Content has not been altered except for formatting.

      Learn more at Review Commons


      Reply to the reviewers

      Authors’ response to reviewers for manuscript “Bacterial killing by complement requires direct anchoring of Membrane Attack Complex precursor C5b-7” (reference #RC-2019-00125)

      Our manuscript entitled “Bacterial killing by complement requires direct anchoring of Membrane Attack Complex precursor C5b-7” has been reviewed by Review Commons. We thank the referees for their interest in our study and are very pleased that the referees consider our findings novel, important and well-designed. Based on the comments given by the referees, we have revised our manuscript and have included two new experimental figures:

      -Experimental data validating that our gating strategy with Sytox blue correlates well with bacterial killing on plate (Fig S1-B)

      -Experimental data validating that non-bactericidal MAC complexes damage the bacterial OM (Fig. S1-C).

      In the response letter below, we respond to the comments raised by the reviewers and explain how we have revised our paper accordingly. All changes into the revised manuscript are clearly highlighted in yellow.

      POINT-TO-POINT REPLY

      Reviewer #1

      (Evidence, reproducibility and clarity (Required)): The paper by Doorduijn et al. addresses a question rarely touched upon in modern studies of the complement system, namely the stability and time-resolved functions of complement component. It extends two earlier reports from the same laboratory, however, with a clear, novel point concerning especially the function of C7. The study embodies several techniques and modes of investigation. From these experiments, the paper contributes significantly to our understanding of the MAC complex is formed and why some bacteria escape this host defense mechanism. Over all the study is very well performed and written. I have only a few major comments.

      Reviewer #1 raises 3 points:

      POINT 1. The AFM pictures shown in Fig. 6D are of outstanding quality. However, it is a disappointment that the outcome of complement incubation was shown only for a complement-resistant E. coli strain. Would it be possible to show the location on the bacterial surface of MAC complexes, or holes, on a complement-susceptible strains? Comparing the visual outcome for such bacteria with locally formed MAC versus C7 replenished would be quite interesting and perhaps important.

      ANSWER 1. Since Fig. 6D represents AFM images of MAC on complement-susceptible E. coli bacteria, we assume that the reviewer is asking why we did not perform AFM experiments on complement-resistant strains? To address this question, it is important to note that we have thus far not succeeded in robustly visualizing MAC complexes under conditions at which bacteria were not killed by MAC complexes (Heesterbeek et al., EMBO J, 2019). While non-bactericidal MAC complexes are present on the bacterial surface as demonstrated with C9 deposition by flow cytometry, we hypothesize that they are not well inserted into the membrane (demonstrated by sensitivity to trypsin) and therefore difficult to resolve by AFM. This is consistent with previous AFM experiments on related pore-forming proteins (Leung et al, 2014, 2017), in which inserted pores were readily detected on supported lipid bilayers, but mobile, non-inserted pores were harder to resolve due to the invasiveness of the AFM measurement and/or insufficient temporal resolution. In the revised manuscript we now better clarify this in line 298-301.

      POINT 2. The flow cytometric analysis of bacterial killing is somewhat simplistic. Usually, staining of BOTH live and dead bacteria is performed. This permits better gating of the relevant populations. Specifically, the gating seems to fit the population in Fig. S1 only poorly, with the gate in some cases simply dividing what otherwise appears to uniform population ("C9 at t=0")

      ANSWER 2. In the revised manuscript, we have now included additional data demonstrating that our gating strategy with Sytox blue correlates well with bacterial killing on plate (new Fig S1-B referred to in line 78-79, 92-93, 96-98 and Supplementals text line 21-24 shows cfu data for Sytox data of Fig 2D). These data correspond with our earlier findings showing that cells gated to be positive for Sytox blue are indeed the relevant population of dead cells (Heesterbeek, EMBO J, 2019). We disagree with the reviewer that the use of a ‘live’ stain is of added value here. Because the outer membrane of Gram-negative bacteria is also a permeability barrier for ‘live’ stains like Syto9, MAC-dependent outer membrane perforation also results in increase in ‘live’ stain during the process of bacterial lysis (also described in Stiefel et al, BMC Microbiol, 2015 PMID: 25881030). We have therefore chosen to only use the Sytox stain in this study as this is a very reliable marker for killing.

      POINT 3. The cited literature is, in general, pertinent and comprehensive. I was surprised, however, that none of the many contributions to field of MAC formation by AF Esser was cited. For instance, the studies over C9 conformation (PMID: 2475785) seem not far away in topic from some of the points raised in the present paper.

      ANSWER 3. The reviewer is correct that the work of AF Esser has indeed focused on the contribution of C9 and C9 polymerization to the lytic activity of the MAC pore. In the revised manuscript, we have therefore now included some of the work done by AF Esser (references 34, 36 and 37) and have discussed this in our discussion (line 305-309). However, it is important to note that much of the work on the importance of C9 polymerization by AF Esser has been performed erythrocytes and single-membrane particles (also the suggested paper by the reviewer). Translation of these studies to the role of C9 conformation and polymerization on bacterial killing is therefore limited, although it does provide clues to what differences might cause the discrepancy observed between lysis of erythrocytes and bacterial killing by MAC pores.

      Reviewer #1 (Significance (Required)): Insight into the concept of locally formed MAC complexes is lacking and the paper clearly adds novel and quantitative data to this point. The paper probably mostly reaches out to an audience interested in the complement system and researchers interested in large protein complexes with conformational changes as part of their function. My own interest lies with complement-mediated protection against bacteria with a special focus on pattern recognition and protein-bacterial surface interactions.

      Reviewer #2

      (Evidence, reproducibility and clarity (Required)): Doorduijn et al. present a study illustrating the importance of rapid C7 interaction with C5b6 for MAC-dependent killing of complement sensitive bacteria. The absence of direct C7 interaction results in a MAC which i) doesn't kill the bacteria, and ii) is sensitive to trypsin. The authors have step by step investigated this issue by using common in vitro-methods with different strains of bacteria, serum, and/or purified complement proteins. Bacterial killing is evaluated by sytox blue influx in flow cytometry. I like this work. The experimental strategy is sound, and the conclusions are convincing are based on the presented data. The data and the methods presented in such a way that they can be reproduced. I have no concerns regarding the design, execution or conclusions.

      Reviewer #2 RAISES 3 POINTS

      POINT 1. My only criticism is on the number of replicates and following statistical analysis: • Overall, the experiments are conducted only three times. With the, in general, large differenced seen between the condition, this may still be acceptable. However, the statistic testing using only N=3 is of low value.

      ANSWER 1. As the reviewer pointed out, with these in vitro studies where the experimental conditions are highly controlled it is common practice to perform three independent experiments when the differences are large.

      POINT 2. The authors have sometimes used paired testing, and sometimes unpaired. For example, Fig. 5A-B is based on paired testing, whereas data in C, which are based on A-B, is tested using unpaired testing. Why so is unclear to me.

      ANSWER 2. We thank the reviewer for this comment, as also for Fig. 5C we should have used a paired analysis, so we have done so accordingly in the revised manuscript (line 725-726).

      POINT 3. Further on in Fig. 5 A-B., ANOVA with Tukey multiple comparison tests is used, which implements testing between all conditions; still, only significance is reported for blue vs. red. If the intention was to only test red vs. blue, a t-test would be better.

      ANSWER 3. As with the previous comment, we have now performed a paired t-test since we only intended to compare C7 at t=0 vs C7 at t=60 in Fig. 5A-B (line 725-726). Moreover, for consistency in our statistical analyses we’ve also applied this to Fig. 3C (line 706-707).

      Reviewer #2 (Significance (Required)): As far as I understand, the presented data is of high significance for the conceptual understanding of the buildup of MAC for bacterial killing on Gram-negative bacteria. I work partly with complement but is not an expert on the terminal pathway.

      Reviewer #3

      (Evidence, reproducibility and clarity (Required)): The study is a follow-up on the paper the same group of scientists published in EMBO J last year. That paper showed that rapid interaction between C5b6 and C7 is necessary for effective killing of Gram negative bacteria. The follow-up this paper makes is to make that case for a series of E. coli strains, showing as part of this that strains of clinical isolate E. coli resistant to complement attack prevent the rapid C5b6-C7 interaction. The story goes that C5 convertase engagement on the surface of targeted bacteria is the necessary context for effective C5>C5b conversion and thence interaction with C6 and C7. The rapid interaction with C7 is necessary because it prevents release/shedding of C5b6 from the bacterial cell surface. Overall, the conclusions seem justified - that C5b6 interaction with C7 stabilises its interaction with the surface and is needed to prevent C5b6 shedding. But this observation needs a mechanical or biophysical framework to be understood properly.

      Reviewer #3 RAISES 5 POINTS

      POINT 1. The authors do not observe non-bactericidal MAC pores/non-lytic MAC by AFM and so I think in this study there is no evidence for their existence. Their depiction in Figure 8b is therefore misleading and I think should be deleted. Indeed, the authors do not know what the structure of the non-bactericidal MAC pores could be, so depicting them in this specific way isn't appropriate. They have no idea what they might be like, if they exist.

      ANSWER 1. We agree with the referee that we do not know the structure of a non-bactericidal MAC pore, and have therefore deleted the speculative structures in Fig. 8B (explained in line 784-785). Although we have no structural information, we do think that non-bactericidal MAC pores exist and our revised manuscript now includes new data to better explain this (Fig S1-C). While our initial manuscript showed that a delayed interaction between C5b6 and C7 results in MAC complexes that cannot perturb the bacterial inner membrane, we now show that these MAC complexes effectively damage the outer membrane (evidenced by leakage of mCherry from the periplasmic space (Fig. S1-C, explained in line 97-98 and Supplementals text line 24-31). This leads us to conclude that there are pores formed in the outer membrane that are not capable of damaging the inner membrane. We think that within this context we can name these ‘non-bactericidal MAC pores’.

      POINT 2. This brings me to another point: it is really unclear to me from this study how the authors envisage the inner bacterial membrane be damaged by MAC attack. Do MAC pores formed in the OM deliver MAC components to the IM? Or what happens - is the damage to the IM indirect? The reason why this is relevant to the possibility of non-bactericidal MAC pores is that it could be these are inserted just like bactericidal pores into the OM but the IM attack is deficient in some way.

      ANSWER 2. Although we agree with the reviewer that exact mechanism by which MAC pores perturb the inner membrane is unanswered, we think this is beyond the scope of this paper which mainly deals with the time-resolved functions of MAC assembly. However, to meet the referees’ critique, we have now more clearly addressed this question in our discussion and speculate on several mechanisms by which the MAC pore could induce bacterial inner membrane damage (line 277 - 288). In short, we hypothesize that OM damage could indirectly trigger IM damage by affecting regulation of osmosis, overall cell envelope stability and/or envelope stress.

      POINT 3. (Significance (Required)). I am intrigued by the difference between MAC assembly on erythrocytes and bacteria. What do the authors believe to be the basis of this difference? It would help understanding of the significance of their work if they could make this clear. Without this kind of attempted explanation the results seem phenomenological - an observation has been made but why this observation occurs, what the important environmental difference is between erythrocyte membranes and the outer membranes of Gram negative bacteria is not addressed. I am looking for some kind of biophysical explanation - specific lipid properties, for example.

      ANSWER 3. We agree that this is intriguing and in our revised manuscript we have included different hypotheses on why MAC assembly on erythrocytes and bacteria could be different. Although differences in composition between the erythrocyte membrane and outer membrane can definitely play a role, our data suggest that the difference is mainly a consequence of the fact that Gram-negative bacteria have two membranes (the outer and inner membrane). In the revised manuscript, the newly added figure (Fig S1-C) supports this, since this figure reveals that MAC pores generated from C5b6 that is generated in the absence of C7 can still damage the bacterial OM. However, despite observing OM damage by measuring leakage of a periplasmic protein, this does not lead to bacterial killing and IM damage. Since we here observe that rapid interaction between C5b6 and C7 is required for bacterial killing and IM damage, we think that efficient anchoring of C5b-7 is primarily relevant in damaging the bacterial IM and subsequently causing bacterial cell death. Finally, we have also mentioned this more specifically in the discussion of the revised manuscript (line 277-288).

      POINT 4. Related, at the end of the Results section the authors say "Altogether, these data indicate that complement-resistant E. coli can prevent complement-dependent killing by MAC pores by preventing efficient anchoring of C5b-234 7 and insertion of MAC pores into the bacterial cell envelope." My immediate response was: 'How? The Discussion needs to consider this.' But it doesn't.

      ANSWER 4. In the revised manuscript, we now explain this more extensively (line 322 – 326). In short, we hypothesize that the composition of the OM, mostly in terms of capsular polysaccharides and lipopolysaccharides, could affect this. We have added additional references supporting the role of these components in complement resistance in multiple Gram-negative species (reference 45-48 and 50).

      POINT 5. I was confused by the term "metastable lipophilic domain" at line 262 on page 10. Do the authors mean the MACPF domain?

      ANSWER 5. We have now more explicitly named this in our discussion as being the MACPF domain and have further elaborated what we meant by metastable (line 263-267).

    1. Emily Messer ENG 491 Bleak House Installment Eleven 02/02/2020 Bleak House Installment Eleven and its Thematic Relevance

      Installment eleven of Bleak House by Charles Dickens begins with chapter thirty-three and ends with chapter thirty-five of forty-eight chapters total. There are twenty installments which were released monthly, making Bleak House an almost two-year endeavor. Situated between a case of spontaneous combustion in installment ten and Esther learning the true identity of her mother in installment twelve, installment eleven runs along a track of eventful and emotion packed installments, just after the middle of the novel. Though it does occur more than halfway through the story, a few of the events in installment eleven set up the later events of the novel, all the way up until the ending. Esther’s illness and recovery, which is the driving force of chapter 35, influence multiple plotlines that lie at the heart Bleak House. Similarly, the confession of love for Mr. Woodcourt, which also takes place at the close of chapter 35, is integral to the ending of Bleak House and its emotional weight. Installment eleven begins with the omniscient narrator describing the court directly after Krooks dies of spontaneous combustion. We learn that Krooks was Mrs. Smallweed’s brother, and therefore the Smallweeds want to inherit Krook’s belongings. We remember that Krooks had a stack of Nemo’s letters and wonder if any may have survived. We also meet the Bagnet family and their learn connection to George Rouncewell while Mr. Tulkhinghorn attempts to collect Mr. George’s debt. Then we switch back to Esther’s narrative, where we learn of her illness and her recovery with resulting scars, her absence from Ada, and an admission of her love for Mr. Woodcourt. Esther writes, “And now I must part with a little secret I have thus far tried to keep. I had thought, sometimes, that Mr. Wood Court loved me; and that if he had been richer, he would perhaps have told me that he loved me, before he went away. I had thought, sometimes, that if he had done so, I should have been glad of it. “(pg. 570) The illness that Esther has contacted, which is decidedly smallpox, was passed down from Jo, to Charley, to her. We learn of all three of these illnesses in installment number ten, and their significance and thematic importance comes to light in installment eleven. Jo, who is poor and resides in Tom All Alone’s, a very impoverished and dirty neighborhood in London, gets the disease first. Then Charley, who is still a poor girl and one of the lower-class characters in Bleak House contracts it from caring for Jo. Then, Esther falls ill after caring for both Jo and Charley. From this, there are two thematic takeaways. First, there is the infection of disease moving its way up the social class but ends with the class just below Ada: people whose perceived birth is low ranking. The disease is not allowed to continue spreading into the upper classed members of the character list. This could be read as a social critique of the idea of birth ranks, calling out how lower classed members of society are seen as a kind of self-contained infection. Secondly, this could fit into the novel’s larger conception of charity. Esther and Charley care for Jo out of compassion: a kind of charity. However, this charitable act winds up giving them smallpox. Similar to the portrayal of Mrs. Jellyby’s neglect at the hands of charity and Mr. Jarndyce’s inability to recognize thanks for his charity, the contraction of disease as a result of charity might be another way Dickens conveys that there is no good or pure form of charity - at least not without consequence. In regard to the effects of her illness, Esther writes “” I had heard my Ada crying at the door day and night I had heard her calling to me that I was cruel and did not love her… But I had only said when I could speak, ‘Never my sweet girl never!’ And I had over and over again reminded Charley that she was to keep my darling from the room whether I lived or died. Charley had been true to me in that time of need, and with her little hand and her great heart had kept the door fast.” Beyond the illness itself, the scars that result from the illness do quite a bit of work for the overall plot going forward from installment eleven. In installment twelve, Lady Dedlock reveals to Esther that she is, in fact, her mother. However, no kind of mother-daughter relationship results from that conversation. This could, in part, be explained by Esther’s complete facial change from the scarring that comes from the illness. Esther’s familiarity to Lady Dedlock is tainted by the scarring, and therefore the scarring becomes a symbol for the years of separation and emotional trauma (especially Esther but also somewhat Lady Dedlock as well) endured. Esther’s scars also inform her decision to accept the proposal made to her by Jarndyce in installment number fourteen, for marriage. Esther believes that her beauty has completely left her as a result of the scarring, and when Jarndyce proposes to her, she feels as though that may be her only chance at marriage. Yes, Esther loves Jarndyce almost like a father, but we have never had any inkling that she is romantically interested in Jarndyce. Therefore, when Jarndyce gives Esther a letter asking her to be his wife, we almost expect, or perhaps wish Esther to say something like, “No! I’m a young and beautiful woman and should not give away a chance at a fulfilling relationship to be the pet of an old man!” However, Esther does not think she is beautiful anymore, not after the scars. When speaking of her scars, Esther writes, “I have thought of this very often. I was now certain of it. I could thank God that it was not a shock to me now. I called Charley back; and when she came – at first pretending to smile, but as she drew nearer to me looking grieved – I took her in my arms and said, ‘it matters very little Charley I hope I can do without my old face very well,’” (pg. 558) Another revelation that comes with installment eleven is the admission of love between Alan Woodcourt and Esther. This has been heavily hinted at, but never outright said until this point. We have already had hurdles to a respective relationship introduced earlier in the novel, such as Mrs. Woodcourt's assertion that Allan should not marry a woman who is not of good birth. This, of course, would not apply to Esther after she learns that she is a Deadlock, but at the time is a clear threat. There is also Mr. Woodcourt’s prolonged absence from Bleak House at the hands of his work as a physician. However, none of the other threats seem as concrete and final as the threat that Woodcourt simply could not be with Esther after she has lost her beauty. This thought leaves the reader with a sadness, which is a perfect setup for the close of the novel where Jarndyce releases Esther from her betrothment and allows her to marry Woodcourt. The emotional weight of learning that Esther, a beloved narrator, can finally be happy with a man she could truly love as a husband is only possible through the many chapters of concealment which lead to the admission at the end of instalment eleven along with the seemingly assured end of any hope with the dramatic change in Esther’s appearance. In reference to how her change in appearance will affect a potential relationship with Woodcourt, Esther writes, “What should I have suffered, if I had had to write to him, and tell him that the poor face he had known as mine was quite gone for me, and then I freely released him from his bondage to one and he had never seen!” (pg. 570) Thematically, the illness and scarring work together to draw parallels between infection and poverty, charity and consequences, emotional scars and physical markings, and loss of beauty and strength of love. The preceding events of the installment, such as the introduction of the Bagnet family and the Smallweed’s connection to Krooks have their importance in the plot of the novel, but the larger thematic overtones of those sections pale in comparison to the importance of the illness. While the illness does first appear in an earlier installment, the resulting scarring and connection to the romance between Woodcourt and Esther does not appear until installment eleven, making this thematic and symbolic groundwork anchored in the eleventh installment.

    2. Allan Woodcourt  (VWKHUEsther

      This is where the potential romantic connect between Allan Woodcourt and Esther is brought to light. The interesting part is that this is also the installment where Woodcourt leaves the country on a voyage to China and India on a ship. We haven't seen very much of him before this, but here we are saying goodbye to him. His exit is almost necessary for the future relationship between Mr. Jarndyce and Esther to even have a chance at beginning -- if Woodcourt had been there all along, through everything (like Esther's illness), Mr. Jarndyce may not have even proposed to Esther in the first place. So, moving his piece off the chessboard of this novel's narrative was significant in itself. We also have hints that Esther's relationship with Woodcourt is more significant than she's letting on.

      Her narrative voice shows she has trouble looking at Woodcourt in an unbiased way; for example, she says things like:

      "I believe -- at least I know -- that he is not rich" as well as "I think -- I mean, he told us -- that he had been in practice three or four years..." (277).

      This obvious attempt to remove herself from the narrative is consistent with her character, but it also shows that her first thought is to portray him the way she subjectively views him, and she must force herself to be more removed.

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      Reply to the reviewers

      We are very grateful to the three reviewers for their useful and constructive comments on our manuscript. All reviewers appreciated that our manuscript provides a good characterization of the KKT2/3 functional domains, especially in solving crystal structures of the KKT2 central domain and revealing the importance of KKT2/3 central domains for their centromere targeting. They also commented that additional experiments (e.g. testing DNA-binding activities using recombinant proteins and examining whether ectopically expressed KKT2 fragments localize at kinetochores transiently) would significantly strengthen the manuscript. In the revised manuscript, we are going to address their comments as follows.

      Reviewer #1:

      From the information presented, it seems like there are only two possibilities to explain the role of the zinc finger domains in directing centromere targeting. First, this could mediate a protein-protein interaction. The authors attempt to assess this using their mass spec experiments, but this does not absolutely rule this out as this interaction may not persist through their purification procedure (low affinity or requires the presence of DNA, such as for a nucleosome).

      Response: We agree with the reviewer’s comment. We will add a sentence to discuss this possibility in the revised manuscript.

      Second, this could reflect direct DNA binding by the zinc finger. Although the existing paper is solid and highlights a role for the zinc finger domains in the localization of these proteins, it would be even better if the authors were to at least assess DNA binding in vitro with their recombinant protein. Comparing its behavior to a well characterized DNA-binding zinc finger protein would be powerful for assessing whether direct DNA binding could be responsible for its centromere localization.

      Response: We have tested DNA-binding activities for the KKT2 central domain from T. brucei, Bodo saltans, and Perkinsela using a fluorescent polarization assay. We tested three different DNA probes (50 bp each) that were fluorescently-labelled: a 50 bp DNA probe from the CIR147 sequence, which is the unit sequence of centromere repeats of several chromosomes in T. brucei (36% GC content), as well as two random DNA sequences of 25% and 74% GC content. We found that the Perkinsela KKT2a central domain binds these three different DNA probes with similar affinities (Kd ~100 nM), suggesting that the Perkinsela KKT2a central domain binds DNA in a sequence-independent manner. Although we have not been able to obtain reliable results for T. brucei and Bodo saltans proteins thus far (due to quenching of fluorescent signals by these proteins), it is likely that the T. brucei KKT2 central domain also binds DNA in a sequence-independent manner given the similarity of the Znf1 structure/sequence among kinetoplastids. This is consistent with the observation that there is no DNA sequence that is commonly found in the centromere of all chromosomes in T. brucei and other kinetoplastids. We are going to add the DNA-binding assay results for the Perkinsela KKT2a central domain in the revised manuscript. We do not feel it is informative to compare the KKT2 Znf1’s behavior to a well characterized DNA-binding zinc finger protein (that binds specific DNA sequence), because Perkinsela KKT2a binds DNA in a sequence-independent manner.

      The code for KKT2 and KKT3 localization is complicated by the multiple regions that contribute to their targeting. This includes both the zinc finger domain that the authors identify here, as well as a second region that appears to act through associations with other constitutive centromere components. Due to this, it feels that there are several aspects of these proteins that are incompletely explored. First, the authors show that the Znf1 mutant in KKT2 localizes apparently normally to centromeres, but is unable to support KKT2 function in chromosome segregation. This suggests that this zinc finger domain could have a separable role in kinetochore function that is distinct from centromere targeting.

      Response: We agree with the reviewer that the mechanism of KKT2 kinetochore localization is complicated because there are at least three distinct domains that contribute to its targeting (Figure 2 in the original manuscript), but we showed that the centromere targeting of the ectopically-expressed KKT2 central domain fragment depends on Znf1 (Figure 6B in the original manuscript). Together with the finding that the Znf1-equivalent domain is essential for the localization of the full length KKT3 protein, we think that a function of the KKT2 Znf1 domain is to promote its centromere localization. In the future, it will be critical to understand the molecular mechanism of how the KKT2 central domain localizes specifically at centromeres.

      Second, although the authors identify these minimal zinc finger regions as sufficient for centromere localization, they do not test whether this behavior depends on the presence of other KKT proteins. This seems like a very important experiment to test whether recruitment of the zinc finger occurs through other factors, or whether it could act directly through binding to DNA or histones.

      Response: We do not have an experimental setup to test whether the centromere localization of KKT2/3 central domains depends on other KKT proteins (i.e. we cannot keep the expression of the central domain to a low level while inducing RNAi constructs at a high level). As an alternative approach, we have been testing the localization dependency of endogenously-tagged full-length KKT2/3 proteins using RNAi against various KKT proteins but our preliminary results have not found any kinetochore protein whose depletion affects the localization of KKT2 or KKT3 at centromeres. Although these results could be explained by inefficient protein depletion, they are consistent with the possibility that KKT2 and KKT3 central domains directly interact with centromere DNA. We could consider adding these data in the revised manuscript, although a significant amount of additional work will be necessary to confirm these results.

      • Based on the description of kinetoplastid centromeres that the authors provide, it is actually unclear to whether these are indeed sequence independent. The authors state that "There is no specific DNA sequence that is common to all centromeres in each organism [Trypanosomes and Leishmania], suggesting that kinetoplastids also determine their kinetochore positions in a sequence-independent manner." However, it remains possible that there are features to this DNA that are responsible for defining the centromere. In principle, enriched clustering of a short motif that may elude sequence comparisons could be responsible for specifying these regions. It would be helpful to use caution with this statement, and I would also encourage the Aikyoshi lab to test this directly in future work, such as using strategies to remove a centromere or alter its position. *

      Response: We agree with the reviewer that we cannot exclude the possibility that there might be an enrichment of a short motif that promotes the localization of kinetochore proteins. We will discuss this possibility in the revised manuscript.

      • It would be helpful to provide a schematic of kinetoplastid kinetochore organization based on their studies to date (possibly in Figure 1) to provide a context for the relationships between the different KKT proteins tested in this paper.*

      Response: While we agree with the referee that a model figure would be helpful, we feel that drawing a model for the overall organization of kinetoplastid kinetochores at this stage could be misleading because we still know very little about it. In fact, our published data (e.g. the microtubule-binding kinetochore protein KKT4 localizes at centromeres throughout the cell cycle and has DNA-binding activities) and our unpublished observations suggest that the design principle of kinetoplastid kinetochores may well be fundamentally different from that of canonical kinetochores in other eukaryotes. We therefore would like to obtain more data before drawing a model of kinetoplastid kinetochores. Instead of a model, we are going to include a summary of localization patterns for kinetoplastid kinetochore proteins in Figure 1 to help orient readers.

      Reviewer #2: The experiments are in general well presented but some could be better controlled: - localization of KKT2 and KKT3 mutants is never verified to be centromeres, we have to believe the dots in the DAPI region are centromeres.

      Response: We have assumed that the KKT2 and KKT3 mutants that had dots very likely localized at centromeres because they behaved similarly to wild-type proteins (i.e. align at metaphase plate in some 2K1N cells and localize at the leading edge of separating chromosomes). We will confirm this assumption by imaging the KKT2/3 mutants with a kinetochore protein marker (e.g. tdTomato-KKT1).

      in some cases mutants are made in full-length (FL) background (viability, sometimes localization), but in other cases only in isolated domains. The former should be done for all assays. This is also important to show that central domain of KKT2 and KKT3 is necessary for localization.

      Response: It is very laborious to create point mutants in full-length background at an endogenous locus. This is why we first tested a number of mutants in our ectopic expression of truncated (for KKT2) or full-length (for KKT3) proteins to identify the most critical mutations, which were subsequently tested in the endogenous context. Although not included in the original manuscript, we have performed an ectopic expression of additional KKT2 mutants (C597A/C600A, C616A/C619A, C624A/C627A, C640A/C643A, and H656A/C660A) in the full-length protein and found that all of them had apparently normal localization pattern, which is consistent with the results we obtained in the endogenous expression experiments (C576A, D622A, and C640A/C643A: Figure 6c in the original manuscript).

      The data of F2 are interpreted to mean that PDB-like domain and middle region get to kinetochores by binding transient KT components, even though KKT2 itself is constitutive. That interpretation would really be strenghthened by showing the KKT2 fragments are now transient also. **

      Response: Our observations suggest that these KKT2 fragments indeed localize at centromeres transiently (from S phase to anaphase). We will confirm this result by imaging with a transiently-localized kinetochore protein, KKT1 tagged with tdTomato, and include in the revised manuscript.

      The paper could do with some attempts to get to this, based on the presented data. For example, does Znf1 bind centromeric DNA, does it bind nucleosomes, is it essential for recruiting the other KKTs, etc.

      Response: As we responded to Reviewer 1, we have found that Perkinsela KKT2a central domain Znf1 has DNA-binding activities. We agree that it will be important to test whether KKT2 binds nucleosomes but it will be necessary for us to reconstitute nucleosomes using recombinant T. brucei histones. It will also be important to test whether KKT2/3 are essential for recruiting other kinetochore proteins but we think that they are beyond the scope of this manuscript.

      Reviewer #3: \*Major Comments:** - No page numbers - this makes it difficult to refer to different parts of the text... *

      Response: We sincerely apologize for the lack of page numbers in the original manuscript. We will add page numbers and line numbers in the revised manuscript.

      Introduction (page 2), fourth-from bottom line: the authors refer here to "regional centromere" but have not defined this term (I assume, as opposed to point-centromeres of budding yeast?). I suggest rephrasing.

      Response: We thank the reviewer for pointing it out. We will rephrase it in the revised manuscript.

      Page 4, bottom: The discussion of KKT2 kinetochore localization brings up a lot or questions. First, can the authors use an assay like yeast two-hybrid to test for pairwise interactions between KKT2 domains and other kinetochore proteins? This could provide direct functional data on the role of these various domains in kinetochore localization.

      Response: Based on the mass spectrometry of immunoprecipitated KKT2 fragments that localized at kinetochores, we are currently trying to identify direct protein-protein interactions between the KKT2 domains and other kinetochore proteins (e.g. does KKT2-DPB directly interact with KKT1, KKT6, or KKT7 proteins?). While we agree that it is important to address these questions, we think that it is beyond the scope of this manuscript because its focus is the characterization of KKT2/3 central domains. As we mentioned in the manuscript, these central domains failed to co-purify with other kinetochore proteins, and the experiment therefore did not give us any clue about how they might localize specifically at centromeres.

      Second, if individual domains are being recruited to kinetochores by their non-constitutive binding partners, wouldn't this be evident if the authors looked at localization at different points in the cell cycle, and/or with dual localization tracking the putative binding partners? Could transient localization of some of the domains explain the intermediate localization phenotype observed for some domains in KKT2?

      Response: As we responded to Reviewer 2, our observations suggest that these KKT2 fragments indeed localize at centromeres transiently (from S phase to anaphase). We will confirm this result by imaging with a transiently-localized kinetochore protein, KKT1 tagged with tdTomato.

      Page 6: The authors note that KKT2 Znf2 bears strong similarity to DNA-binding canonical Zinc fingers, and even note the high conservation of some putative DNA-binding residues. Have the authors tested for DNA binding by this protein?

      Response: As we responded to Reviewer 1 and 2, we used a fluorescence polarization assay and found that the Perkinsela KKT2a central domain binds DNA in a sequence-independent manner.

      Can the authors at least model DNA binding and see if that would result in a clash, given the packing of Znf2 against the larger Znf1?

      Response: As suggested, we superimposed the structure of Bodo saltans KKT2 Znf2 with that of a zinc finger 268 bound to DNA (PDB:1AAY), which shows a possible mechanism by which Znf2 might bind DNA. It also revealed a clash between DNA and Znf1 (in the crystal packing of the solved structure), implying that the position of Znf2 would need to change in order to bind DNA. We will add a supplementary figure showing a hypothetical DNA-binding mechanism by Znf2 and discuss the possibility of a necessary structural change in the Znf1 position to accommodate the DNA binding by Znf2.

      \*Minor Comments:** - Page 5: I'm skeptical as to whether these zinc-binding domains, especially Znf1, should really be referred to as "fingers". *

      Response: To our knowledge, the word “zinc finger” could be used for any protein that binds one or more zinc ions. Given that we still do not understand the molecular mechanism by which this domain functions, we wanted to use a very general term, Znf1. However, we do appreciate the reviewer’s point that calling this domain as a zinc finger could be misleading, so we will refer Znf1 and Znf2 in the original manuscript as the CL domain (for centromere localizing domain) and a classic C2H2 zinc finger in the revised manuscript.

      Page 8: At the beginning of the section describing KKT3 cellular experiments, I think the authors need to make it much more explicit that T. brucei KKT3 shares both Znf1 and Znf2 with KKT2.

      Response: We will add the suggested sentence before describing the functional assay for KKT3.

      Figure S1A: The gap between lanes in the middle of the major peak is really confusing (it's not even clear that this is two different SDS-PAGE gels next to one another). I initially thought that KKT2 was in both peaks, given the labeling of this figure. I suggest labeling the lanes specifically, or cropping the picture, to avoid confusion.

      Response: As suggested, we will prepare an image that shows only those lanes (from two separate gels) that were used for loading protein samples. We also like to retain the whole gel images in the same figure because those gels have rather low background signal (even without any contrast manipulation).

    1. The discourse community I’m going to be talking about is social media. Gathering information isn’t as easy as it looks. I’m going to be doing some research online,reading articles so I have back up evidence. I want to focus more on social networking sites and how that affects our lives in everyday life. We may not realize but a lot of problems start from these sites such as facebook, instagram, snapchat and much more. 

      One of the difficulties for choosing this as a discourse community is the idea of shared goals. I think this is important so please do address it in the paper.

      I found this essay for you that might help. Please take a look. https://pdfs.semanticscholar.org/dd19/4239a42bb7989fc8b041e599113604e71dbd.pdf

    1. Do you think that depression is “fashionable?” And by criticising the sufferers you can deter others from “joining in”? Granted, we hear more about depression than we used to these days, but then we know what it is now. We see a lot more photos from Mars these days, because we have the means of doing so now, not because it’s suddenly trendy. Perhaps you are trying to deter anyone else who might read your views from considering suicide themselves? Given that statistics suggest that one in four people suffer some sort of mental health problem, this isn’t that unlikely an occurrence. But if someone is genuinely depressed and feels their life is worthless, seeing that others consider their feeling selfish can surely only emphasise their own self-loathing and bleakness? It suggests that people will hate them even in death. Maybe you know some people who have “attempted” suicide purely for attention? Fair enough; a debatable conclusion, but even if you’re right, so what? Surely someone who succeeds at committing suicide is a genuine sufferer who deserves our sympathy? Perhaps you feel that those expressing sorrow and sadness are wrong and you need to show them that you know better, no matter how upsetting they may find it? And this is unselfish behaviour how, exactly?

      Throughout the rest of the article, the tone shifts majorly. It started off as an informative piece, trying to explain how depression affects people differently and what they go through before making a life altering decision. But now, it is almost like the writer is attacking the people on the other side of the argument. They are not providing explanations of information, just questions that can be taken as offensive. Certainly this does not show professionalism.

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      Author Comments:

      ­Response to Reviewers Comments

      We would like to thank all reviewers for carefully considering our manuscript and providing useful suggestions/ideas. The general consensus was that our study provides an important conceptual advance that reveals a new way of thinking about kinetochore phosphatases. However, in light of our surprising findings, it was suggested that additional experiments would be required to fully validate our conclusions. In particular, it was seen as important to test whether PLK1 can activate MPS1 from the BUB complex and to confirm that PP1 and PP2A are effectively inhibited in situations where MELT dephosphorylation can occur normally (Figure 3).

      In general, we agree with these and the other points raised by the reviewers, therefore we plan to address all comments as outlined in detail below.

      The major new additions to the final paper will be the following:

      1) Experiments to test how BUB-bound PLK1 affects MPS1 activity.

      2) Experiments to determine the efficiency of phosphatase inhibition in figure 3.

      3) Experiments to test whether maintaining PLK1 at the BUB complex causes SAC silencing defects

      4) Evolutionary analysis demonstrating that the PLK1 and PP2A-binding modules have co-evolved in the kinetochore BUB complex. This analysis, which has been performed already, strengthens our manuscript because it provides additional independent evidence for a functional relationship between PLK1 and PP2A on the BUB complex.


      Reviewer #1 **Minor comments:** 1) The authors propose that PP1-KNL1 and BUBR1-bound PP2A-B56 continuously antagonise PLK1 association with the BUB complex by dephosphorylating the CDK1 phosphorylation sites on BUBR1 (pT620) and BUB1 (pT609). It is therefore expected that converting these residues to aspartate would increase PLK1 recruitment. It would be interesting to verify if this hypothesis fits with the proposed model.

      Response: The general idea to maintain PLK1 at the BUB complex is a good one, but unfortunately polo-box domains do not bind to acidic negatively charged residues. Instead we will attempt to maintain PLK1 at the BUB complex using alternatively approaches (as suggested by reviewer 2).

      2) In Figure 1E, are the mean values for BubR1WT+BubWT and BubR1WT+Bub1T609 both normalized to 1? If so, this fails to reveal the contribution of Bub1 T609 for the recruitment of PLK1 when PP2A-B56 is allowed to localize at kinetochores.

      Response: The values will be updated and normalised to the BubR1WT+BUB1WT control. We have also performed additional experiments already and overall the results reveal a small reduction in kinetochore PLK1 following BUB1-T609A mutation and a larger reduction upon combined BUBR1-T620A mutation.

      3) What underlies the increase in Bub1 levels at unattached kinetochores of siBubR1 cells (Figure S1C?) Is this caused by an increase in Bub1 T609 phosphorylation and consequently unopposed PLK1 recruitment, which consequently increases MELT phosphorylation?

      Response: We suspect that PLK1 is not the cause of the increased BUB1 levels because PLK1 kinetochore levels are actually decreased in this situation (Figure S1A).

      4) Although the immunoblotting from Figure S1D indicates that BubR1T620A and Bub1T609A are expressed at similar levels as their respective WT counterparts, some degree of single-cell variability is expected to occur. As a complement to Figure 1B,C and Figure S1E,F could the authors plot the kinetochore intensity of BubR1 pT620 and Bub1T609 relative to the YFP-BubR1 and YFP-Bub1 signal, respectively?

      Response: There is indeed variability in the level of re-expression of BUBR1/BUB1 on a single cell level, which can at least partially explain the variation on BUBR1-pT620 and BUB1-pT609 observed within in each condition. We can upload these scatter plots at resubmission and include in the supplementary, if required.

      5) The authors nicely show that excessive PLK1 levels at the BUB complex are able to maintain MELT phosphorylation and the SAC (independently of MPS1) when KNL1-localised phosphatases are removed (Figures 2A,B). However, it should be noted that PLK1 is able to promote MPS1 activation at kinetochores and so, whether AZ-3146 at 2.5 uM efficiently inhibits MPS1 under conditions of excessive PLK1 recruitment should be confirmed. Can the authors provide a read-out for MPS1 activation status or activity (other than p-MELTs) to exclude a potential contribution of residual MPS1 activity in maintaining the p-MELTs and SAC?

      Response: This is a good point because although PLK1 can phosphorylate the MELTs it can also activate MPS1, although it is unknown whether it can do this from the BUB complex. We had left a dotted line in Figure 4B to include this possibility, but we will now test this directly with additional experiments.

      6) To examine whether PLK1 removal is the major role of PP1-KNL1 and PP2A-B56 in the SAC or whether they are additionally needed to dephosphorylate the MELTs, the authors monitored MELT dephosphorylation when MPS1 was inhibited immediately after 30-minute of BI2356. This revealed similar dephosphorylation kinetics, irrespective of compromised PP1-KNL1 or PP2A-B56 activity, thus suggesting that these pools of phosphatases are not required to dephosphorylate MELTs. To confirm this and exclude phosphatase redundancy, the authors simultaneously depleted all PP1 and B56 isoforms or treated cells with Calyculin A to inhibit all PP1 and PP2A phosphatases. In both of these situations, the kinetics of MELT dephosphorylation was indistinguishable from wild type cells if MPS1 and PLK1 were inhibited together. These observations led to the conclusion that neither PP1 or PP2A are required to dephosphorylate the MELT motifs. Instead they are needed to remove PLK1 from the BUB complex. This set of experiments is well-designed and the results support the conclusion. However, it would be of value if the authors provide evidence for the efficiency of PP1 and B56 isoforms depletion and for the efficiency of phosphatase inhibition by Calyculin A. An alternative read-out for the activity of PP1 and PP2A-B56 (other than p-MELT dephosphorylation) clearly confirming that both phosphatases are compromised when MPS1 and PLK1 are inhibited together could make a stronger case in excluding the contribution of residual PP1 or PP2A to the observed dephosphorylation of MELT motifs.

      Response: This is also a good point. We had attempted many different combinations in Figure 3 to inhibit PP1/PP2A activity as efficiently as possible. This is especially important considering the “negative” results on pMELT are very surprising. However, we will now test how efficiently we have inhibited PP1 and PP2A phosphatase function in these experiments.

      Reviewer #2 (Evidence, reproducibility and clarity (Required)): **Major comments:** 1) In its current state I am not convinced that the key conclusions are fully supported by the experiments and alternative conclusions/interpretations can be drawn. For example the level of MELT phosphorylation will be determined by the balance of kinase and phosphatase activity and if they do not achieve 100% inhibition of Mps1 in their assays then they are not strictly monitoring dephosphorylation kinetics in their assays. If the combination of Mps1 and Plk1 inhibition then more strongly inhibits Mps1 then dephosphorylation kinetics becomes faster. Thus subtle differences in Mps1 activity under their different conditions could lead to misleading conclusions but in its present state a careful analysis of Mps1 activity is not provided. This lack of complete inhibition also applies to the phosphatases and the experiments in Figure 3E indicates that their Calyculin preparation is not really active as at steady state MELT phosphorylation levels are much less affected than in for instance BubR1 del PP2A (Figure 2A as an example). Thus they likely still have phosphatase activity in the experiment in figure 3E making it difficult to draw the conclusions they do. A more careful analysis of kinase and phosphatase activities in their different perturbations would be recommendable and should be possible within a reasonable time frame.

      Response: These are good points and we will now more carefully assess MPS1 and PP1/PP2A activities.

      2) A more stringent test of their model would also be needed. What happens if Plk1 is artificially maintained in the Bub complex? The prediction would be that SAC silencing should be severely delayed even when Mps1 is inhibited. This is a straightforward experiment to do that should not take too long. If the polobox can bind phosphoSer then one could also make BubR1 T620S to slow down dephosphorylation of this site (PPPs work slowly on Ser while Cdk1 have almost same activity for Ser and Thr).

      Response: These are good suggestions and we will try to see if maintaining PLK1 at the BUB complex produces effects on the SAC.

      3) Another issue is the relevance of Plk1 removal under normal conditions. As their quantification shows in figure 1D-E (I think there is something wrong with figure 1E - should likely be Bub1) the contribution of BubR1 T620 and Bub1 T609 to Plk1 kinetochore localisation seems minimal. Thus upon SAC satisfaction there is not really a need to remove Plk1 through dephosphorylation as it is already at wild type levels. It is only in their BubR1 and KNL1 mutants that there is this effect so one has to question the impact in a normal setting. This is consistent with the data in Figure S1D showing no phosphorylation of these sites under unperturbed conditions.

      Response: The major finding of this study is that kinetochore phosphatases are primarily needed to supress PLK1 activity on the BUB complex and thereby prevent excessive MELT phosphorylation. The relevance of this continued PLK1 removal under normal conditions is clear, because when it cannot occur (i.e. if the phosphatases are removed) then the SAC cannot be silenced unless PLK1 is inhibited. Therefore, whilst it is true that PLK1 localisation to the BUB complex is low under normal conditions, that is because the phosphatases are working to keep it that way. The relevance of that continual removal is an interesting, but in our opinion, separate question that will require a new body of work to resolve. One possibility is that PLK1 recruitment is a continual dynamic process, that is perhaps coupled to a particular stage in MCC assembly. For example, PLK1 could bind the BUB complex to recruit PP2A to BUBR1, before being immediately removed by PP2A. In this sense, PLK1 binding could still be functionally important even if it is only occurs transiently and steady state PLK1 levels are low. We will add a line to the discussion to highlight that it would be interesting to test PLK1 dynamics on the BUB complex in future.

      4) They write that in the absence of phosphatase activity Plk1 becomes capable of supporting SAC independently (of Mps1 is implied). They do not show this - only that MELT phosphorylation is maintained. As Mps1 has other targets required for SAC activity I would rephrase this.

      Response: Good point, this will be rephrased.

      Reviewer #2 (Significance (Required)): The advance is clearly conceptual and provides a new way of thinking about the kinetochore localized phosphatases. These phosphatases and the SAC have been immensely studied but this work brings in a new angle. The discussion would benefit from some evolutionary perspectives as the PP1 and PP2A-B56 binding sites are very conserved but the Plk1 docking sites on Bubs less so. This will be of interest to people in the field of cell division and researchers interested in phospho-mediated signaling.

      Response: Since the paper was submitted, we performed evolutionary analysis to examine this point. We discovered that the PLK1 docking sites are surprisingly well conserved and, in fact, they appear to have co-evolved within the same region of MAD/BUB along with the PP2A-B56 binding motif. We believe this new data strengthens our manuscript because it argues strongly for an important functional relationship between PLK1 and PP2A. A new figure containing this evolutionary analysis will be included in the final version.

      Reviewer #3 **Major comments:**

      1. An important limitation of this study is that KNL1 dephosphorylation at MELT repeats is monitored only by indirect immunofluorescence using phospho-specific antibodies. Thus, reduction of phospho-KNL1 kinetochore signals could be due to protein turnover at kinetochores, rather than to dephosphorylation. This is a serious issue that could be addressed by checking KNL1 dephosphorylation during time course experiments by western blot using phospho-specific antibodies, as previously done (Espert et al., 2014).

      Response: This is an important point that we feel is best addressed by examining total KNL1 levels at kinetochores (instead of simply total cellular levels by western blots). The reason is that KNL1 could potentially still be lost from kinetochores even if the total protein is not degraded. In all experiments involving YFP-KNL1 we observe no change in kinetochore KNL1 levels and this data will be included in the final version. We will also perform new experiments to examine total KNL1 levels in the BUBR1-WT/DPP2A situation to test whether KNL1 kinetochore levels are similarly maintained in these cells following MPS1 inhibition.

      1. For obvious technical reasons, the shortest time point at which authors compare KNL1 dephosphorylation upon MPS1-PLK1 inhibition is 5 minutes. Based on immunofluorescence data, authors conclude that kinetics of KNL1 dephosphorylation are similar when kinases are inhibited, independent of whether or not kinetochore-bound phosphatases are active. However, in most experiments (e.g. Fig. 3B, 3C, 3E) lower levels of MELT phosphorylation are detected after 5 minutes of kinase inhibition when phosphatases are present than when they are absent, suggesting that phosphatases likely do contribute to KNL1 dephosphorylation. I suspect that differences between the presence and absence of phosphatases might even be more obvious if authors were to look at shorter time points, when phosphatases conceivably accomplish their function. I would therefore suggest that the authors tone down their conclusions, as their data complement but do not disprove the previous model.

      Response: We appreciate that small differences can be seen in figure 3B and 3E at the 5-minute timepoint (between the WT and phosphatase inhibited situations). This may reflect a role for the phosphatases in dephosphorylation or in the ability of drugs such as BI-2536 (3B) or Calyculin A (3E) to fully inhibit their targets in the short timeframe. We will perform additional experiments to examine MPS1 and phosphatase activity under these conditions, in response to comments by reviewers 1 and 2. In the final version we will carefully interpret the new and existing data and, if required, modify the conclusions appropriately.

      1. In all experiments cells are kept mitotically arrested through nocodazole treatment, which is not quite a physiological condition to study SAC silencing. This could potentially mask the real contribution of phosphatases in MELT dephosphorylation. Indeed, it is possible that higher amounts of phosphatases are recruited to kinetochores during SAC silencing than during SAC signalling (e.g. during SAC signalling Aurora B phosphorylates the RVSF motif of KNL1 to keep PP1 binding at low levels; Liu et al., 2010). What would happen in a nocodazole wash-out? Would phosphatases be dispensable in these conditions for normal kinetics of MELT dephosphorylation and anaphase onset if PLK1 is inhibited?

      Response: All SAC silencing assays where performed in nocodazole for 2 main reasons: 1) PP2A-B56, PP1 or PLK1 can all regulate kinetochore-microtubule attachments, and thereby control the SAC indirectly. Therefore, performing our assays in the absence of microtubules allows us to make specific and direct conclusions about SAC regulation; 2) Previous work on pMELT regulation by PP1/PP2A in human cells was also performed following MPS1 inhibition in nocodazole (Espert et al 2014, Nijenhuis et al, 2014). Therefore, we are able to directly compare the contribution of PLK1 to the previously observed phenotypes, which allowed us to conclude that PLK1 has a major influence.

      Nevertheless, we appreciate the point that the influence of PLK1 could, in theory, be different during a normal mitosis when microtubule attachment can form. Therefore, we will attempt to address whether PLK1 inhibition can bypass a requirement for PP1/PP2A in SAC silencing during an unperturbed mitosis.

      Other data are overinterpreted. For instance, the evidence that CDK1-dependent phosphorylation sites in Bub1 and BubR1 is enhanced when PP1 and PP2A-B56 are absent at kinetochores suggests but does not "demonstrate that PP1-KNL1 and BUBR1-bound PP2A-B56 antagonise PLK1 recruitment to the BUB complex by dephosphorylating key CDK1 phosphorylation sites on BUBR1 (pT620) and BUB1 (pT609)(Figure 1F)". Similarly, the claim "when kinetochore phosphatase recruitment is inhibited, PLK1 becomes capable of supporting the SAC independently" referred to Fig. 2C-D is an overstatement, as residual MPS1 kinase could be still active in the presence of the AZ-3146 inhibitor.

      Response: These are good points and the indicated statements will be reworded.

      **Minor comments:**

      1. In many graphs (Fig. 1A-C, Fig. 2A,C) relative kinetochore intensities are quantified over "CENPC or YFP-KNL1". Authors should clarify when it is one versus the other.

      Response: This will be clarified in the axis and in the methods.

      1. The drawing in Fig. 1F depicts the action of PP1 and PP2A-B56 in antagonising PLK1 at kinetochores. Thus, the output should be SAC silencing, rather than activation.

      Response: The SAC symbol will be removed from the schematic to avoid confusion and because it is not actually the focus of figure 1 anyway.

      1. In the Discussion authors speculate that KNL1 dephosphorylation relies on a constitutive phosphatase with unregulated basal activity. Would a phosphatase be needed at all when MPS1 and PLK1 are inhibited? Could phosphorylated KNL1 be actively degraded?

      Response: We will insert total KNL1 immunofluorescence quantification so show that KNL1 KT levels are not decreased in this situation. KNL1 remains anchored at kinetochore but the MELTs must be dephosphorylated to remove the BUB complex.

      1. What happens to MPS1 when KNL1-bound PP1 and BUBR1-bound PP2A are absent? Do its kinetochore levels increase as observed for PLK1? And what about the kinetochore levels of Bub1 and BubR1?

      Response: We have demonstrated previously that BUB1/BUBR1 increase in this situation in line with the pMELTs (Nijenhuis et al 2014;l Smith et al, 2019) – these papers will be referenced in relation to this. We will also address the effect of phosphatase removal on MPS1 activity, in response to comments by reviewers 1 and 2.

  13. Feb 2020
    1. Good job, Paul. A surprisingly analytical mind for your age . I like how you incorporate ideas and arguments into your essay. And you are right.... Sometimes I think...Human is the most selfish being on the surface of the earth...Or ..is he? Aren't we animals with high intelligence and conscience ?Aren't we guided by the ancestral instinct of conservation? Like animals protecting their teritoires , we went to war from the earliest times to settle our boundaries. Like males of so many species ,men have killed each other ( literaly or only relational speaking) for women, and , of course, since human is more complex than most animals , women would kill each other as well for men. Just like in the wildest jungle, the strongest will survive .But what has consciense done to us? Killing and "jungle law" do exist in animal's world , but that world is a balanced one. Our world is a world that is constantly in contradiction with itself, for one we think , and one we say , and one we wish , but one we do..We are advocates of good and mercy but in the mean time we do so much harm...To ourselves , to each other , to the planet . What animal would kill for pleasure? What animal would destroy its own habitat ?What animals would trade the fundamental need of sex for the fundamenal need for food (which translated in human terms=money )? We admit publicly that violince and hatred are bad , but in the mean time we promote these all over the place in mass media. And , perhaps not surprinsingly , most of the times, the most popular tv programs are the ones that contain this kind of material.we keep denying the animal part in ourselves , but in the mean time we bring it to the surface , and not in just any form, but in the form of the wildest beast. What has conscience done to us? Or , better said, what have we done with conscience? We are always able to choose , but why so often this right to choose given by thought is manifested in its rather negative aspect , than its positive one? And if something can be done , than why don't we do something about it? That's why I like classical music;becaue it is not infested with any of these malicious things . Classical music usually brings to the light the other side of human , the great one , the kind and superior one.The one that is suppose to prove that we evolved from a primitive , wild stage to a more refined one. Like Shinichi Suzuki said , if we would nurture our children with more noble music , than our society would be a different kind of society. You need never apologize for speaking your heart. More often it's silence that conspires to lead a society in the wrong direction. I liked your response, but I don't like the question. There's too much polarized either/or thinking and discourse in the media already. People who survived either of the World Wars in Europe might disagree that today's wars are worse. And as a woman, I can't help but think of the changes that have made my current life possible--I mean, we're not even 100 years out from when I wouldn't have been able to vote, or go to college. I agree with you, something has to be done. But something has always had to be done. Larisa, thank you for sharing your view and comments on this. Laurie, those are two of the greatest sentences I've heard in a long time. Karen, thank you for showing me what someone with an interesting view thinks. I was going to include some things about how women are still somewhat treated unfairly but then my essay would have had another few hundred words. And that's what was hard with showing my view, and the opposition. Especially if I would have used a subject like female rights. The arguement in my opinion would be that it's gotten better, but still exists widely. Which does not change into a persuasive essay very well. And I don't think that any war is more horrible than another. If you look at the people who are killed, each death is going to affect some one on such a deep personal level. A death could be a child losing a parent or a parent losing a child. It will always be something different, yet the same. Paul, thanks for putting your essay up here. I enjoyed reading it! You pose a good argument and write much better than I ever could when I was your age. A suggestion to make your argument stronger: Your central thesis is that the world is changing for the worst, because of hate and discrimination, and how these lead to conflict. In your essay you have supplied good evidence of this and have argued it well, and on its own it stands up as an argument. But, to keep in mind: you haven't shown how this is different from the way things were in the past, and thus not completely supporting that things have changed for the worst. Hate, discrimination, and violence resulting from those two things have been around forever and forever. In fact, one could argue that the world is improving because of the increased awareness of and attempts to eradicate hate and prejudice in comparison to the past (also for example improvement in woman's rights, etc. like Karen suggested). To solidify your argument you would need to show how the hate and violence today is worse than earlier, ie. through comparisons of then/now, showing escalation of conflict, maybe something about today's technology and ease of killing, etc. But all in all, a good essay! If you write any more, will you also post them up here? Thank you so much for telling me what I need to fix and ways to do so! It is very helpful. Being able to see the faults in my writing and how to fix it is something that I really need to work on and you've just helped me in that. And yes, if it's ok with Laurie and everyone else, I'll post up more essays in the future if you'd like. I can not be more original than the others in my comments so GOOD JOB! You should send this to Obama... so that he has an idea of what is doing the young generation that is too often seen as "a buch of lazies with no heart" lol Great! Anne-Marie I think that is a beautiflly written essay, mirroring a lot of the hatred and discrimination that exist today. However, the topic assigned was whether these things are getting worse. That would be hard to say because hatred, fear, anger, aggressivity, etc. have always been around. Your essay would be stronger if you would explain why you think that things are changing for the worst. Access to weapons and to international news reports, as well as violence shown so widely on TV and video games, may make us more "violence-friendly." I am sure that there are many more reasons. I agree with Laurie. Silence gives consent. I like one of the slogans used by the Friends (Quakers): War is not the answer. Understanding and compassion can be very powerful at blocking violence. As you can tell by the discussion about your blog, your thinking is very mature. Thanks for posting your essay. Anne Marie, I started reading Obama's book The Audacity of Hope recently, and the Introduction put me very much in mind of Paul's essay here and some of the responses. I think Obama would be very sympathetic to these ideas. One of the additional themes of that book, at least in my interpretation, is that in order to move forward, we need to get away from simplistic polarized formulations like "is it better or worse?" I think it is to Paul's credit that he was able to make such an interesting essay out of a not-so-great assignment question. This entry has been archived and is no longer accepting comments.

      This is the extra stuff.

    1. First the most famous example of an indisputably (or so you would think) fake news story that has had real-world consequences.

      This type of online activity has been around since the beginning. Well maybe not the complete beginning but you understand... The anonymity and falsehoods are not only currently allowed but also sort of asked for by design. That is to say that there will always be con men and there will always be platforms for them to work within. This is just the modern equivalent of forging a check. Regardless, that is not to say that it does not make an impact. It does! But it currently only does so due to our lack of strong digital literacy. We need to teach that now more than ever but I think that in a way we already are. Younger kids these days have a great grasp on what may be real or fake and have even made a sort of entertainment game out of it with online story telling and ARGs. We just need to continue to strengthen our understanding of evolved digital literacy as a form of prevention and truth seeking.

    1. r adults to understand the languages that children may prefer, such as creative expression and play. Adults may have neglected and forgotten these languages in the course of growin

      This is so true. We can't expect children to be experts with something they have not had exposure to. Most preschools I think understand this, but as soon as they enter kindergarten this notion is lost.

    2. face

      I feel like we don't give children enough credit in the way that they think. They may not think as deep as we can as adults, but they are not incapable of thinking in a way that is abstract.

    1. Students in many fields, for example astronomy, oceanography, and genomics, are often already proficient in analyzing large, complex data sets. That’s an attractive skill to many employers.

      50% agree 50% disagree. While scientific papers are a niche, I still think there is great value in the process of learning how to write and present results. That is a skill we cannot lose as scientists. And I don't think this article is strictly suggesting we step away 100% from journal articles, but I do think journals may reach a broader audience? As a student, we read a lot of papers and those papers contribute to our learning and sometimes we just want a quick look at results or methods or simply the introduction. Personally, I don't want to run code or a software program to see results if I'm just looking to write a sentence or two for an introduction or an abstract. Again, I think maybe the shift should be to have these codes / softwares available to complement the paper. Not the other way around.

    1. PowerThe ability to have one’s will carried out despite the resistance of others. refers to the ability to have one’s will carried out despite the resistance of others.

      In addition to what is power, let's say you are at work and you run into the CEO of the company. Instantly, you might get nervous. You act different, you might walk different. The CEO represents a person of "power" so you may quickly think of something nice or professional to say as you walk closer to them. You will act a little different than you would around you other co-workers that you talk to on a daily basis. There are many other examples of this power we encounter everyday.

    1. [Note: this preprint has been peer reviewed by eLife. The decision letter after peer review, based on three reviews, follows. The decision was sent on 19 May 2019.]

      Summary

      This paper describes five cryo-EM structures of ribosomal complexes apparently representing different stages of RF2-catalyzed translation termination. The novel observations here are that the tip of domain 3 of RF2 undergoes a rearrangement from an a-helical conformation to a b-hairpin conformation during termination that likely facilitates exit of the newly synthesized protein from the ribosomal polypeptide exit tunnel and that the ribosome can undergo two thermally activated, spontaneous conformational changes, a relative rotation of the ribosomal subunits and a swiveling of the 'head' domain of the small subunit, during termination that likely facilitate dissociation of RF2 from the ribosome. These are interesting observations that significantly extend our understanding of how class I RFs and ribosome conformational changes drive important steps during termination and, as such, all three reviewers recommended publication provided the following comments are addressed adequately.

      Essential Revisions

      1) The maps provided through the eLife system seemed to be unsharpened, as they showed very little detail. However, even after sharpening them with a B-factor of -100A2, they still did not show the expected features for their respective resolutions. My suspicion is that FREALIGN has been used to overfit the data. This should be addressed in the revision. It should be indicated whether gold-standard separation of halves of the data sets were used in the final refinements, or whether those were limited to a specific spatial frequency (like was done in the classifications). If the latter, those frequencies should also be stated in the manuscript, and they should be significantly lower than the claimed resolutions.

      In addition: a lot of basic cryo-EM information is missing: the authors should include: a) at least one micrograph image b) some representative 2D class averages c) local resolution maps of the five structures. Also, because the density of important parts of the maps seems to be a lot worse than the resolution claimed, it would be good to explicitly mention the local resolution of the important features discussed in the main text. d) for each structure, some zoomed-in figures with the density on top of the molecular model. These figures should be chosen as to validate the resolution claim. For example, in structures I, II and V, the RNA bases should be well separated (they do so at 3.6A), and in structures III and IV beta-strands should be well separated, and many (larger) side chains should be visible. In addition, some panels with density for the most important features of each structure should be shown. e) FSC curves between the refined PDB models and the cryo-EM maps are missing from the manuscript. These should be included. In addition, to evaluate potential overfitting of the models in the maps, for each structure, the authors should also include the FSC curves between a model that was refined in half-map1 versus half-map1, as well as the FSC curve between _thesame model versus half-map2.

      2) There appear to be many self-citations, and there are also a few places where relevant citations are missing or are mis-cited. There are too many to list individually, but, just a few examples: Page 4: the only citation for the phrase "recent biophysical and biochemical findings suggest a highly dynamic series of termination events" is a Rodnina paper. There are many, earlier papers from Ehrenberg, Gonzalez, Puglisi, Green, Joseph, etc. that should be cited here. Page 5: The only citation for the sentence "By contrast, biochemical experiments showed..." is a Green paper. There are earlier papers from Ehrenberg characterizing the effects of the GGQ-->GAQ mutations on the ability of RF3 to accelerate the dissociation of class I RFs from termination complexes that should be cited here. Page 5: There's a sentence that refers to X-ray, cryo-EM, and smFRET studies, but only provides citations to two smFRET studies (Casy et al, 2018 and Sternberg et al, 2009); Page 5: Moazed and Noller, 1989 identified and characterized the P/E hybrid state, but they didn't report that a deacylated P-site tRNA 'samples' the P/E hybrid state 'via a spontaneous intersubunit rotation'--that was later work from Noller and Ha; etc. There are several other instances of missing citations or mis-citations. We would ask that the authors review their citations with an eye for excessive self-citations and for missing citations or mis-citations. In this context, "Ensemble-EM" is also cited as a specific method in the introduction (Abeyrathne et al., 2016; Loveland et al., 2017). However, this method is more commonly known as (3D) classification of cryo-EM images, and there are many older, more appropriate citations.

      3) The sample imaged is a model sample generated by in vitro assembly with purified components of a termination complex. In order to mimic a bona fide termination complex, a short messenger RNA with a strong Shine-Dalgarno sequence followed by a start codon and immediately after by a stop codon was used (mRNA sequence: 5'-GGC AAG GAG GUA AAA AUG UGA AAAAAA-3'). Similar constructs were used to crystallize termination complexes in the past and it has been proven by smFRET experiments that, at least regarding ribosomal inter-subunit dynamics, this model sample behaves similarly to a real termination complex with a peptide linked to the P site tRNA. However, the nature of this model sample should be apparent for the non-specialist reader, highlighting its similarities with a real termination complex but also its possible limitations, especially regarding the "artificial" nature of having a stop codon so close to the Shine-Dalgarno sequence, a situation that never happens in real mRNAs. The authors should explicitly acknowledge this and discuss its implications in the main text.

      4) The authors set up a couple of somewhat 'strawman' arguments in claiming that: (i) there are discrepancies in the X-ray, cryo-EM, and smFRET literature with regard to whether ribosomes can undergo intersubunit rotation while bound to class I RFs or whether the non-rotated conformation of the ribosome is stabilized by bound class I RFs and (ii) class I RFs are able to terminate translation and dissociate from the ribosome without the aid of RF3. In the case of (i), it is obviously possible for class I RF-bound ribosomes to undergo intersubunit rotation while still favoring the non-rotated conformation of the ribosome. Moreover, there are enough differences between the cited studies, both in terms of the experimental conditions as well as the technical limitations associated with the various experimental techniques, that it is easy to rationalize differences with regard to whether the class I RF-bound ribosomes would be expected to undergo intersubunit rotation and/or whether the researchers would have been able to capture/observe intersubunit rotation. In the case of (ii), decades of biochemistry from Buckingham, Ehrenberg, Green, and others had already demonstrated that class I RFs are able to terminate translation and dissociate from the ribosome without the aid of RF3, and that the role of RF3 in termination is to accelerate the spontaneous dissociation of the class I RFs. If the authors want to highlight discrepancies in the literature, they should frame them in the context of differences between the studies, experimental design, limitations of the approaches/techniques in the cited papers that might account for such discrepancies. Re-writing this paragraph also in the light of addressing the missing citations and mis-citations pointed out under (2) will further help in toning these arguments down, which would strengthen the manuscript's scholarship.

      5) Class I RFs are post-translationally methylated at the Q residue of the GGQ motif of domain 3 and Buckingham, Ehrenberg, and others have shown that this methylation accelerates and/or facilitates class I-catalyzed termination both in vitro and in vivo. Nonetheless, Svidritskiy et al do not report whether and to what extent their RF2 is methylated. Was RF2 overexpressed in a manner that ensured homogeneous methylation or lack of methylation? If they are overexpressing prfB and not overexpressing prmC, it is likely that they have a mix of methylated and unmethylated RF2. Assuming they are using the wt E. coli prfB gene, then the residue at position 246 is a T, rather than an A or S, and Buckingham has shown that, in the wt T246 background, a lack of methylation at Q252 is either seriously detrimental in richer media or lethal in more minimal media. It was felt that a discussion of this issue was not needed in the main text, but that it would be helpful if the authors would include the important/relevant experimental details in the Methods section, for example, did they use the T246 wt E. coli variant of RF2; and did they overexpress prmC along with prfB?

      6) Structure I is denoted and treated as a pre-termination complex, but that does not seem at all possible given that the sample was prepared by incubating a pre-termination complex for 30 min in the presence of excess RF2, conditions that Figure 1-Figure Supplement 3 suggest results in robust termination. Structure I is more likely the non-rotated conformation of a post-termination complex that is in equilibrium with its rotated counterpart, Structure V. Based on my reading of the manuscript, it is likely that the authors understand this point, but are nonetheless using this structure as a mimic/analog of a pre-termination complex. If so, I think this is fine, but the authors should explicitly state that this is what they are doing. Related to this, the authors should clarify the description of their activity assay, show the raw data, and/or report 'Released [S35]-fMet (%)' instead of 'Released [S35]-fMet, CPM' on the y-axis of Figure 1-Figure Supplement 3; as the activity assay is currently described, reported, and plotted, it is impossible to determine whether RF2 is 1% or 99% active in termination.

      7) The final sentence of the manuscript reads: "Translation termination and recycling of the release factors and the ribosome therefore rely on the spontaneous ribosome dynamics, triggered by local rearrangements of the universally conserved elements of the peptidyl-transferase and decoding centers". There are a couple of problems with this sentence as written. First, smFRET experiments by Gonzalez, Puglisi, and Rodnina have previously shown that "Translation termination and recycling of the release factors and the ribosome therefore rely on the spontaneous ribosome dynamics" and the relevant articles should therefore be cited here. Moreover, given the data are static structures solved using a sample that is at equilibrium, it is not clear how the authors determined that these spontaneous ribosome dynamics were "triggered by local rearrangements of the universally conserved elements of the peptidyl-transferase and decoding centers". Isn't it equally possible, given the data presented, that the local rearrangements were triggered by the ribosome dynamics?

    1. I do not think I would.

      I find it pretty compelling the way the speaker spends the entire poem dismissing the concept of love as something essential to life, and yet in the end she still states that she would probably not trade love for nourishment. She shows how often, we may be well aware that our emotions are not completely rational, and yet we still struggle to control them.

    1. Alternatively, by saying that researchers pursue taxpayer funding (rather than scientific funding) the author may be making the implicit assumption that researchers compete to work on projects in the taxpayers’ interest, rather than scientific interest. What do you think?

      i would not agree to this framing...

      i would put it this way... researchers, just like any other humans , are not immune to rewards and punishment, (or STRUCTURE) of their professions...

      if we (the PUBLIC) wish researchers to pursue scientific (however defined) interest, then the structure must be modified accordingly...

      who created the current structure? public and private elites... in democratic states, the public certainly have power over public elites, so they should pressure these public elites to change the structure...

      now, why is the public concerned with science and scientific advancement? to my understanding, this concern is often expressed in state's constitution, which created the public in each particular state...

      then we come to the issue what if the public interest is sometimes against scientific interest? well, this is where (democratic) negotiation takes place... scientists must convince the public that science is useful for them, in all its seeming 'uselessness'...

      many times the scientists only need to convince the public elites for this, not the public as a whole... but i think it's good if scientists are always in touch with the public as a whole with regards to their work if they use public funding...

      in conclusion, i had fun reading the comments, both at the original site and ea forum, because some commenters 'pierced the veil of ideology' behind the original article, and some of the back-and-forth were hilarious with my knowledge of 'money and the state'... would love to expand on this later when i have time... :)

    1. Look to her, Moor, if thou hast eyes to see:She has deceived her father, and may thee

      I think that this line can be described as an indirect foreshadow of future events. Othello in the end believes that Desdemona betrayed him even though we the audience know what really went on.

    1. As college students on the threshold of the academic community, many of you probably think that research is about finding answers to a question given to you by your professor. While this may have been true in high school, we would like to propose that a more meaningful research

      This is something that I always though was true, I remember there would be times were I would be so stressed because I did not know if what I was writing was correct, as I was always focusing on what the right answer was for the question being asked or prompt.

    1. This Nicholas gan mercy for to crye,                     This Nicholas began to cry for mercy, 3289         And spak so faire, and profred him so faste,                     And spoke so fair, and pressed his suit so fast, 3290         That she hir love hym graunted atte laste,                     That she granted him her love at the last, 3291         And swoor hir ooth, by Seint Thomas of Kent,                     And swore her oath, by Saint Thomas of Kent, 3292         That she wol been at his comandement,                     That she will be at his commandment, 3293         Whan that she may hir leyser wel espie.                     When she may well espy her opportunity. 3294         "Myn housbonde is so ful of jalousie                     "My husband is so full of jealousy 3295         That but ye wayte wel and been privee,                     That unless you wait patiently and are secretive, 3296         I woot right wel I nam but deed," quod she.                     I know right well I am as good as dead," said she. 3297         "Ye moste been ful deerne, as in this cas."                     "You must been very secret in this matter."

      This seems like an important piece of the work to me as it shows a major theme in British Literature. Deception is something that I have found to be predominant in many works that we have read. I also think this becomes a major turning point in the story where the plot thickens and the main conflict will occur.

    1. When redefining learning experiences, we cannot do so at the expense of human connection. The most powerful technologies known to human did more than simply minimize complexity or do something new and catchy: they enhance communication, making it easier for individuals to connect with one another

      Will technology preserve or enhance human connection in the classroom is by far a great question. Technology does make it easier to connect you leave an imprint everywhere you go online. I think as much as it enhances communication it also hinders it.

    1. Below him, in the town among the trees, Where friends of other days had honored him,

      Below him are his friends that are now dead, this supports that he is writing a poem of loneliness. BUT, what I find from this poem is that, even thought dying is undeniably depressing, NOT dying is even MORE depressing when you are of a certain age. This is almost reassuring, for no one like to think about when they are going to leave this planet, worried that we will kicking and screaming as death takes us away; but here we get a sense that this feared experience may never come. This poem is of a man that is depressed not because death is near, but because death has yet to come.