6,780 Matching Annotations
  1. Sep 2020
    1. We have to limit fortunes. Our present plan is that we will allow no one man to own more that $50,000,000. We think that with that limit we will be able to carry out the balance of the program. It may be necessary that we limit it to less than $50,000,000. It may be necessary, in working out of the plans that no man’s fortune would be more than $10,000,000 or $15,000,000. But be that as it may, it will still be more than any one man, or any one man and his children and their children, will be able to spend in their lifetimes; and it is not necessary or reasonable to have wealth piled up beyond that point where we cannot prevent poverty among the masses.

      The thing about “limiting” fortunes is that there is always going to be a loophole. Offshore accounts or something similar to that or them putting assets in other people’s names like their newborn children or some relative. To me it does not seem achievable.

    2. I now ask those who read this circular to help us at once in this work of giving life and happiness to our people — not a starvation dole upon which someone may live in misery from week to week. Before this miserable system of wreckage has destroyed the life germ of respect and culture in our American people let us save what was here, merely by having none too poor and none too rich. The theory of the Share Our Wealth Society is to have enough for all, but not to have one with so much that less than enough remains for the balance of the people.

      Reading this made me think about the current situation we are facing today. I understand that billionaires earned their money, but to have that much money is outrageous. I also understand that it is their money, and they can use it however they want but, I do not think that this is fair. They do not get taxed as we do. For instance, the CEO of Amazon is worth $185 billion. He and other billionaires could literally donate 1% of their wealth, and cover the very needed PPE’s, education, etc. A lot of billionaires have the power to eradicate homelessness and other problems in America, but I believe that they are selfish and greedy. As stated previously, it is their money and they could do anything desired however, there should be no room for an individual to accumulate that much wealth. With this whole COVID-19 issue, many businesses shut down except the big corporations. These corporations made billions from a pandemic, which is insane. A lot of people lost their livelihood and jobs yet these corporations made bank from it, which shows the uneven wealth distribution.

      Regarding the past, I can see where Huey Long is coming from. The wealth distribution at that time seemed to be unfairly distributed. He wants the non-wealthy individuals to obtain the same opportunities rich people could get such as education. The only part I do not agree with Huey Long is the having a complete balance of middle class. There would be no room for progression and development. Everybody would be paid the same regardless of what education level and status. What I got from this was a cashier could make the same as a doctor. I think the balance of poor and rich is necessary but, I do not agree with super wealthy individuals (billionaires).

    3. We have to limit fortunes. Our present plan is that we will allow no one man to own more that $50,000,000. We think that with that limit we will be able to carry out the balance of the program. It may be necessary that we limit it to less than $50,000,000. It may be necessary, in working out of the plans that no man’s fortune would be more than $10,000,000 or $15,000,000. But be that as it may, it will still be more than any one man, or any one man and his children and their children, will be able to spend in their lifetimes; and it is not necessary or reasonable to have wealth piled up beyond that point where we cannot prevent poverty among the masses.

      On paper, this is an excellent concept and something that I think everyone would love to see. Why does any one person need that much money? Why does this concept not work? Why isn't this the standard? If everyone could just sit at home watching TV and having money dropped off, why would anyone work? One major reason is that without incentive, no one would work and there would be no wealth to redistribute. You also gain more than just money from accomplishing something yourself. You build values and morals. You create self worth. Without that, who are you?

    4. It is impossible for the United States to preserve itself as a republic or as a democracy when 600 families own more of this Nation’s wealth—in fact, twice as much—as all the balance of the people put together. Ninety-six percent of our people live below the poverty line, while 4 percent own 87 percent of the wealth. America can have enough for all to live in comfort and still permit millionaires to own more than they can ever spend and to have more than they can ever use; but America cannot allow the multimillionaires and the billionaires, a mere handful of them, to own everything unless we are willing to inflict starvation upon 125,000,000 people.

      Huey Long makes some valid and strong points that still hold true till this day. For example, although we are a Constitutional republic, currently, one percent of people own thirty eight percent of our entire nation's wealth. I think Huey Long presents some socialistic ideals throughout his two speeches, but I think his overall goal was to take money from the very wealthy that earned exorbitant amounts of wealth and distribute that wealth to those that needed it most, to make our country's people stronger and make our country's distribution of wealth more just. Fast forward to present day. The pandemic has crushed many small businesses, that the American economy greatly relies on and yet other large corporations have made huge exponential financial gains because of the pandemic and increased online consumerism. Unemployment rate in the coming months may exceed eleven percent, meaning that 1 in 8 U.S. workers will not have a viable job. Instead of letting big businesses get richer with billions of dollars, or tax payers funding stimulus payments that our children and our children's children will be paying off huge national debt in years to come, why not have the elite business owners donate some of their excessive wealth for the better of other U.S. citizens. After all, it is the U.S. citizens that through consumerism made them billionaires in the first place. Mr. Long did have a good point in my opinion. These companies deserve their wealth for what they created, but I think they should give back some of it to help sustain our economy which will enable them to keep prospering in the long haul and help U.S. citizens that are in or will soon be in dire need of financial help.

    5. We have to limit fortunes. Our present plan is that we will allow no one man to own more that $50,000,000. We think that with that limit we will be able to carry out the balance of the program. It may be necessary that we limit it to less than $50,000,000. It may be necessary, in working out of the plans that no man’s fortune would be more than $10,000,000 or $15,000,000. But be that as it may, it will still be more than any one man, or any one man and his children and their children, will be able to spend in their lifetimes; and it is not necessary or reasonable to have wealth piled up beyond that point where we cannot prevent poverty among the masses.

      You have to pause for a moment to consider the impact this would have on America's middle class -- people would be free to pursue entrepreneurship in businesses about which they are passionate, people would be able to change careers without the fear of bankrupting themselves. This would create a lot of movement in the middle class and class mobility.

    6. We have to limit fortunes. Our present plan is that we will allow no one man to own more that $50,000,000. We think that with that limit we will be able to carry out the balance of the program. It may be necessary that we limit it to less than $50,000,000. It may be necessary, in working out of the plans that no man’s fortune would be more than $10,000,000 or $15,000,000. But be that as it may, it will still be more than any one man, or any one man and his children and their children, will be able to spend in their lifetimes; and it is not necessary or reasonable to have wealth piled up beyond that point where we cannot prevent poverty among the masses.

      So again, this passage sounds very socialist in nature. My question is How exactly would you go about making this a reality? It kind of makes me think of politics today. I won't lie, I like the idea Bernie Sanders proposed with regard to getting rid of all the student debt. While that would benefit me as an individual, I understand why some people were very much against it. The same question I had for him, that I have for Huey Long is: How would you make this work? For the people who had mountains of debt, but found a way to pay them off, would you give them some money back for their troubles? I think the idea of making sure everyone gets a fair chance is amazing- in theory. In reality, I don't think it's that simple.

    7. We have to limit fortunes. Our present plan is that we will allow no one man to own more that $50,000,000. We think that with that limit we will be able to carry out the balance of the program. It may be necessary that we limit it to less than $50,000,000. It may be necessary, in working out of the plans that no man’s fortune would be more than $10,000,000 or $15,000,000. But be that as it may, it will still be more than any one man, or any one man and his children and their children, will be able to spend in their lifetimes; and it is not necessary or reasonable to have wealth piled up beyond that point where we cannot prevent poverty among the masses. …

      So it's very interesting that he starts at 50 million and by the end lands 10-15 million and later even lower. When you can start limiting others money and spend their money by taxing it's easy to limit money.

    8. We have to limit fortunes. Our present plan is that we will allow no one man to own more that $50,000,000. We think that with that limit we will be able to carry out the balance of the program. It may be necessary that we limit it to less than $50,000,000. It may be necessary, in working out of the plans that no man’s fortune would be more than $10,000,000 or $15,000,000. But be that as it may, it will still be more than any one man, or any one man and his children and their children, will be able to spend in their lifetimes; and it is not necessary or reasonable to have wealth piled up beyond that point where we cannot prevent poverty among the masses.

      This is a very interesting idea from Huey Long. It could be that the reason for this is that he was born in the poor north of Louisiana before he gained education, money and fame. He proposed to make things equal for everyone and mentioned that people should not have more than what they will need to provide for themselves and their families. This according to him will prevent poverty with the entire population

    9. We have to limit fortunes. Our present plan is that we will allow no one man to own more that $50,000,000. We think that with that limit we will be able to carry out the balance of the program. It may be necessary that we limit it to less than $50,000,000. It may be necessary, in working out of the plans that no man’s fortune would be more than $10,000,000 or $15,000,000. But be that as it may, it will still be more than any one man, or any one man and his children and their children, will be able to spend in their lifetimes; and it is not necessary or reasonable to have wealth piled up beyond that point where we cannot prevent poverty among the masses.

      He believes that if ones wealth is limited to 50,000 so that there will be plenty of money to go around for everyone, and if there isn't enough to go around then ones wealth might need to be lowered. He is saying why should one have so much money just sitting there piled up while people are going without. He believes that is not helping out one another with wealth just sitting there being unused.

    10. We have to limit fortunes. Our present plan is that we will allow no one man to own more that $50,000,000. We think that with that limit we will be able to carry out the balance of the program. It may be necessary that we limit it to less than $50,000,000. It may be necessary, in working out of the plans that no man’s fortune would be more than $10,000,000 or $15,000,000. But be that as it may, it will still be more than any one man, or any one man and his children and their children, will be able to spend in their lifetimes; and it is not necessary or reasonable to have wealth piled up beyond that point where we cannot prevent poverty among the masses

      This was part of Huey P. Long's plan of sharing the wealth in America. He was suggesting a plan to limit the amount of money an individual had to have. Setting a cap would have enabled the distribution of wealth among people by taking money from the rich and distributing. He believed that the few rich people had accumulated a lot of wealth piled up while the majority of Americans were living in poverty.

    1. Reviewer #2:

      This paper reports a thorough critique of the ABCD stop-signal data set. It identifies a set of eight problems that severely limits the utility of the ABCD stopping data. In particular, the first two (which are essentially the same problem) invalidate estimates of SSRT based on the independent race model because of violations of the context independence assumption of that model. The remaining issues are more minor in the sense that while potentially problematic they either affect a very small percentage of the data and so can be dealt with by removing the affected trials or participants, or do not appear to be problematic in practice.

      The authors have provided a valuable service to the research community in systematically and thoroughly cataloguing these issues, although we think it is fair to say that a number of people (including the present reviewers) have been aware of the key design issue caused by the stop signal replacing the go signal for quite some time and have been working on solutions.

      Below we have a few suggestions for clarifications, but overall the paper is very clear and well written.

      Although the paper mentions that "new models for stopping must be developed to accommodate context dependence (Bissett et al., 2019), the latter of which we consider to be of utmost importance to advancing the stop-signal literature", it does not discuss such models and neither does it show the potentially severe consequences of context independence violations in the ABCD data set.

      All our more substantive comments relate to "Retroactive Suggestions For Issue 1". First, the authors write: "Given the above, if analyzing or disseminating existing ABCD stopping data, we would recommend caution in drawing any strong conclusions from the stopping data, and any results should be clearly presented with the limitation that the task design encourages context dependence and therefore stopping behavior (e.g., SSRT) and neuroimaging contrasts may be contaminated".

      We feel that this recommendation is too lenient and would suggest the following alternative: Unless the ABCD community conclusively shows that the design flaw does not distort conclusions based on SSRT estimates (or any other stop-signal measure), researchers should not use the ABCD data set to estimate SSRTs at all.

      Second, the authors suggest removing subjects who have severe violations as evidenced by mean stop-failure RT > mean no-stop-signal RT. We are concerned that this recommendation impacts on the representativeness of the sample. Also, this recommendation ignores the fact that violations are not an all-or-none phenomenon but are a matter of degree and can come in varying shapes and sizes.

      Third, the authors recommend that "any results be verified when only longer SSDs are used, perhaps only SSDs > 200ms". Figure 3 does not seem to support the recommended cut-off of 200ms: at 200ms accuracy is still far from asymptotic.

      In general, we feel that recommendations based on removing participants and trials are not sufficient. Such practices will affect the representativeness of the sample and will increase estimation uncertainty and hence decrease power. We believe that the only way to solve Issue 1 is by developing measurement models that can account for the dependence of the go and the stop process.

    1. Author Response

      Reviewer #1:

      This paper addresses the very interesting topic of genome evolution in asexual animals. While the topic and questions are of interest, and I applaud the general goal of a large-scale comparative approach to the questions, there are limitations in the data analyzed. Most importantly, as the authors raise numerous times in the paper, questions about genome evolution following transitions to asexuality inherently require lineage-specific controls, i.e. paired sexual species to compare with the asexual lineages. Yet such data are currently lacking for most of the taxa examined, leaving a major gap in the ability to draw important conclusions here. I also do not think the main positive results, such as the role of hybridization and ploidy on the retention and amount of heterozygosity, are novel or surprising.

      We agree with the reviewer that having the sexual outgroups would improve the interpretations; this is one of the points we make in our manuscript. Importantly however, all previous genome studies of asexual species focus on individual asexual lineages, generally without sexual species for comparison. Yet reported genome features have been interpreted as consequences of asexuality (e.g., Flot et al. 2013). By analysing and comparing these genomes, we can show that these features are in fact lineage-specific rather than general consequences of asexuality. Unexpectedly, we find that asexuals that are not of hybrid origin are largely homozygous, independently of the cellular mechanism underlying asexuality. This contrasts with the general view that cellular mechanisms such as central fusion (which facilitates heterozygosity retention between generation) promotes the evolutionary success of asexual lineages relative to mechanisms such as gamete duplication (which generate complete homozygosity) by delaying the expression of the recessive load. We also do not observe the expected relationship between cellular mechanism of asexuality and heterozygosity retention in species of hybrid origin. Thus we respectfully disagree that our results are not surprising. Reviewer #2 found our results “interesting” and a “potentially important contribution”, and reviewer #3 wrote that we “call into question the generality of the theoretical expectations, and suggest that the genomic impacts of asexuality may be more complicated than previously thought”.

      We also make it very clear that some of the patterns we uncover (e.g. low TE loads in asexual species) cannot be clearly evaluated with asexuals alone. Our study emphasizes the importance of the fact that asexuality is a lineage-level trait and that comparative analyses using asexuals requires lineage-level replication in addition to comparisons to sexual species.

      References

      Flot, Jean-François, et al. "Genomic evidence for ameiotic evolution in the bdelloid rotifer Adineta vaga." Nature 500.7463 (2013): 453-457.

      Reviewer #2:

      [...] Major Issues and Questions:

      1) The authors choose to refer to asexuality when describing thelytokous parthenogenesis. Asexuality is a very general term that can be confusing: fission, vegetative reproduction could also be considered asexuality. I suggest using parthenogenesis throughout the manuscript for the different animal clades studied here. Moreover, in thelytokous parthenogenesis meiosis can still occur to form the gametes, it is therefore not correct to write that "gamete production via meiosis... no longer take place" (lines 57-58). Fertilization by sperm indeed does not seem to take place (except during hybridogenesis, a special form of parthenogenesis).

      We will clarify more explicitly what asexuality refers to in our manuscript. Notably our study does not include species that produce gametes which are fertilized (which is the case under hybridogenesis, which sensu stricto is not a form of parthenogenesis). Even though many forms of parthenogenesis do indeed involve meiosis (something we explain in much detail in box 2), there is no production of gametes.

      2) The cellular mechanisms of asexuality in many asexual lineages are known through only a few, old cytological studies and could be inaccurate or incomplete (for example Triantaphyllou paper of 1981 of Meloidogyne nematodes or Hsu, 1956 for bdelloid rotifers). The authors should therefore mention in the introduction the lack of detailed and accurate cellular and genetic studies to describe the mode of reproduction because it may change the final conclusion.

      For example, for bdelloid rotifers the literature is scarce. However the authors refer in Supp Table 1 to two articles that did not contain any cytological data on oogenesis in bdelloid rotifers to indicate that A. vaga and A. ricciae use apomixis as reproductive mode. Welch and Meselson studied the karyotypes of bdelloid rotifers, including A. vaga, and did not conclude anything about absence or presence of chromosome homology and therefore nothing can be said about their reproduction mode. In the article of Welch and Meselson the nuclear DNA content of bdelloid species is measured but without any link with the reproduction mode. The only paper referring to apomixis in bdelloids is from Hsu (1956) but it is old and new cytological data with modern technology should be obtained.

      We will correct the rotifer citations and thank the reviewer for picking up the error. We agree that there are uncertainties in some cytological studies, but the same is true for genomic studies (which is why we base our analyses as much as possible on raw reads rather than assemblies because the latter may be incorrect). We in fact excluded cytological studies where the findings could not be corroborated. For example, we discarded the evidence for meiosis and diploidy by Handoo at al. 2004 for its incompatibility with genomic data because this study does not provide any verifiable evidence (there are no data or images, only descriptions of observations). We provide all the references in the supplementary material concerning the cytological evidence used.

      3) In the section on Heterozygosity, the authors compute heterozygosity from kmer spectra analysis from reads to "avoid biases from variable genome assembly qualities" (page 16). But such kmer analysis can be biased by the quality and coverage of sequencing reads. While such analyses are a legitimate tool for heterozygosity measurements, this argument (the bias of genome quality) is not convincing and the authors should describe the potential limits of using kmer spectra analyses.

      We excluded all the samples with unsuitable quality of data (e.g. one tardigrade species with excessive contamination or the water flea samples for insufficient coverage), and T. Rhyker Ranallo Benavidez, the author of the method we used, collaborated with us on the heterozygosity analyzes. However, we will clarify the limitations of the method for species with extremely low or high heterozygosity (see also comment 5 of this reviewer).

      4) The authors state that heterozygosity levels “should decay over time for most forms of meiotic asexuality". This is incorrect, as this is not expected with "central fusion" or with "central fusion automixis equivalent" where there is no cytokinesis at meiosis I.

      Our statement is correct. Note that we say “most” and not “all” because certain forms of endoduplication in F1 hybrids result in the maintenance of heterozygosity. Central fusion is expected to fully retain heterozygosity only if recombination is completely suppressed (see for example Suomalainen et al. 1987 or Engelstädter 2017).

      5) I do not fully agree with the authors’ statement that: "In spite of the prediction that the cellular mechanism of asexuality should affect heterozygosity, it appears to have no detectable effect on heterozygosity levels once we control for the effect of hybrid origins (Figure 2)." (page 17)

      The scaling on Figure 2 is emphasizing high values, while low values are not clearly separated. By zooming in on the smaller heterozygosity % values we may observe a bigger difference between the "asexuality mechanisms". I do not see how asexuality mechanism was controlled for, and if you look closely at intra group heterozygosity, variability is sometimes high.

      It is expected that hybrid origin leads to higher heterozygosity levels but saying that asexuality mechanism is not important is surprising: on Figure 2 the orange (central fusion) is always higher than yellow (gamete duplication).

      As we explain in detail in the text, the three comparatively high heterozygosity values under spontaneous origins of asexuality (“orange” points in the bottom left corner of the figure) are found in an only 40-year old clone of the Cape bee. Among species of hybrid origin, we see no correlation between asexuality mechanism and heterozygosity. These observations suggest that the asexuality mechanism may have an impact on genome-wide heterozygosity in recent incipient asexual lineages, but not in established asexual lineages.

      Also, the variability found within rotifers could be an argument against a strong importance of asexuality origin on heterozygosity levels: the four bdelloid species likely share the same origin but their allelic heterozygosity levels appears to range from almost 0 to almost 6% (Fig 2 and 3, however the heterozygosity data on Rotaria should be confirmed, see below).

      We prefer not using the data from rotifers for making such arguments, given the large uncertainty with respect to genome features in this group (including the possibility of octoploidy in some species which we describe in the supplemental information). One could even argue that the highly variable genome structure among rotifer species could indicate repeated transitions to asexuality and/or different hybridization events, but the available genome data would make all these arguments highly speculative.

      The authors’ main idea (i.e. asexuality origin is key) seems mostly true when using homoeolog heterozygosity and/or composite heterozygosity which is not what most readers will usually think as "heterozygosity". This should be made clear by the authors mostly because this kind of heterozygosity does not necessarily undergo the same mechanism as the one described in Box 2 for allelic heterozygosity. If homoeolog heterozygosity is sometimes not distinguishable from allelic heterozygosity, then it would be nice to have another box showing the mechanisms and evolution pattern for such cases (like a true tetraploid, in which all copies exist).

      The heterozygosity between homoeologs is always high in this study while it appears low between alleles, but since the heterozygosity between homeologs can only be measured when there is a hybrid origin, the only heterozygosity that can be compared between ALL the asexual groups is the one between alleles.

      By definition, homoeologs have diverged between species, while alleles have diverged within species. So indeed divergence between homoeologs will generally exceed divergence between alleles. We will consider adding expected patterns in perfect tetraploid species for Box 2.

      Both in the results and the conclusion the authors should not over interpret the results on heterozygosity. The variation in allelic heterozygosity could be small (although not in all asexuals studied) also due to the age of the asexual lineages. This is not mentioned here in the result/discussion section..

      We explain in section Overview of species and genomes studied that age effects are important but that we do not consider them quantitatively because age estimates are not available for the majority of asexual species in our paper.

      6) Regarding the section on Heterozygosity structure in polyploids

      There is inconsistency in many of the numbers. For example, A. vaga heterozygosity is estimated at 1.42% in Figure 1, but then appears to show up around 2% in Figure 2, and then becomes 2.4% on page 20. It is unclear is this is an error or the result of different methods.

      It is also unclear how homologs were distinguished from homeologs. How are 21 bp k-mers considered homologous? In the method section. the authors describe extracting unique k-mer pairs differing by one SNP, so does this mean that no more than one SNP was allowed to define heterozygous homologous regions? Does this mean that homologues (and certainly homoeologs) differing by more than 5% would not be retrieved by this method. If so, then It is not surprising that for A.vaga is classified as a diploid.

      Figure 1 a presents the values reported in the original genome studies, not our results. This is explained in the corresponding figure legend. Hence, 1.42 is the value reported by Flot at al. 2013. 2.4 is the value we measure and it is consistent in Figures 2 and 3.

      We used k-mer pairs differing by one SNP to estimate ploidy (smudgeplot). The heterozygosity estimates were estimated from kmer spectra (GenomeScope 2.0). The kmers that are found in 1n must be heterozygous between homologs, as the homoeolog heterozygosity would produce 2n kmers, We used the kmer approach to estimate heterozygosity in all other cases than homoeologs of rotifers, which were directly derived from the assemblies. We explain this in the legend to Figure 3, but we will add the information also to the Methods section for clarification.

      The result for A. ricciae is surprising and I am still not convinced by the octoploid hypothesis. In Fig S2. there is a first peak at 71x coverage that still could be mostly contaminants. It would be helpful to check the GC distribution of k-mers in the first haploid peak of A. ricciae to check whether there are contaminants. The karyotypes of 12 chromosomes indeed do not fit the octoploid hypothesis. I am also surprised by the 5.5% divergence calculated for A. ricciae, this value should be checked when eliminating potential contaminants (if any). In general, these kind of ambiguities will not be resolved without long-read sequencing technology to improve the genome assemblies of asexual lineages.

      We understand the scepticism of the reviewer regarding the octoploidy hypothesis, but it is important to note that we clearly present it as a possible explanation for the data that needs to be corroborated, i.e., we state that the data are better consistent with octo- than tetraploidy. Contamination seems quite unlikely, as the 71.1x peak represents nearly exactly half the coverage of the otherwise haploid peak (142x). Furthermore, the Smudgeplot analysis shows that some of the kmers from the 71x peak pair with genomic kmers of the main peaks. We also performed KAT analysis (not presented in the manuscript) showing that these kmers are also represented in the decontaminated assembly. We will add this clarification regarding possible contamination to the supplementary materials.

      7) Regarding the section on palindromes and gene conversion

      The authors screened all the published genomes for palindromes, including small blocks, to provide a more robust unbiased view. However, the result will be unbiased and robust if all the genomes compared were assembled using the same sequencing data (quality, coverage) and assembly program. While palindromes appear not to play a major role in the genome evolution of parthenogenetic animals since only few palindromes were detected among all lineages, mitotic (and meiotic) gene conversion is likely to take place in parthenogens and should indeed be studied among all the clades.

      We agree with the reviewer that gene conversion might be one of the key aspects of asexual genome evolution. Our study merely pointed out that genomes of asexual animals do not show organisation in palindromes, indicating that palindromes might not be of general importance in asexual genome evolution. Note also that we clearly point out that these analyses are biased by the quality of the available genome assemblies.

      8) Regarding the section on transposable elements

      The authors are aware that the approach used may underestimate the TEs present in low copy numbers, therefore the comparison might underestimate the TE numbers in certain asexual groups.

      Yes. We clearly explain this limitation in the manuscript. The currently available alternatives are based on assembled genomes, so the results are biased by the quality of the assemblies (and similarities to TEs in public databases) and our aim was to broadly compare genomes in the absence of assembly-generated biases.

      9) Regarding the section on horizontal gene transfer. For the HGTc analysis, annotated genes were compared to the UniRef90 database to identify non-metazoan genes and HGT candidates were confirmed if they were on a scaffold containing at least one gene of metazoan origin. While this method is indeed interesting, it is also biased by the annotation quality and the length of the scaffolds which vary strongly between studies.

      Yes, this is true and we explain many limitations in the supplemental information, but re-assembling and re-annotating all these genomes would be beyond reasonable computational possibilities.

      10) Regarding the use of GenomeScope2.0

      When homologues are very divergent (as observed in bdelloid rotifers) GenomeScope probably considers these distinct haplotypes as errors, making it difficult to model the haploid genome size and giving a high peak of errors in the GenomeScope profile. Moreover, due to the very divergent copies in A. vaga, GenomeScope indeed provides a diploid genome (instead of tetraploid).

      For A. vaga, the heterozygosity estimated par GenomeScope2.0. on our new sequencing dataset is 2% (as shown in this paper). This % corresponds to the heterozygosity between k-mers but does not provide any information on the heterogeneity in heterozygosity measurements along the genome. A limitation of GenomeScope2.0. (which the authors should mention here) is that it is assuming that the entire genome is following the same theoretical k-mer distribution.

      The model of estimating genome wide heterozygosity indeed assumes a random distribution of heterozygous loci and indeed is unable to estimate divergence over a certain threshold, which is the reason why we used genome assemblies for the estimation of divergence of homoeologs. Regarding estimates in all other genomes, the assumptions are unlikely to fundamentally change the output of the analysis. GenomeScope2 is described in detail in a recent paper (Ranallo-Benavidez et al. 2019), where the assumption that heterozygosity rates are constant across the genome is explicitly mentioned.

      References

      Engelstädter, Jan. "Asexual but not clonal: evolutionary processes in automictic populations." Genetics 206.2 (2017): 993-1009.

      Flot, Jean-François, et al. "Genomic evidence for ameiotic evolution in the bdelloid rotifer Adineta vaga." Nature 500.7463 (2013): 453-457.

      Handoo, Z. A., et al. "Morphological, molecular, and differential-host characterization of Meloidogyne floridensis n. sp.(Nematoda: Meloidogynidae), a root-knot nematode parasitizing peach in Florida." Journal of nematology 36.1 (2004): 20.

      Suomalainen, Esko, Anssi Saura, and Juhani Lokki. Cytology and evolution in parthenogenesis. CRC Press, 1987.

      Ranallo-Benavidez, Timothy Rhyker, Kamil S. Jaron, and Michael C. Schatz. "GenomeScope 2.0 and Smudgeplots: Reference-free profiling of polyploid genomes." BioRxiv (2019): 747568. 

      Reviewer #3:

      Jaron and collaborators provide a large-scale comparative work on the genomic impact of asexuality in animals. By analysing 26 published genomes with a unique bioinformatic pipeline, they conclude that none of the expected features due to the transition to asexuality is replicated across a majority of the species. Their findings call into question the generality of the theoretical expectations, and suggest that the genomic impacts of asexuality may be more complicated than previously thought.

      The major strengths of this work is (i) the comparison among various modes and origins of asexuality across 18 independent transitions; and (ii) the development of a bioinformatic pipeline directly based on raw reads, which limits the biases associated with genome assembly. Moreover, I would like to acknowledge the effort made by the authors to provide on public servers detailed methods which allow the analyses to be reproduced. That being said, I also have a series of concerns, listed below:

      We thank this reviewer for the relevant comments and for providing many constructive suggestions in the points below. We will take them into account for our final version of the manuscript.

      1) Theoretical expectations

      As far as I understand, the aim of this work is to test whether 4 classical predictions associated with the transition to asexuality and 5 additional features observed in individual asexual lineages hold at a large phylogenetic scale. However, I think that these predictions are poorly presented, and so they may be hardly understood by non-expert readers. Some of them are briefly mentioned in a descriptive way in the Introduction (L56 - 61), and with a little more details in the Boxes 1 and 2. However, the evolutive reasons why one should expect these features to occur (and under which assumptions) is not clearly stated anywhere in the Introduction (but only briefly in the Results & Discussion). I think it is important that the authors provide clear-cut quantitative expectations for each genomic feature analysed and under each asexuality origin and mode (Box 1 and 2). Also highlighting the assumptions behind these expectations will help for a better interpretation of the observed patterns.

      We will clarify the expectations for non expert readers.

      2) Mutation accumulation & positive selection

      A subtlety which is not sufficiently emphasized to my mind is that the different modes of asexuality encompass reproduction with or without recombination (Box 2), which can lead to very different genetic outcomes. For example, it has been shown that the Muller's ratchet (the accumulation of deleterious mutations in asexual populations) can be stopped by small amounts of recombination in large-sized populations (Charlesworth et al. 1993; 10.1017/S0016672300031086). Similarly a new recessive beneficial mutation can only segregate at a heterozygous state in a clonal lineage (unless a second mutation hits the same locus); whereas in the presence of recombination, these mutations will rapidly fix in the population by the formation of homozygous mutants (Haldane's Sieve, Haldane 1927; 10.1017/S0305004100015644). Therefore, depending on whether recombination occurs or not during asexual reproduction, the expectations may be quite different; and so they could deviate from the "classical predictions". In this regard, I would like to see the authors adjust their conclusions. Moreover, it is also not very clear whether the species analysed here are 100% asexuals or if they sometimes go through transitory sexual phases, which could reset some of the genomic effects of asexuality.

      Yes, the predictions regarding the efficiency of selection are indeed influenced by cellular modes of asexuality. Adding some details or at least a good reference would certainly increase the readability of the section. We thank the reviewer for this suggestion.

      3) Transposable elements

      I found the predictions regarding the amount of TEs expected under asexuality quite ambiguous. From one side, TEs are expected not to spread because they cannot colonize new genomes (Hickey 1982); but on the other side TEs can be viewed as any deleterious mutation that will accumulate in asexual genome due to the Muller's ratchet. The argument provided by the authors to justify the expectation of low TE load in asexual lineages is that "Only asexual lineages without active TEs, or with efficient TE suppression mechanisms, would be able to persist over evolutionary timescales". But this argument should then equally be applied to any other type of deleterious mutations, and so we won't be able to see Muller's ratchet in the first place. Therefore, not observing the expected pattern for TEs in the genomic data is not so surprising as the expectation itself does not seem to be very robust. I would like the authors to better acknowledge this issue, which actually goes into their general idea that the genomic consequences of asexuality are not so simple.

      Indeed, the survivorship bias should affect all genomic features. Nothing that is incompatible with the viability of the species will ever be observed in nature. Perhaps the difference between Muller’s ratchet and the dynamics of accumulation of transposable elements (TEs) is that TEs are expected to either propagate very fast or not at all (Dolgin and Charlesworth 2006), while the effects of Muller’s ratchet are expected to vary among different populations and cellular mechanisms of asexuality. We will rephrase the text to better reflect the complexity of the predicted consequences of TE dynamics.

      4) Heterozygosity

      Due to the absence of recombination, asexual populations are expected to maintain a high level of diversity at each single locus (heterozygosity), but a low number of different haplotypes. However, as presented by the authors in the Box 2, there are different modes of parthenogenesis with different outcomes regarding heterozygosity: (1) preservation at all loci; (2) reduction or loss at all loci; (3) reduction depending on the chromosomal position relative to the centromere (distal or proximal). Therefore, the authors could benefit from their genome-based dataset to explore in more detail the distribution of heterozygosity along the chromosomes, and further test whether it fits with the above predictions. If the differing quality of the genome assemblies is an issue, the authors could at least provide the variance of the heterozygosity across the genome. The mode #3 (i.e. central fusions and terminal fusions) would be particularly interesting as one would then be able to compare, within the same genome, regions with large excess vs. deficit of heterozygosity and assess their evolutive impacts.

      Moreover, the authors should put more emphasis on the fact that using a single genome per species is a limitation to test the subtle effects of asexuality on heterozygosity (and also on "mutation accumulation & positive selection"). These effects are better detected using population-based methods (i.e. with many individuals, but not necessarily many loci). For example, the FIS value of a given locus is negative when its heterozygosity is higher than expected under random mating, and positive when the reverse is true (Wright 1951; 10.1111/j.1469-1809.1949.tb02451.x).

      We agree with the reviewer that the analysis of the distribution of heterozygosity along the chromosomes would be very interesting. However, the necessary data is available only for the Cape honey bee, and its analysis has been published by Smith et al. 2018. Calculating the probability distribution of heterozygosities would be possible, but it would require SNP calling for each of the datasets. Such an analysis would be computationally intensive and prone to biases by the quality of the genome assemblies.

      5) Absence of sexual lineages

      A second limit of this work is the absence of sexual lineages to use as references in order to control for lineage-specific effects. I do not agree with the authors when they say that "the theoretical predictions pertaining to mutation accumulation, positive selection, gene family expansions, and gene loss are always relative to sexual species [...] and cannot be independently quantified in asexuals." I think that this is true for all the genomic features analysed, because the transition to asexuality is going to affect the genome of asexual lineages relative to their sexual ancestors. This is actually acknowledged at the end of the Conclusion by the authors.

      To give an example, the authors say that "Species with an intraspecific origin of asexuality show low heterozygosity levels (0.03% - 0.83%), while all of the asexual species with a known hybrid origin display high heterozygosity levels (1.73% - 8.5%)". Interpreting these low vs. high heterozygosity values is difficult without having sexual references, because the level of genetic diversity is also heavily influenced by the long term life history strategies of each species (e.g. Romiguier et al. 2014; 10.1038/nature13685).

      I understand that the genome of related sexual species are not available, which precludes direct comparisons with the asexual species. However, I think that the results could be strengthened if the authors provided for each genomic feature that they tested some estimates from related sexual species. Actually, they partially do so along the Result & Discussion section for the palindromes, transposable elements and horizontal gene transfers. I think that these expectations for sexual species (and others) could be added to Table 1 to facilitate the comparisons.

      Our statement "the theoretical predictions pertaining to mutation accumulation, positive selection, gene family expansions, and gene loss are always relative to sexual species [...] and cannot be independently quantified in asexuals." specifically refers to methodology: analyses to address these predictions require orthologs between sexual and asexual species. We fully agree that in addition to methodological constraints, comparisons to sexual species are also conceptually relevant - which is in fact one of the major points of our paper. We will clarify these points.

      6) Regarding statistics, I acknowledge that the number of species analysed is relatively low (n=26), which may preclude getting any significant results if the effects are weak. However, the authors should then clearly state in the text (and not only in the reporting form) that their analyses are descriptive. Also, their position regarding this issue is not entirely clear as they still performed a statistical test for the effect of asexuality mode / origin on TE load (Figure 2 - supplement 1). Therefore, I would like to see the same statistical test performed on heterozygosity (Figure 2).

      We will unify the sections and add an appropriate test everywhere where suited.

      7) As you used 31 individuals from 26 asexual species, I was wondering whether you make profit of the multi-sample species. For example, were the kmer-based analyses congruent between individuals of the same species?

      Unfortunately, some of the 31 individuals do not have publicly available reads (some of the root-knot nematode datasets are missing), others do not have sufficient quality (the coverage for some water flea samples is very low). Our analyses were consistent for the few cases where we have multiple datasets available.

      References

      Dolgin, Elie S., and Brian Charlesworth. "The fate of transposable elements in asexual populations." Genetics 174.2 (2006): 817-827.

      Smith, Nicholas MA, et al. "Strikingly high levels of heterozygosity despite 20 years of inbreeding in a clonal honey bee." Journal of evolutionary biology 32.2 (2019): 144-152.

    2. Reviewer #3

      Jaron and collaborators provide a large-scale comparative work on the genomic impact of asexuality in animals. By analysing 26 published genomes with a unique bioinformatic pipeline, they conclude that none of the expected features due to the transition to asexuality is replicated across a majority of the species. Their findings call into question the generality of the theoretical expectations, and suggest that the genomic impacts of asexuality may be more complicated than previously thought.

      The major strengths of this work is (i) the comparison among various modes and origins of asexuality across 18 independent transitions; and (ii) the development of a bioinformatic pipeline directly based on raw reads, which limits the biases associated with genome assembly. Moreover, I would like to acknowledge the effort made by the authors to provide on public servers detailed methods which allow the analyses to be reproduced. That being said, I also have a series of concerns, listed below:

      1) Theoretical expectations.

      As far as I understand, the aim of this work is to test whether 4 classical predictions associated with the transition to asexuality and 5 additional features observed in individual asexual lineages hold at a large phylogenetic scale. However, I think that these predictions are poorly presented, and so they may be hardly understood by non-expert readers. Some of them are briefly mentioned in a descriptive way in the Introduction (L56 - 61), and with a little more details in the Boxes 1 and 2. However, the evolutive reasons why one should expect these features to occur (and under which assumptions) is not clearly stated anywhere in the Introduction (but only briefly in the Results & Discussion). I think it is important that the authors provide clear-cut quantitative expectations for each genomic feature analysed and under each asexuality origin and mode (Box 1 and 2). Also highlighting the assumptions behind these expectations will help for a better interpretation of the observed patterns.

      2) Mutation accumulation & positive selection.

      A subtlety which is not sufficiently emphasized to my mind is that the different modes of asexuality encompass reproduction with or without recombination (Box 2), which can lead to very different genetic outcomes. For example, it has been shown that the Muller's ratchet (the accumulation of deleterious mutations in asexual populations) can be stopped by small amounts of recombination in large-sized populations (Charlesworth et al. 1993; 10.1017/S0016672300031086). Similarly a new recessive beneficial mutation can only segregate at a heterozygous state in a clonal lineage (unless a second mutation hits the same locus); whereas in the presence of recombination, these mutations will rapidly fix in the population by the formation of homozygous mutants (Haldane's Sieve, Haldane 1927; 10.1017/S0305004100015644). Therefore, depending on whether recombination occurs or not during asexual reproduction, the expectations may be quite different; and so they could deviate from the "classical predictions". In this regard, I would like to see the authors adjust their conclusions. Moreover, it is also not very clear whether the species analysed here are 100% asexuals or if they sometimes go through transitory sexual phases, which could reset some of the genomic effects of asexuality.

      3) Transposable elements.

      I found the predictions regarding the amount of TEs expected under asexuality quite ambiguous. From one side, TEs are expected not to spread because they cannot colonize new genomes (Hickey 1982); but on the other side TEs can be viewed as any deleterious mutation that will accumulate in asexual genome due to the Muller's ratchet. The argument provided by the authors to justify the expectation of low TE load in asexual lineages is that "Only asexual lineages without active TEs, or with efficient TE suppression mechanisms, would be able to persist over evolutionary timescales". But this argument should then equally be applied to any other type of deleterious mutations, and so we won't be able to see Muller's ratchet in the first place. Therefore, not observing the expected pattern for TEs in the genomic data is not so surprising as the expectation itself does not seem to be very robust. I would like the authors to better acknowledge this issue, which actually goes into their general idea that the genomic consequences of asexuality are not so simple.

      4) Heterozygosity.

      Due to the absence of recombination, asexual populations are expected to maintain a high level of diversity at each single locus (heterozygosity), but a low number of different haplotypes. However, as presented by the authors in the Box 2, there are different modes of parthenogenesis with different outcomes regarding heterozygosity: (1) preservation at all loci; (2) reduction or loss at all loci; (3) reduction depending on the chromosomal position relative to the centromere (distal or proximal). Therefore, the authors could benefit from their genome-based dataset to explore in more detail the distribution of heterozygosity along the chromosomes, and further test whether it fits with the above predictions. If the differing quality of the genome assemblies is an issue, the authors could at least provide the variance of the heterozygosity across the genome. The mode #3 (i.e. central fusions and terminal fusions) would be particularly interesting as one would then be able to compare, within the same genome, regions with large excess vs. deficit of heterozygosity and assess their evolutive impacts.

      Moreover, the authors should put more emphasis on the fact that using a single genome per species is a limitation to test the subtle effects of asexuality on heterozygosity (and also on "mutation accumulation & positive selection"). These effects are better detected using population-based methods (i.e. with many individuals, but not necessarily many loci). For example, the FIS value of a given locus is negative when its heterozygosity is higher than expected under random mating, and positive when the reverse is true (Wright 1951; 10.1111/j.1469-1809.1949.tb02451.x).

      5) Absence of sexual lineages.

      A second limit of this work is the absence of sexual lineages to use as references in order to control for lineage-specific effects. I do not agree with the authors when they say that "the theoretical predictions pertaining to mutation accumulation, positive selection, gene family expansions, and gene loss are always relative to sexual species [...] and cannot be independently quantified in asexuals." I think that this is true for all the genomic features analysed, because the transition to asexuality is going to affect the genome of asexual lineages relative to their sexual ancestors. This is actually acknowledged at the end of the Conclusion by the authors.

      To give an example, the authors say that "Species with an intraspecific origin of asexuality show low heterozygosity levels (0.03% - 0.83%), while all of the asexual species with a known hybrid origin display high heterozygosity levels (1.73% - 8.5%)". Interpreting these low vs. high heterozygosity values is difficult without having sexual references, because the level of genetic diversity is also heavily influenced by the long term life history strategies of each species (e.g. Romiguier et al. 2014; 10.1038/nature13685).

      I understand that the genome of related sexual species are not available, which precludes direct comparisons with the asexual species. However, I think that the results could be strengthened if the authors provided for each genomic feature that they tested some estimates from related sexual species. Actually, they partially do so along the Result & Discussion section for the palindromes, transposable elements and horizontal gene transfers. I think that these expectations for sexual species (and others) could be added to Table 1 to facilitate the comparisons.

      6) Regarding statistics, I acknowledge that the number of species analysed is relatively low (n=26), which may preclude getting any significant results if the effects are weak. However, the authors should then clearly state in the text (and not only in the reporting form) that their analyses are descriptive. Also, their position regarding this issue is not entirely clear as they still performed a statistical test for the effect of asexuality mode / origin on TE load (Figure 2 - supplement 1). Therefore, I would like to see the same statistical test performed on heterozygosity (Figure 2).

      7) As you used 31 individuals from 26 asexual species, I was wondering whether you make profit of the multi-sample species. For example, were the kmer-based analyses congruent between individuals of the same species?

    3. Reviewer #2

      This paper is interesting because it is studying, through a comparative genomic approach, how asexuality affects genome evolution in animal lineages while focusing on the same features. Such an extensive comparison can, in principle, distinguish the common consequences of asexuality, in contrast to previous studies that focused on few asexual species (or only one). It is interesting that the authors did not find a universal genomic feature of "asexual" species. This is a potentially important contribution to the field of the evolution of reproductive systems.

      However, I am concerned about limitations and potential biases in many of the specific genomic features analysed, and resultant difficulties in drawing any general conclusions from these analyses. For example, the heterozygosity analyses need to be more clearly explained and the potential limits of the methods used discussed further. The use of kmer spectra analyses as opposed to genome assemblies is understandable, but these are biases here that were not discussed. I am also concerned about the impact of low read quality and low coverage genomic data, and whether issues with genome assembly affect the conclusions. There are also issues about conclusions related to species of hybrid origin as there are numerous "unknown" cases and cytological data is lacking for many of the studied animal groups (therefore the authors should be cautious on the evidence of reproduction mode).

      Ideally, all the genomes of the asexual animal clades studied should have been sequenced and assembled using the same method which would make this comparative study much stronger. We realize this may not yet be practical, but the absence of such data must temper the conclusions. It is nevertheless the first article including and comparing many distinct parthenogenetic animal clades and the main result that no common universal genomic feature of parthenogenesis is, with caveats, interesting.

      Major Issues and Questions:

      1) The authors choose to refer to asexuality when describing thelytokous parthenogenesis. Asexuality is a very general term that can be confusing: fission, vegetative reproduction could also be considered asexuality. I suggest using parthenogenesis throughout the manuscript for the different animal clades studied here. Moreover, in thelytokous parthenogenesis meiosis can still occur to form the gametes, it is therefore not correct to write that "gamete production via meiosis... no longer take place" (lines 57-58). Fertilization by sperm indeed does not seem to take place (except during hybridogenesis, a special form of parthenogenesis).

      2) The cellular mechanisms of asexuality in many asexual lineages are known through only a few, old cytological studies and could be inaccurate or incomplete (for example Triantaphyllou paper of 1981 of Meloidogyne nematodes or Hsu, 1956 for bdelloid rotifers). The authors should therefore mention in the introduction the lack of detailed and accurate cellular and genetic studies to describe the mode of reproduction because it may change the final conclusion.

      For example, for bdelloid rotifers the literature is scarce. However the authors refer in Supp Table 1 to two articles that did not contain any cytological data on oogenesis in bdelloid rotifers to indicate that A. vaga and A. ricciae use apomixis as reproductive mode. Welch and Meselson studied the karyotypes of bdelloid rotifers, including A. vaga, and did not conclude anything about absence or presence of chromosome homology and therefore nothing can be said about their reproduction mode. In the article of Welch and Meselson the nuclear DNA content of bdelloid species is measured but without any link with the reproduction mode. The only paper referring to apomixis in bdelloids is from Hsu (1956) but it is old and new cytological data with modern technology should be obtained.

      3) In the section on Heterozygosity, the authors compute heterozygosity from kmer spectra analysis from reads to "avoid biases from variable genome assembly qualities" (page 16). But such kmer analysis can be biased by the quality and coverage of sequencing reads. While such analyses are a legitimate tool for heterozygosity measurements, this argument (the bias of genome quality) is not convincing and the authors should describe the potential limits of using kmer spectra analyses.

      4) The authors state that heterozygosity levels “should decay over time for most forms of meiotic asexuality". This is incorrect, as this is not expected with "central fusion" or with "central fusion automixis equivalent" where there is no cytokinesis at meiosis I.

      5) I do not fully agree with the authors’ statement that: "In spite of the prediction that the cellular mechanism of asexuality should affect heterozygosity, it appears to have no detectable effect on heterozygosity levels once we control for the effect of hybrid origins (Figure 2)." (page 17)

      The scaling on Figure 2 is emphasizing high values, while low values are not clearly separated. By zooming in on the smaller heterozygosity % values we may observe a bigger difference between the "asexuality mechanisms". I do not see how asexuality mechanism was controlled for, and if you look closely at intra group heterozygosity, variability is sometimes high.

      It is expected that hybrid origin leads to higher heterozygosity levels but saying that asexuality mechanism is not important is surprising: on Figure 2 the orange (central fusion) is always higher than yellow (gamete duplication). Also, the variability found within rotifers could be an argument against a strong importance of asexuality origin on heterozygosity levels: the four bdelloid species likely share the same origin but their allelic heterozygosity levels appears to range from almost 0 to almost 6% (Fig 2 and 3, however the heterozygosity data on Rotaria should be confirmed, see below).

      The authors’ main idea (i.e. asexuality origin is key) seems mostly true when using homoeolog heterozygosity and/or composite heterozygosity which is not what most readers will usually think as "heterozygosity". This should be made clear by the authors mostly because this kind of heterozygosity does not necessarily undergo the same mechanism as the one described in Box 2 for allelic heterozygosity. If homoeolog heterozygosity is sometimes not distinguishable from allelic heterozygosity, then it would be nice to have another box showing the mechanisms and evolution pattern for such cases (like a true tetraploid, in which all copies exist).

      The heterozygosity between homoeologs is always high in this study while it appears low between alleles, but since the heterozygosity between homeologs can only be measured when there is a hybrid origin, the only heterozygosity that can be compared between ALL the asexual groups is the one between alleles.

      Both in the results and the conclusion the authors should not over interpret the results on heterozygosity. The variation in allelic heterozygosity could be small (although not in all asexuals studied) also due to the age of the asexual lineages. This is not mentioned here in the result/discussion section.

      6) Regarding the section on Heterozygosity structure in polyploids.

      There is inconsistency in many of the numbers. For example, A. vaga heterozygosity is estimated at 1.42% in Figure 1, but then appears to show up around 2% in Figure 2, and then becomes 2.4% on page 20. It is unclear is this is an error or the result of different methods.

      It is also unclear how homologs were distinguished from homeologs. How are 21 bp k-mers considered homologous? In the method section. the authors describe extracting unique k-mer pairs differing by one SNP, so does this mean that no more than one SNP was allowed to define heterozygous homologous regions? Does this mean that homologues (and certainly homoeologs) differing by more than 5% would not be retrieved by this method. If so, then It is not surprising that for A. vaga is classified as a diploid.

      The result for A. ricciae is surprising and I am still not convinced by the octoploid hypothesis. In Fig S2. there is a first peak at 71x coverage that still could be mostly contaminants. It would be helpful to check the GC distribution of k-mers in the first haploid peak of A. ricciae to check whether there are contaminants. The karyotypes of 12 chromosomes indeed do not fit the octoploid hypothesis. I am also surprised by the 5.5% divergence calculated for A. ricciae, this value should be checked when eliminating potential contaminants (if any). In general, these kind of ambiguities will not be resolved without long-read sequencing technology to improve the genome assemblies of asexual lineages.

      7) Regarding the section on palindromes and gene conversion.

      The authors screened all the published genomes for palindromes, including small blocks, to provide a more robust unbiased view. However, the result will be unbiased and robust if all the genomes compared were assembled using the same sequencing data (quality, coverage) and assembly program. While palindromes appear not to play a major role in the genome evolution of parthenogenetic animals since only few palindromes were detected among all lineages, mitotic (and meiotic) gene conversion is likely to take place in parthenogens and should indeed be studied among all the clades.

      8) Regarding the section on transposable elements.

      The authors are aware that the approach used may underestimate the TEs present in low copy numbers, therefore the comparison might underestimate the TE numbers in certain asexual groups.

      9) Regarding the section on horizontal gene transfer.

      For the HGTc analysis, annotated genes were compared to the UniRef90 database to identify non-metazoan genes and HGT candidates were confirmed if they were on a scaffold containing at least one gene of metazoan origin. While this method is indeed interesting, it is also biased by the annotation quality and the length of the scaffolds which vary strongly between studies.

      10) Regarding the use of GenomeScope2.0.

      When homologues are very divergent (as observed in bdelloid rotifers) GenomeScope probably considers these distinct haplotypes as errors, making it difficult to model the haploid genome size and giving a high peak of errors in the GenomeScope profile. Moreover, due to the very divergent copies in A. vaga, GenomeScope indeed provides a diploid genome (instead of tetraploid).

      For A. vaga, the heterozygosity estimated par GenomeScope2.0. on our new sequencing dataset is 2% (as shown in this paper). This % corresponds to the heterozygosity between k-mers but does not provide any information on the heterogeneity in heterozygosity measurements along the genome. A limitation of GenomeScope2.0. (which the authors should mention here) is that it is assuming that the entire genome is following the same theoretical k-mer distribution.

    1. In that case, we might want to think about how one clarification or another might affect our assessment.  We can specify in our assessment that we support the argument if it means what we think it means but not if it means something else.

      Not too clear. What do you mean by "one clarification or another?" Do you mean a clarification needed in a side point as opposed to a central point? And how does the next sentence follow? This last sentence in the little paragraph doesn't seem to have to do with the first. Are the three sentences three different examples of how the problem may not be too serious? In that case, you have to make that clearer.

    1. Author Response

      Reviewer #1:

      The Lambowitz group has developed thermostable group II intron reverse transcriptases (TGIRTs) that strand switch and also have trans-lesion activity to provide a much wider view of RNA species analyzed by massively parallel RNA sequencing. In this manuscript they use several improvements to their methodology to identify RNA biotypes in human plasma pooled from several healthy individuals. Additionally, they implicate binding by proteins (RBPs) and nuclease-resistant structures to explain a fraction of the RNAs observed in plasma. Generally I find the study fascinating and argue that the collection of plasma RNAs described is an important tool for those interested in extracellular RNAs. I think the possibility that RNPs are protecting RNA fragments in circulation is exciting and fits with elegant studies of insects and plants where RNAs are protected by this mechanism and are transmitted between species.

      I have one major comment for the authors to consider. In my view the use of pooled plasma samples prevented the important opportunity to provide a glimpse on human variation in plasma RNA biotypes. This significantly limits the use of this information to begin addressing RNA biotypes as biomarkers. While I realize that data from multiple individuals represents a significant undertaking and may be beyond the scope of this manuscript, I urge the authors to do two things: (1) downplay the significance of the current study on the development of biomarkers in the current manuscript (e.g., in the abstract and discussion - e.g., "The ability of TGIRT-seq to simultaneously profile a wide variety of RNA biotypes in human plasma, including structured RNAs that are intractable to retroviral RTs, may be advantageous for identifying optimal combinations of coding and non-coding RNA biomarkers for human diseases."). (2) Carry out an analysis in multiple individuals - including racially diverse individuals - very important information will come of this - similar to C. Burge's important study in Nature ~2008 where it was clear that there is important individual variation in alternative splicing decisions - very likely genetically determined. This second suggestion could be added here or constitute a future manuscript.

      The identification of biomarkers in human plasma is an important application of this study, as was noted by reviewer 3 -- "Overall, this study provided a robust dataset and expanded picture of RNA biotypes one can detect in human plasma. This is valuable because the findings may have implications in biomarker identification in disease contexts." The present manuscript lays the foundation for such applications, which we have been carrying out in parallel. In one such study in collaboration with Dr. Naoto Ueno (MD Anderson), we used TGIRT-seq to identify combinations of mRNA and non-coding RNA biomarkers in FFPE-tumor slices, PBMCs and plasma from inflammatory breast cancer patients compared to non-IBC breast cancer patients and healthy controls (manuscript in preparation; data presented publicly in seminars), and in another, we explored the potential of using full-length excised intron (FLEXI) RNAs as biomarkers. In the latter study, we identified >8,000 FLEXI RNAs in different human cell lines and tissues and found that they are expressed in a cell-type specific manner, including hundreds of differences between matched tumor and healthy tissues from breast cancer patients and cell lines. A manuscript describing the latter findings was submitted for publication after this one and has been uploaded as a pertinent related manuscript. This new manuscript follows directly from the last sentence of the present manuscript and fully references the BioRxiv preprint currently under review for eLife.

      Reviewer #2:

      Yao et al used thermostable group II intron reverse transcriptase sequencing (TGIRT-seq) to study apheresis plasma samples. The first interesting discovery is that they had identified a number of mRNA reads with putative binding sites of RNA-binding proteins. A second interesting discovery from this work is the detection of full-length excised intron RNAs.

      I have the following comments:

      1) One doubt that I have is how representative is apheresis plasma when compared with plasma that one obtains through routine centrifugation of blood. The authors have reported the comparison of apheresis plasma versus a single male plasma in a previous publication. I think that to address this important question, a much increased number of samples would be necessary.

      Detailed comparison of plasma prepared by apheresis to that prepared by centrifugation would require a separate large-scale study, preferably by multiple laboratories using different methods to prepare plasma. However, our impression both from our findings and from the literature (Valbonesi et al. 2001, cited in the manuscript) is that apheresis-prepared plasma has very low levels of cellular contamination (required to meet clinical standards) compared to plasma prepared by centrifugation, even with protocols designed to minimize contamination from intact 4 or broken cell (e.g., preparing plasma from freshly drawn blood, centrifugation into a Ficoll cushion to minimize cell breakage, and carefully avoiding contamination from sedimented cells).

      We do have additional information about the degree of variation in protein-coding gene transcripts detected by TGIRT-seq in plasma samples prepared by centrifugation from five healthy females controls in our collaborative study with Dr. Naoto Ueno (M.D. Anderson; see above), and we have added it to the manuscript citing a manuscript in preparation with permission from Dr. Ueno (p. 10, beginning line 6 from bottom) as follows:

      “The identities and relative abundances of different protein-coding gene transcripts in the apheresis-prepared plasma were broadly similar to those in the previous TGIRT analysis of plasma prepared by Ficoll-cushion sedimentation of blood from a healthy male individual (Qin et al., 2016) (r = 0.62-0.80; Figure 3C) and between high quality plasma samples similarly prepared from five healthy females in a collaborative study with Dr. Naoto Ueno, M.D. Anderson (r = 0.53-0.67; manuscript in preparation).” See Author Response Image below.

      2) For the important conclusion of the presence of binding sites of RNA-binding proteins in a proportion of apheresis plasma mRNA molecules, the authors need to explore whether there is any systemic difference in terms of mapping quality (i.e. mapping quality scores in alignment results) between RBP binding sites and non-RBP binding sites, so that any artifacts of peaks caused by the alignment issues occurring in RNA-seq analysis could be revealed and solved subsequently. Furthermore, it would be prudent to perform immunoprecipitation experiments to confirm this conclusion in at least a proportion of the mRNA.

      We have added a figure panel comparing MAPQ scores for reads from peaks containing RBP-binding site to other long RNA reads (Figure 4–figure supplement 2A) and have added further details about the methods used to obtain peaks with high quality reads, including the following (p. 13, beginning line 3 from the bottom).

      “After further filtering to remove read alignments with MAPQ <30 (a cutoff that eliminates reads mapping equally well at more than one locus) or ≥5 mismatches from the mapped locus, we were left with 950 high confidence peaks ranging in size from 59 to 1,207 nt with ≥5 high quality read alignments at the peak maximum (Supplementary File).”

      3) In Fig. 2D, one can observe that there are clearly more RNA reads in TGIRT-seq located in the 1st exon of ACTB, compared with SMART-seq. Is there any explanation? Will this signal be called as a peak (a potential RBP binding site) in the peak calling analysis (MACS2)? Is ACTB supposed to be bound by a certain RBP?

      The higher coverage of the ACTB 5'-exon in the TGIRT-seq datasets reflects in part the more uniform 5' to 3' coverage of mRNA sequences by TGIRT-seq compared to SMART-seq, which is biased for 3'-mRNA sequences that have poly(A) tails (current Figure 3F). The signal in the first exon of ACTB was in fact called as a peak by MACS2 (peak ID#893, Supplementary file), which overlapped an annotated binding site for SERBP1 (see Supplementary File).

      4) For Fig 2A, it would be informative for the comparison of RNA yield and RNA size profile among different protocols if the author also added the results of TGIRT-seq.

      Figure 3D (previously Figure 2A) shows a bioanalyzer trace of PCR amplified cDNAs obtained by SMART-Seq. These cDNAs correspond to 3' mRNA sequences that have poly(A) tails and are not comparable to the bioanalyzer profiles of plasma RNA (Figure 1–figure supplement 1) or read span distributions in the TGIRT-seq datasets (Figure 1B), which are dominated by sncRNAs. The coverage plots for protein-coding gene transcripts show that TGIRT-seq captures mRNA fragments irrespective of length that span the entire mRNA sequence, whereas SMART-seq is biased for 3' sequences linked to poly(A) (Figure 3F). We also note that coverage plots and mRNAs detected by TGIRT-seq remain similar, even if the plasma RNA is chemically fragmented prior to TGIRT-seq library construction (Figure 3F and Figure 3–figure supplement 2).

      5) As shown in Figure 4 C (the track of RBP binding sites), it seems quite pervasive in some gene regions. How many RBP binding sites from public eCLIP-seq results are used for overlapping peaks present in TGIRT-seq of plasma RNA? What percentage of plasma RNA reads have fallen within RBP binding sites? Are those peaks present in TGRIT-seq significantly enriched in RBPs binding regions?

      Some of these points are addressed under Reviewer 1-comment #4. Additionally, we noted that 109 RBP-binding sites were searched in the original analysis, and we have now added further analyses for 150 RBPs currently available in ENCODE eCLIP datasets with and without irreproducible discovery rate (IDR) analysis (Figure 6 and Figure 6–figure supplement 1). We have also added a tab to the Supplementary File identifying the 109 and 150 RBPs whose binding sites were searched. The requested statistical analysis has been added in Figure 4–figure supplement 2C. The analysis shows that enrichment of RBP-binding site sequences in the 467 called peaks was statistically significant (p<0.001) (p. 14, para. 3, last sentence).

      6) Since there is a considerable portion of TGIRT-seq reads related to simple repeat, one possible reason is likely the high abundance of endogenous repeat-related RNA species in plasma. Nonetheless, have authors studied whether the ligation steps in TGIRT-seq have any biases (e.g. GC content) when analyzing human reference RNAs and spike ins (page 4, paragraph 2)?

      We have added a note to the manuscript indicating that although repeat RNAs constitute a high proportion of the called peaks, they do not constitute a similarly high proportion of the total RNA reads (Figure 1C; p. 18, para. 2, first sentence). The TGIRT-seq analysis of human reference RNAs and spike-ins showed that TGIRT-seq recapitulates the relative abundance of human transcripts and spike-in comparably to non-strand-specific TruSeq v2 and better than strand-specific TruSeq v3 (Nottingham et al. RNA 2016). Subsequently, we used miRNA reference sets for detailed analysis of TGIRT-seq biases, including developing a computer algorithm for bias correction based on a random forest regression model that provides insight into different factors that contribute to these biases (Xu et al. Sci. Report. 2019). Overall GC content does not make a significant contribution to TGIRT-seq biases (Figure 9 of Xu et al. Sci. Report, 2017). Instead, biases in TGIRT-seq are largely confined to the first three nucleotides at the 5'-end (due to bias of the thermostable 5' App DNA ligase used for 5' RNA-seq adapter addition) and the 3' nucleotide (due to TGIRT-template switching). These end biases are not expected to significantly impact the quantitation of repeat RNAs.

      7) As described in Figure 2 legend, there are 0.25 million deduplicated reads for TGIRT-seq reads assigned to protein-coding genes transcripts which are far less than 2.18 million reads for SMART-seq. The authors need to discuss whether the current protocol of TGIRT-seq would cause potential dropouts in mRNA analysis, compared with SMART-seq?

      We have added the following to the manuscript (p. 11, para. 1, line 15).

      “The larger number of mRNA reads compared to TGIRT-seq (0.28 million) largely reflects that SMART-seq selectively profiles polyadenylated mRNAs, while TGIRT-seq profiles mRNAs together with other more abundant RNA biotypes. In addition, ultra low input SMART-Seq is not strand-specific, resulting in redundant sense and antisense strand reads (Figure 3–figure supplement 1).”

      The manuscript contains the following statement regarding potential drop outs (p. 11, para. 2, line 1).

      “A scatter plot comparing the relative abundance of transcripts originating from different genes showed that most of the polyadenylated mRNAs detected in DNase I-treated plasma RNA by ultra low input SMART-Seq were also detected by TGIRT-seq at similar TPM values when normalized for protein-coding gene reads (r=0.61), but with some, mostly lower abundance mRNAs undetected either by TGIRT-seq or SMART-Seq, and with SMART-seq unable to detect non-polyadenylated histone mRNAs, which are relatively abundant in plasma (Figure 3E and Figure 3–figure supplement 1).”

      8) While scientific thought-provoking, the practical implication of the current work is still unclear. The authors have suggested that their work might have applications for biomarker development. Is it possible to provide one experimental example in the manuscript?

      We addressed the relevance of the manuscript to biomarker identification and noted parallel studies that supports this application in the response to reviewer 1--comment 1. We have also modified the final paragraph of the Discussion (p. 30, para. 2).

      “The ability of TGIRT-seq to simultaneously profile a wide variety of RNA biotypes in human plasma, including structured RNAs that are intractable to retroviral RTs, may be advantageous for identifying optimal combinations of coding and non-coding RNA biomarkers that could then be incorporated in target RNA panels for diagnosis and routine monitoring of disease progression and response to treatment. The finding that some mRNAs fragments persist in discrete called peaks suggests a strategy for identifying relatively stable mRNA regions that may be more reliably detected than other more labile regions in targeted liquid biopsies. Finally, we note that in addition to their biological and evolutionary interest, short full-length excised intron RNAs and intron RNA fragments, such as those identified here, may be uniquely well suited to serve as stable RNA biomarkers, whose expression is linked to that of numerous protein-coding genes."

      Reviewer #3:

      In this work, Yao and colleagues described transcriptome profiling of human plasma from healthy individuals by TGIRT-seq. TGIRT is a thermostable group II intron reverse transcriptase that offers improved fidelity, processivity and strand-displacement activity, as compared to standard retroviral RT, so that it can read through highly structured regions. Similar analysis was performed previously (ref. 20), but this study incorporated several improvements in library preparation including optimization of template switching condition and modified adapters to reduce primer dimer and introduce UMI. In their analysis, the authors detected a variety of structural RNA biotypes, as well as reads from protein-coding mRNAs, although the latter is in low abundance. Compared to SMART-Seq, TGIRT-seq also achieved more uniform read coverage across gene bodies. One novel aspect of this study is the peak analysis of TGIRT-seq reads, which revealed ~900 peaks over background. The authors found that these peaks frequently overlap with RBP binding sites, while others tend to have stable predicted secondary structures, which explains why these regions are protected from degradation in plasma. Overall, this study provided a robust dataset and expanded picture of RNA biotypes one can detect in human plasma. This is valuable because the findings may have implications in biomarker identification in disease contexts. On the other hand, the manuscript, in the current form, is relatively descriptive, and can be improved with a clearer message of specific knowledge that can be extracted from the data.

      Specific points:

      1) Several aspects of bioinformatics analysis can be clarified in more detail. For example, it is unclear how sequencing errors in UMI affect their de-duplication procedure. This is important for their peak analysis, so it should be explained clearly.

      We have added details of the procedure used for de-duplication to the following paragraph in Materials and methods (p. 35, para. 2).

      “Deduplication of mapped reads was done by UMI, CIGAR string, and genome coordinates (Quinlan, 2014). To accommodate base-calling and PCR errors and non-templated nucleotides that may have been added to the 3' ends of cDNAs during TGIRT-seq library preparation, one mismatch in the UMI was allowed during deduplication, and fragments with the same CIGAR string, genomic coordinates (chromosome start and end positions), and UMI or UMIs that differed by one nucleotide were collapsed into a single fragment. The counts for each read were readjusted to overcome potential UMI saturation for highly-expressed genes by implementing the algorithm described in (Fu et al., 2011), using sequencing tools (https://github.com/wckdouglas/sequencing_tools ).”

      Also, it is not described how exon junction reads (when mapped to the genome) are handled in peak calling, although the authors did perform complementary analysis by mapping reads to the reference transcriptome.

      We have added this to first sentence of the paragraph describing peak calling against the transcriptome reference (p. 16, line 4), which now reads as follows:

      "Peak calling against the human genome reference sequence might miss RBP-binding sites that are close to or overlap exon junctions, as such reads were treated by MACS2 as long reads that span the intervening intron."

      2) Overall, the authors provided convincing data that TGIRT-seq has advantages in detecting a wide range of RNA biotypes, especially structured RNAs, compared to other protocols, but these data are more confirmatory, rather than completely new findings (e.g., compared to ref. 20).

      As indicated in the response to Reviewer 1, comment 2, we modified the first paragraph of the Discussion to explicitly describe what is added by the present manuscript compared to Qin et al. RNA 2016 (p. 24, para. 2). Additionally, further analysis in response to the reviewers' comments resulted in the interesting finding that stress granule proteins comprised a high proportion of the RBPs whose binding sites were enriched in plasma RNAs (to our knowledge a completely new finding), consistent with a previously suggested link between RNP granules, EV packing, and RNA export (p. 16, last sentence; data shown in Figure 6 and Figure 6–figure supplement 1). Also highlighted in the Discussion p. 26, last sentence, continuing on p. 27).

      3) The peak analysis is more novel. The authors observed that 50% of peaks in long RNAs overlap with eCLIP peaks. However, there is no statistical analysis to show whether this overlap is significant or simply due to the pervasive distribution of eCLIP peaks. In fact, it was reported by the original authors that eCLIP peaks cover 20% of the transcriptome.

      We have added statistical analysis, which shows that the enrichment of RBP-binding sites in the 467 called peaks is statistically significant at p<0.001 (p. 14, para. 3, last sentence; Figure 4–Figure supplement 2C), as well as scatter plots identifying proteins whose binding sites were more highly represented in plasma than cellular RNAs or vice versa (p. 16, last two sentences; Figure 6 and Figure 6-figure supplement 1).

      Similarly, the authors found that a high proportion of remaining peaks can fold into stable secondary structures, but this claim is not backed up by statistics either.

      First, near the beginning of the paragraph describing these findings, we added the following to provide a guide as to what can and can't be concluded by RNAfold (p. 17, line 6 from the bottom).

      "To evaluate whether these peaks contained RNAs that could potentially fold into stable secondary structures, we used RNAfold, a tool that is widely used for this purpose with the understanding that the predicted structures remain to be validated and could differ under physiological conditions or due to interactions with proteins."

      Second, at the end of the same paragraph, we have added the requested statistics (p. 18, para. 1, last sentence).

      "Subject to the caveats above regarding conclusions drawn from RNAfold, simulations using peaks randomly generated from long RNA gene sequences indicated that enrichment of RNAs with more stable secondary structures (lower MFEs) in the called RNA peaks was statistically significant (p≤0.019; Figure 4–figure supplement 2D)."

      4) Ranking of RBPs depends on the total number of RBP binding sites detected by eCLIP, which is determined by CLIP library complexity and sequencing depth. This issue should be at least discussed.

      We have added scatter plots in Figure 6 and Figure 6–figure supplement 1, which show that the relative abundance of different RBP-binding sites detected in plasma differs markedly from that for cellular RNAs in the eCLIP datasets (both for the 109 RBPs searched initially and for 150 RBPs with or without irreproducible discovery rate (IDR) analysis from the ENCODE web site,) As mentioned in comments above, this analysis identified a number of RBP-binding sites that were substantially enriched in plasma RNAs compared to cellular RNAs or vice versa and led to what we think is the important new finding that plasma RNAs are enriched binding sites for a number of stress granule proteins (Figure 6 and Figure 6–figures supplement 1). We thank the reviewers for this and related comments that led to this additional analysis.

      5) Enrichment of RBP binding sites and structured RNA in TGIRT-seq data is certainly consistent with one's expectation. However, the paper can be greatly improved if the authors can make a clearer case of what is new that can be learned, as compared to eCLIP data or other related techniques that purify and sequence RNA fragments crosslinked to proteins. What is the additional, independent evidence to show the predicted secondary structures are real?

      Compared to CLIP and related methods, peak calling enables more facile identification of candidate RBPs and putatively structured RNAs for further analysis and may be particularly useful for the vanishingly small amounts of RNA present in plasma and other bodily fluids. New findings resulting from peak calling in the present manuscript include that plasma RNAs are enriched in binding sites for stress granule proteins (see above) and the discovery of a variety of novel RNAs, including the full-length excised intron RNAs first identified here and subsequently studied in cellular RNAs in the Yao et al. pertinent submitted manuscript. We also note that peak calling enables the identification of protein-protected and structured mRNA regions that are relatively stable in plasma and may be more reliably detected in targeted liquid biopsy assays than are more labile mRNA regions (p. 17, para. 1, last sentence; and p. 30, para. 2, beginning on line 5).

      6) The authors should probably discuss how alignment errors can potentially affect detection of repetitive regions.

      In the Empirical Bayes method that we used for the analysis of repeats, repeat sequences were quantified by aggregate counts irrespective of the genomic locus to which they mapped (Materials and methods, p. 38, para. 2, line 5), which should not be affected by alignment errors.

      7) Many figures are IGV screenshots, which can be difficult to follow. Some of them can probably be summarized to deliver the message better.

      Some IGV-based figures are crucial for showing key features of the RNAs that are called as peaks (e.g., the predicted secondary structures of the full-length excised intron RNAs and intron RNA fragments). However, in the process of reformatting, we have switched in and added non-IGV main text figures including Figure 2 (microbiome analysis), Figure 3 (TGIRT-seq versus SMART-Seq), Figure 4 (repeats), and Figure 6 (new figure comparing relative abundance of RBP-binding sites in plasma versus cells).

    1. On the third day Abraham looked up and saw the place far away.

      I would imagine that on a long, excruciating journey, Abraham could have been suffering silently. Trying to make peace with sacrificing your child is a heavy weight to carry, aside from the material weight of their supplies. Additionally, if we view Abraham’s silence as obedience, I think it’s important to remember that obeying a command and trusting the one who gave it to you are different things. Abraham’s actions may have more to do with his relationship to God than his true feelings.

    1. What hap-pens in practice is evidence of this: we take delight in viewing the most accurate possible in1ages of objects which in then1s~lves cause distress when we see them (e.g. the shapes of the lowest species of 6 3,2 EARLY.HISTORY animal, and corpses)

      At first I had trouble understanding Aristotle's point here. Then, after one or two rereads I didn't think this take remained true today, mainly because I was so hung up on the idea of taking delight in imitations of objects that cause us "distress." My initial thought was that nobody would like replications or pictures of images they don't like, but the more I thought about it I've realized that our society may actually have a fascination with replications of distressing objects. The best example that came to mind was how prolific images of skulls are in our world today. While a real life skull, with the exception of being seen in museums or even class rooms for historical/educational purposes, would be offputing to many, replications of skulls have become increasingly popularized. From skulls being popular images as jewelry, to popular halloween decorations/costumes, to being used in hazard/warning signs, skull imagery can be seen everywhere, today. While this is only one example (another prominent example that comes to mind is how gruesome cartoons can be, which people like, even though the real life image wouldn't be appealing), I found it fascinating how this claim has remained so true throughout history.

    1. “moral education”

      <br>

      Analytic notes:

      The primacy of moral education in the design of the French primary school system is evidenced in the writings of Francois Guizot, the French Minister of Education who during the July Monarchy drafted the 1833 Loi that established, for the first time, a centrally-controlled system of primary schools, with a centralized curriculum and primary school teachers trained and recruited according to central government directives. The centrality of moral education as a motivation to centralize the control and expand the provision of primary schooling in France is also discussed by Weber (1976). The 1833 Loi is especially important because it was followed by the fastest expansion of primary schooling observed in French history (Grew and Harrigan 1991).

      That the Prussian primary school system focused mainly on moral and religious education was perceived to be true among those who admired and those who criticized Prussian schools. Among the admirers was Horace Mann, a U.S. education reformer who played a key role in the development of a system of common schools in Massachusetts during the 1830s and 40s. Mann devoted two of the twelve Annual Reports he wrote as Secretary of the Massachusetts Board of Education entirely to the importance of moral education. He highlighted Prussia as an example of an education system that shapes moral values. The Prussian emphasis on moral and religious education noticed by Mann continued to prevail at the beginning of the 20th century according to Thomas Alexander, a professor at the George Peabody College of Teachers who spent between 1912 and 1913 studying Prussian schools directly as the basis for his book The Prussian Elementary Schools, published in 1919.

      Source excerpts:

      In an essay discussing the perceived benefits of the 1833 Loi, Guizot (1860) writes: “The state must provide primary education to all families and give it to those who cannot afford it; and in this he does more for the moral life of peoples than he can do for their material condition. This is the true principle on this point, and it was the one adopted by my bill.” (p. 63-64) “Primary education is not a panacea which cures all the moral maladies of the people . . . ; it is a salutary or harmful power according to whether it is well or badly directed and contained within its limits or pushed out of its mission. . . . The law of June 28, 1833, has received various modifications . . .; but all its principles, all its essential dispositions, have remained upright and in force. Founded by this law, primary education is now, among us, a public institution and an acquired fact. There remains, surely, much to be done for the good government of the schools, to make it dominant in their bosom the influences of religion and order, of faith and law, which make both the dignity and the safety of a people.” (p.86)

      And continues: “If it is true that the attachment of the citizens to the fundamental laws of the state and to the sovereign who is its leader, is the most energetic power and surest safeguard of society, if it is true that there where this feeling has existed, it has produced miracles, and that its absence has brought about the greatest evils, it is the duty as well as the interest of the government to favor and direct its development. But this feeling can only arise from the concordance of public doctrines and national habits with political institutions, the nature and principles of government. . . . When men have learned from childhood to understand the fundamental laws of the country and to respect its sovereign, the sovereign and the laws become a kind of property which is dear to them, and they do not refuse the obligations that it imposes upon them: when the government has taken care to propagate, in favor of national education, in the relations of religion, morality, politics, etc., the doctrines which suit his nature and his direction, these doctrines soon acquire a power against which come the failures of liberty of mind and all seditious attempts. Thus the public mind is formed, thus a true patriotism is maintained, thus fortifying and consolidating societies and thrones.” (Guizot 1860, 86).

      Weber (1976, 329): “Official reports coupled poor education with rude, brutal ways. Where schooling did not take hold, ‘ways are coarse, characters are violent, excitable, and hotheaded, troubles and brawls are frequent.’ The school was supposed to improve manners and customs, and soothe the savage breast. The polite forms it inculcated ‘softened the savagery and harshness natural of peasants.’ Improved behavior and morality would be attributed to the effects of schooling.”

      Mann (1844, 22): “If Prussia can pervert the benign influence of education to the support of arbitrary power, we surely can employ them for the support and perpetuation of republican institutions… If a moral power over the understandings and affections of the people may be turned to evil, may it not also be employed for good?”

      Alexander (1919, v): “A careful study of the Prussian school system will convince any unbiased reader that the Prussian citizen cannot be free to do and act for himself; that the Prussian is to a large measure enslaved through the medium of his school; that his learning, instead of making him his own master, forges the chain by which he is held in servitude; that the whole scheme of Prussian elementary education is shaped with the express purpose of making ninety-five out of every hundred citizens subservient to the ruling house and to the state.”

      In a later chapter that focuses on the curriculum and teaching methods, Alexander refers to the influential ideas of Johann Friedrich Herbart, a German philosopher, psychologist and educator, author of The Moral or Ethical Revelation of the World: The Chief Aim of Education (1804), and Prussian commissioner of education. Alexander (1919, 258) writes that “With Herbart and his followers, the aim and end of instruction was the formation of moral and religious character. It was not Herbart’s intention merely to furnish the child with a definite number of concepts by the form and content of the instruction and subject matter, but rather to shape thereby the will and directly to affect the moral nature and attitude of the child.”

      In the conclusion, Alexander (1919, 540) writes: “Chief among the avowed aims of the Volksschule is the formation of moral and religious character. There is no doubt that the reason religion is made one of the chief subjects of instruction in the elementary schools is to teach the lesson of obedience to authority which is the basis of the German state.” And continues, “There are then leaders and followers. The leaders think and do; the followers merely do. The schools are organized for the express purpose of producing just these types . . . They educate the individual for the state; we [Americans] make the state for the individual. The lesson to learn here is this. The German sets definitely his national aims. Those in authority shape every resource to reach that goal. The schools were molded to meet the needs of the state.” (Alexander 1919, 538-39).

      Full citations:

      Alexander, Thomas. 1919. The Prussian elementary schools. New York: The Macmillan company.

      Grew, Raymond, and Patrick J. Harrigan. 1991. School, state, and society: the growth of elementary schooling in nineteenth-century France: a quantitative analysis. Ann Arbor: University of Michigan Press.

      Guizot, Francois. 1860. Memoires pour servir a l'histoire de mon temps, T.3. Paris: Michel Levy freres.

      Mann, Horace. 1844. Seventh Annual Report on the Board of Education. Boston.

      Weber, Eugen. 1976. Peasants into Frenchmen: The Modernization of Rural France, 1870-1914. Stanford, California: Stanford University Press.

    2. “moral education”

      <br>

      Analytic notes:

      The primacy of moral education in the design of the French primary school system is evidenced in the writings of Francois Guizot, the French Minister of Education who during the July Monarchy drafted the 1833 Loi that established, for the first time, a centrally-controlled system of primary schools, with a centralized curriculum and primary school teachers trained and recruited according to central government directives. The centrality of moral education as a motivation to centralize the control and expand the provision of primary schooling in France is also discussed by Weber (1976). The 1833 Loi is especially important because it was followed by the fastest expansion of primary schooling observed in French history (Grew and Harrigan 1991).

      That the Prussian primary school system focused mainly on moral and religious education was perceived to be true among those who admired and those who criticized Prussian schools. Among the admirers was Horace Mann, a U.S. education reformer who played a key role in the development of a system of common schools in Massachusetts during the 1830s and 40s. Mann devoted two of the twelve Annual Reports he wrote as Secretary of the Massachusetts Board of Education entirely to the importance of moral education. He highlighted Prussia as an example of an education system that shapes moral values. The Prussian emphasis on moral and religious education noticed by Mann continued to prevail at the beginning of the 20th century according to Thomas Alexander, a professor at the George Peabody College of Teachers who spent between 1912 and 1913 studying Prussian schools directly as the basis for his book The Prussian Elementary Schools, published in 1919.

      Source excerpts:

      In an essay discussing the perceived benefits of the 1833 Loi, Guizot (1860) writes: “The state must provide primary education to all families and give it to those who cannot afford it; and in this he does more for the moral life of peoples than he can do for their material condition. This is the true principle on this point, and it was the one adopted by my bill.” (p. 63-64) “Primary education is not a panacea which cures all the moral maladies of the people . . . ; it is a salutary or harmful power according to whether it is well or badly directed and contained within its limits or pushed out of its mission. . . . The law of June 28, 1833, has received various modifications . . .; but all its principles, all its essential dispositions, have remained upright and in force. Founded by this law, primary education is now, among us, a public institution and an acquired fact. There remains, surely, much to be done for the good government of the schools, to make it dominant in their bosom the influences of religion and order, of faith and law, which make both the dignity and the safety of a people.” (p.86)

      And continues: “If it is true that the attachment of the citizens to the fundamental laws of the state and to the sovereign who is its leader, is the most energetic power and surest safeguard of society, if it is true that there where this feeling has existed, it has produced miracles, and that its absence has brought about the greatest evils, it is the duty as well as the interest of the government to favor and direct its development. But this feeling can only arise from the concordance of public doctrines and national habits with political institutions, the nature and principles of government. . . . When men have learned from childhood to understand the fundamental laws of the country and to respect its sovereign, the sovereign and the laws become a kind of property which is dear to them, and they do not refuse the obligations that it imposes upon them: when the government has taken care to propagate, in favor of national education, in the relations of religion, morality, politics, etc., the doctrines which suit his nature and his direction, these doctrines soon acquire a power against which come the failures of liberty of mind and all seditious attempts. Thus the public mind is formed, thus a true patriotism is maintained, thus fortifying and consolidating societies and thrones.” (Guizot 1860, 86).

      Weber (1976, 329): “Official reports coupled poor education with rude, brutal ways. Where schooling did not take hold, ‘ways are coarse, characters are violent, excitable, and hotheaded, troubles and brawls are frequent.’ The school was supposed to improve manners and customs, and soothe the savage breast. The polite forms it inculcated ‘softened the savagery and harshness natural of peasants.’ Improved behavior and morality would be attributed to the effects of schooling.”

      Mann (1844, 22): “If Prussia can pervert the benign influence of education to the support of arbitrary power, we surely can employ them for the support and perpetuation of republican institutions… If a moral power over the understandings and affections of the people may be turned to evil, may it not also be employed for good?”

      Alexander (1919, v): “A careful study of the Prussian school system will convince any unbiased reader that the Prussian citizen cannot be free to do and act for himself; that the Prussian is to a large measure enslaved through the medium of his school; that his learning, instead of making him his own master, forges the chain by which he is held in servitude; that the whole scheme of Prussian elementary education is shaped with the express purpose of making ninety-five out of every hundred citizens subservient to the ruling house and to the state.”

      In a later chapter that focuses on the curriculum and teaching methods, Alexander refers to the influential ideas of Johann Friedrich Herbart, a German philosopher, psychologist and educator, author of The Moral or Ethical Revelation of the World: The Chief Aim of Education (1804), and Prussian commissioner of education. Alexander (1919, 258) writes that “With Herbart and his followers, the aim and end of instruction was the formation of moral and religious character. It was not Herbart’s intention merely to furnish the child with a definite number of concepts by the form and content of the instruction and subject matter, but rather to shape thereby the will and directly to affect the moral nature and attitude of the child.”

      In the conclusion, Alexander (1919, 540) writes: “Chief among the avowed aims of the Volksschule is the formation of moral and religious character. There is no doubt that the reason religion is made one of the chief subjects of instruction in the elementary schools is to teach the lesson of obedience to authority which is the basis of the German state.” And continues, “There are then leaders and followers. The leaders think and do; the followers merely do. The schools are organized for the express purpose of producing just these types . . . They educate the individual for the state; we [Americans] make the state for the individual. The lesson to learn here is this. The German sets definitely his national aims. Those in authority shape every resource to reach that goal. The schools were molded to meet the needs of the state.” (Alexander 1919, 538-39).

      Full citations:

      Alexander, Thomas. 1919. The Prussian elementary schools. New York: The Macmillan company.

      Grew, Raymond, and Patrick J. Harrigan. 1991. School, state, and society: the growth of elementary schooling in nineteenth-century France: a quantitative analysis. Ann Arbor: University of Michigan Press.

      Guizot, Francois. 1860. Memoires pour servir a l'histoire de mon temps, T.3. Paris: Michel Levy freres.

      Mann, Horace. 1844. Seventh Annual Report on the Board of Education. Boston.

      Weber, Eugen. 1976. Peasants into Frenchmen: The Modernization of Rural France, 1870-1914. Stanford, California: Stanford University Press.

    1. Reviewer #3:

      General assessment:

      In this research article, authors claim that HIP1 plays an important role in promoting the proliferative ability of prostate cancer cells by HIP1-STAT3-GDF15 signaling axis activation. HIP1 overexpression increased STAT3 signaling in response to FGF2 receptor activation and increased GDF15 transcription. The increase in GDF15 protein secretion was dependent on HIP1 and STAT3 expression and was shown to have paracrine growth-promoting effects. Although some of the information is new, the relevance and importance of this information is inconclusive and not supported from the data presented in this article.

      Major Comments:

      This paper needs a substantial amount of revision, as indicated below.

      A. Novelty:

      HIP-1 has been extensively studied in cancer including prostate cancer (Rao et al., 2002). Its role in STAT3 signaling has also been demonstrated (Hsu et al, 2015). This study is not very novel.

      B. Major comments:

      1) Figures 1A, S1: Changes in p-AMPK1α, and p-Akt are very profound in this array, however, the authors indicate that "By contrast to our validation of STAT3 phosphorylation by Western blotting, it was not possible to detect increased levels of p-AMPK1α (T174), p-Akt (S473) or p-PLC-γ1 when we attempted to validate these by blotting (Supplementary Figure S1D-F)." Why do the authors think this is happening? Did the authors use the same experimental conditions for the array and validation experiments? These apparent discrepancies need further clarification.

      2) Figure 1E: the authors show that shHIP1#2 caused a modest knockdown of HIP1, while shHIP1#1 induced a dramatic reduction in HIP1 protein level, however, both the shRNAs significantly inhibited pSTAT3 to the same extent. This indicates that total knockdown (KD) of HIP1 is not necessary to completely shut-down the activity of pSTAT3. How does this translate to the biological functions of HIP1?

      3) How come DMSO treatment blocks the phosphorylation of ERK1/2 in lane 2 of Fig 1(F)?

      4) Figure S1F: pSTAT3 western blot: the authors should indicate which band they considered positive for p-STAT3; if it's the lower band why was there no activity in lane 4?

      5) Fig 2A and 2B should be repeated in HIP1 knockout cells.

      6) What is the endogenous level of HIP1 and GDF15 in prostate cancer cell lines vs. normal prostate epithelial cells? Why was HIP1 overexpressed in LNCaP cells? Was the level of HIP1 expression low in LNCaP and PNT1A, when compared in a panel of prostate cancer cell lines? Did the authors observe any differential expression of HIP1 and GDF15 in hormone sensitive vs. hormone resistant prostate cancer cells?

      7) GDF15 is a very ambiguous biomarker of cancer as its levels are even higher in the case of mental disorders including psychosis (for reference https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5554200/ ). And from this study, it is not even clear that the GDF15 upregulation is just one of the several outcomes of the activation of this signaling axis or if it is the only consequence of this signaling axis to promote the growth of cancer cells by increasing paracrine signaling. An experiment in GDF15 knockout cells/mice can document the role of this axis in a more precise manner.

      8) It has been shown that wt p53 significantly reduces STAT3 tyrosine phosphorylation and inhibits STAT3 DNA binding activity in prostate cancer cell lines that express both constitutively active STAT3 and mutant p53 protein. The authors have claimed that the increase in STAT3 phosphorylation is due to HIP1 expression. All three of the cell lines evaluated in this paper have different p53 status and show differences in expression of activated STAT3. Is the expression of HIP1 independent of the status of p53?

      9) Figure 3: Does STAT3 silencing (siRNA/stattic) downregulate HIP1, and does this decrease STAT3 activation over time? Also, does STAT3 silencing or treatment with WP1066 inhibit HIP1-induced tumor growth in vivo?

      10) The role of GDF15 in prostate cancer is likely stage specific. It may promote early stages of tumorigenesis, but suppress the progression of advanced prostate cancers. The authors claim that HIP1 overexpression is mediated by stat3 activation, which leads to increased secretion of GDF15. Does expression of HIP1 correlate with the expression of GDF15 and does this also associate with stage-specific progression of prostate cancer?

      11) How was cellular transformation studied and confirmed? Did HIP1 cause transformation of normal prostate cells?

      12) Fig 1B: HIP1 western blot is not clear, please quantify 1C, 1D, 1E.

      13) Most of the studies are done only in one cell line which is not adequate.

      14) What is the clinical relevance of this study? The authors should study clinical samples along with multiple cell lines.

      15) Several of the Western blot figures need better quality blots; Figs 1E (FGFR), S2C (all).

    1. Bacon says we live through screens. What are these screens? They are part of our normal way of looking at the world, or rather our normal way of seeing the world without looking at it, for Bacon’s claim is that a real seer who looked at the world would notice it to be fairly violent—not violent as narrative surface but somehow violently composed underneath the surface, having violence as its essence

      Bacon's perspective on the world is illustrated through his philosophical belief of living through screens. A screen can be utilized as a divider, but also something one may look through, similar to glass. Based on environmental conditions and/or state of the glass, it can either be transparent, opaque, or cracked. Those who innocently perceive the world are divided from the rest of the corrupt world, not being able to clearly see through the glass/screen. Unlike the normal way of seeing the world, "real seers" can see through the glass/screen clearly. The perspective is based on the conditions one lived through, causing their glass to be transparent or opaque. Do you think the idea of a screened existence is accurate? Why or why not?

    2. Bacon has another term for this catastrophizing: he calls it “destroying clarity with clarity.”(8) Not just in his use of color but in the whole strategy of his compositions, he wants to make us see something we don’t yet have eyes for. He goes inside clarity to a place of deeper refreshment, where clarity is the same and yet differs from itself, which may be analogous to the place inside a word where it falls silent in its own presence

      Bacon "destroys clarity with clarity" by going into depth of the compositions. He does this by not just going through what people can see, such as the "colors", but he goes into a deeper meaning by exaggerating all the points to get his message across. I think Bacon chooses to do this because he wants people to think about the underlying message that he is trying to bring across and have people think more deeply about the meaning. How does Bacon give new perspective to people?

    1. “Hear me, illustrious children of Earth and Sky, 645 that I may speak what my spirit within my breast prompts me to speak. For now a very long space we are fighting, each in opposition to other, concerning victory and power, all our days, the Titan gods and as many of us are sprung from Kronos. 650 Now you must show against the Titans in deadly fight both mighty force and hands invincible, in gratitude for our mild loving-kindness, namely, after how many sufferings you came back again to the light, from afflictive bondage, through our counsels, from the murky gloom.”

      I think it's really interesting how each god/goddess was born with their personality traits, alongside their powers. Here, we witness that Zeus is a natural-born leader, from his radiating confidence through this speech to the instinct to lead the other gods into a fight and into victory. It seemed fit that he should take place as rulers of the heavens.

    2. length the father of gods and men addressed them: “Hear me, illustrious children of Earth and Sky, 645 that I may speak what my spirit within my breast prompts me to speak. For now a very long space we are fighting, each in opposition to other, concerning victory and power, all our days, the Titan gods and as many of us are sprung from Kronos. 650 Now you must show against the Titans in deadly fight both mighty force and hands invincible, in gratitude for our mild loving-kindness, namely, after how many sufferings you came back again to the light, from afflictive bondage, through our counsels, from the murky gloom.” Thus he spoke; and him in turn blameless Kottos addressed in answer

      I think it's pretty cool to see that these gods are in many ways like humans. Their fight for justice, for fair treatment in the face of tyranny has been seen all throughout human history. This passage actually reads as a "pep talk" or a speech to rally the troops and to build camaraderie with one another.

    1. This is a very strong case. Grant Torquemada his premises, and the conclusion follows irresistibly that torturing and burning people may be a duty and a kindness. You can hardly say he was a wicked man for living up more courageously than others to what he and they believed in common, or for acting sincerely in what he believed to be the interest of his victims. We cling, nevertheless, to our conviction that he did wrong, and atrocious wrong. And the question presses more urgently than before, where does the wrongness lie?

      Very interesting perspective -- this reminded me of Donald Trump and what people reference as the 'silent majority'. I think that where the wrongness lies can be subjective based on morals; some people are willing to overlook more controversial choices than others are which is where a divide is created

    Annotators

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      Reply to the reviewers

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

      The manuscript describes two advances. First is the technical development for a protein targeting system called PInT that brings a target protein close to (~320 bp) a DNA sequence of interest. The idea is that localisation of the target protein allows one to distinguish its effects on the DNA sequence either in cis (when targeted) or in trans (when not targeted but expressed at the same level). Since targeting is conveyed by simply adding the small molecule ABA to the experiment, it is easy to compare the two situations. This is a clever idea and it is substantiated by data showing that the components of PInT do not affect triplet repeat instability or gene expression of GFP, into whose gene the PInT system is placed. Moreover, targeting is shown to enable enzymatic activity in the targeted region. Using the DNA methylase DNMT1, there are local increases in DNA methylation. Similarly, targeting the histone deacetylase HDAC5 results in local decreases in histone H3 acetylation.

      We thank the reviewer for a thoughtful and helpful review.

      What is not clear from these experiments, however, is whether the targeted proteins can interact normally with partner proteins to form functional complexes. One necessary control is to add ChIP for at least one interacting protein each for DNMT1 and for HDAC5 and show that targeting permits normal protein-protein interactions. This experiment is straightforward as specific interacting proteins are known and good antibodies to precipitate those proteins are available.

      This is a good suggestion and we plan on doing this experiment in our 59B-Y-HDAC5 and 89B-Y-DNMT1 lines with and without ABA using interacting proteins. The exact interacting protein to be used will depend on the antibodies availability and quality, which we will test. We will start with UHRF1 and HDAC3 for PYL-Dnmt1 and PYL-HDAC5, respectively.

      Overall, PInT would likely be useful for many groups studying the effects of chromatin modifiers on a DNA sequence of interest.

      The second advance is conceptual and is focused more specifically on triplet repeat expansions. The manuscript describes experiments that measure genetic instability of long CAG-CTG repeats with and without protein targeting. The results show that allele size distributions are not significantly affected by targeting either DNMT1 or HDAC5. One curious outcome that is not discussed is contraction frequency in the HDAC5 experiment. Zero contractions are reported compared to 10-20% contractions in the other two experiments. Authors need to provide an explanation.

      Lack of contractions in this experiment is likely due to the lower number of repeats in this line (59 vs 89/91). It is known that longer repeats display higher frequency of contractions, and contractions are rarely seen in short repeats (Larson et al Neurobiology of Disease 2015, Gomes-Pereira et al PLOS Genet 2007, Morales et al HMG 2020). Albeit, the threshold may be different in our HEK293-derived cells. Of note, we had a clone of 89B-Y-HDAC5 that did not express the expected amount of GFP for unknown reasons and we did not use it here. However, small pool PCRs using this line with 89 repeats showed that contractions were indeed present. Although we cannot rule out that the reason for the contractions is the unknown mutation(s), it suggests that the difference is due to the size of the expansion. We have added a comment in the methods section.

      It reads: “We have noted that cell lines with repeats that are mildly expanded (e.g., 59 CAGs) have fewer contractions than longer ones. This is consistent with several studies in the context of DM1 and HD [82], albeit the size threshold for seeing more contractions may be shorter in HEK293-derived cells than in mice.”

      The major issue with this set of experiments is that there is no positive control where instability is shown to be clearly manipulated. A knockdown of FAN1 would be the most likely avenue to pursue for identifying a positive control. This is straightforward to perform since successful FAN1 knockdowns have been described in the literature.

      We agree that a positive control to show that the model behaves as expected is necessary. We will add the experiments proposed by the reviewer in the revised version of the manuscript.

      The manuscript also looks at effects on gene expression measured by GFP fluorescence intensity. The potential significance is to see if disease-causing genes with expanded triplet repeats can be silenced by targeting chromatin-modifying enzymes. In the examples tested here, the answer seems to be no. Expression of DNMT1 or HDAC5 reduce fluorescence even in the absence of targeting. Upon targeting, there is a small further decrease, but the expanded triplet repeat resists this further decrease. Domain analysis of HDAC5 indicates that protein-protein interactions, not deacetylase activity, are important for silencing. The key interaction may be with HDAC3, since small molecule inhibition of HDAC3 relieved repeat length-dependent silencing by HDAC5. It was very curious that targeting HDAC3 actually increased expression, instead of silencing. The explanation for this observation was inadequate.

      We have added the following paragraph to the discussion to address this.

      It reads: “We found that targeting of PYL-HDAC3 increases gene expression slightly, independently of repeat size and in the presence of an inhibitor of its catalytic activity. Although this appears counterintuitive, several studies suggest that this is not unexpected. Specifically, HDAC3 has an essential role in gene expression during mouse development that is independent of its catalytic activity [73]. Moreover, HDAC3 binds more readily to genes that are highly expressed in both human and yeast cells [74,75]. The mechanism or function of HDACs binding to highly expressed genes are currently unknown.”

      The claim on page 16 final paragraph that the manuscript 'settled a central question for both HDAC5 and DNMT1 and their involvement in CAG/CTG repeat instability' is not supported by the data. Most of the results are negative so it is premature to claim the question is 'settled'.

      We have rephrased all the conclusions about this in the text, emphasizing that we find no evidence of a role in cis, rather than stating that there is no role in cis.

      Overall, with appropriate modifications described here, these experiments would be of interest with regards to potential therapies of triplet repeat expansion diseases, where silencing the expanded gene is the goal.

      **Minor concerns**

      P 4, last line. 59 bp should read 59 repeats - This is now fixed.

      P 5, line 2. 38 bp of what? This is now amended. It reads: “The CAG/CTG repeats affect splicing of the reporter in a length-dependent manner, with longer repeats leading to more robust insertion of an alternative CAG exon that includes 38 nucleotides downstream of the CAG, creating a frameshift [30].”

      P 10, first paragraph. DNA methylation levels rise from ~10% to ~20% with DNMT1 targeting. Is there a good precedent in the literature that the magnitude of this increase can be expected to be biologically meaningful?

      To our knowledge, it is the first time that DNMT1 is used for targeted epigenome editing. This is therefore the first evidence that targeting DNMT1 leads to silencing of a reporter construct. Nevertheless, this reviewer’s comment stands: is an increase in DNA methylation of 10 to 20% biologically relevant? The answer to this is yes, changes in 10-20% are known to have functional impact on gene expression in various settings (for example see the recent study in developing oocytes by Li et al Nature 2018). Furthermore, there is evidence that DNMT1 has weak de novo activity (Li et al Nature 2018, Wang et al Nat Genet 2020), consistent with a small increase in CpG methylation upon targeting. We now acknowledge in the discussion that one reason for the lack of effect upon targeting may be that the changes in CpG methylation are not dramatic enough. We also point out more clearly that changes of 10 to 20% are correlated with changes in repeat instability (Dion et al HMG 2008). We have amended the text to reflect this.

      The results now reads “To do so, we performed bisulfite sequencing after targeting PYL-DNMT1 for 30 days. This led to changes of 10 to 20% in the levels of CpG methylation, a modest increase(Fig. 3C), which is in line with the weak de novo methyltransferase activity of DNMT1 (for example see [39,40]). Similar changes in levels of CpG methylation in Dnmt1 heterozygous ovaries and testes were seen to correlate with changes in repeat instability in vivo [31].”

      The discussion now states: “It should be pointed out that there remains the possibility that DNMT1 targeting did not lead to large enough changes in CpG methylation to affect repeat instability.”

      P12 first paragraph. Text describing Fig 5 is confusing. First, GFP expression is referred to in terms of fold decrease, but subsequently in percent. Second, the ABA-induced silencing looks to reduce expression from about 0.6 to 0.5 of control. I presume this is where the claim of 16% comes from but it was not clear. Indeed, this is what we mean.

      We now state: “In 16B-Y-DNMT1 cells, ABA treatment decreased GFP expression by 2.2-fold compared to DMSO treatment alone. Surprisingly, ABA-induced silencing was 1.8 fold compared to DMSO alone, or 16% less efficient in 89B-Y-DNMT1 than in 16B-Y-DNMT1 cells.”

      P 15 paragraph 2. Where does the P value of 0.78 come from? Fig 7B shows no corresponding value. The P-value in figure 7B has now been corrected.

      Reviewer #1 (Significance (Required)):

      See above.

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

      **Summary:**

      We still do not know whether epigenetics contributes to repeat instability and/or transcriptional activity in unstable CAG/CTG repeat associated pathologies. The aim of this manuscript is to examine whether induced binding of DNMT1 (CpG methylation) or HDAC5 (histone H3 acetylation) modulates CAG/CTG repeat instability and/or gene silencing upon expansion. For this the authors developed a highly sophisticated reporter system (PlnT) that allows to recruit a specific chromatin modifying enzyme (DNMT1/ HDAC5) to a GFP reporter near a CAG/CTG expansion, in the course of transcription (Dox-inducible promoter). This is to determine whether the CTGs, when lengthened and transcribed, become unstable or impede gene activity via epigenetic modifications.

      We appreciate the reviewer highlighting the importance of the question that we address here and the usefulness of PInT.

      **Findings:**

      1.Binding of DNMT1 to the reporter results in a modest increase (~10%) in local DNA methylation, with no change in repeat instability.

      3.Targeting HDAC5 to the reporter results in local reduction in histone H3 acetylation, with no effect on repeat stability.

      4.DNMT1/HDAC5 binding reduces GFP intensity differentially, in normal but not expanded alleles.

      5.The N-terminal domain of HDAC5, when mutated, abolishes the reduction in GFP expression levels.

      6.RGFP966 abolishes the allele-specific effect of HDAC5, resulting in a general decrease in GFP expression regardless of repeat tract size

      7.CTG expanded alleles abolish the reduction in GFP repression by HDAC5 via HDAC3 activity

      **Conclusions:**

      Based on the results using the PlnT reporter assay, the authors claim that:

      1.HDAC5 and DNMT1 do not affect repeat instability in cis

      2.Expanded CAG/CTGs reduce the efficiency of gene silencing by targeting DNMT1/HDAC5 to the locus

      3.Gene silencing that is mediated by HDAC5 recruitment can be abolished by inhibition of HDAC3 activity

      Unfortunately, none of the claims in this manuscript are convincing.

      We note that in the comments below the reviewer does not include a reason why he/she does not find the claims convincing. We therefore cannot address this criticism.

      **General Comments:**

      The major drawback of the PlnT experimental approach is that it ignores the importance of the flanking regions and the genomic organization of the endogenous locus. This is a major concern as it makes the conclusions irrelevant to the related loci. In the case of myotonic dystrophy type 1, for example, the reporter should reside within a CpG island, should be positioned immediately next to CTCF binding site(s), and should be transcribed bi-directionally.

      HDAC3 and DNMT1 were found to have effects on repeat instability both at reporters, which do not harbour flanking sequences from disease loci, and indeed at endogenous loci in vivo (Dion et al HMG 2008, Debacker et al PLoS Biol 2012, Suelves et al Sci Rep 2017, Williams et al PNAS 2020). This highlights the fact that cis elements from disease loci are not required for chromatin modifiers to affect repeat instability.

      The reviewer is suggesting a very interesting set of experiments where specific sequences may be added to our reporter and tested for their influence on gene expression and on repeat instability. PInT is ideally suited for this and we have now added a paragraph highlighting this in the discussion. We have also highlighted that the current study aims to isolate the repeats from its cis-elements to specifically side-step potential locus-specific effects and to look for chromatin modifiers that would be useful for epigenome editing for as many loci as possible.

      Furthermore, only large expansions (at least several hundred copies) can trigger heterochromatin at the DM1 locus. None of these features are recapitulated by the PlnT reporter assay, making it difficult to draw any conclusion regarding the role of these chromatin modifying enzymes to the locus.

      This is true for DM1 but untrue for other disease loci. For example, we have shown that there are changes in the flanking chromatin marks at the SCA1 locus of a mouse model with 145 repeats (Dion et al HMG 2008), DNA methylation is also affected near a SCA7 transgene with 92 CAG repeats (Libby et al PLoS Genet 2008) and transgenes containing CAG repeats (without the flanking sequences) lead to silencing regardless of where the transgene is integrated in the genome (Saveliev et al Nature 2003). Moreover, HDAC5 had effects on repeat expansion in a cell-based shuttle system containing as few as 22 CAG repeats (Gannon et al NAR 2012), again suggesting that chromatin modifiers affect repeat instability in a wide range of repeat sizes. We have reviewed this in Dion and Wilson TiG 2009.

      In fact, the authors state in their Discussion that "targeting a chromatin modifying peptide to different loci can have very different effects"!

      This is indeed the case and the reason why we sought to control for locus-specific effects using an exogenous reporter.

      To better substantiate their conclusions the authors must set up an improved model system that takes into account the flanking regions and the 3D genomic organization of the locus (TADs). The preferable approach would be to insert a reporter cassette by homologous recombination into the differentially methylated/acetylated regions near the repeats, and compare between normal vs. expanded alleles.

      We would like to point out that we have recently published a study where we looked at 3D chromatin folding at the DM1, HD, and the GFP transgene used here. We did not find any evidence for changes in TADs that would underlie changes in repeat instability at these loci (Ruiz Buendia et al Sci Advances 2020). We therefore do not think that it would be important to further manipulate 3D genomic organization in this context.

      To be clear, we are not denying that cis elements are likely to have an effect, there is plenty of evidence supporting this. Rather, we are using a reporter assay to disentangle the potential locus-specific (or cis-element specific) effects from the trans-activating factors. In short, we focus on the trans-acting factors rather than on the cis-elements, as suggested by the reviewer.

      We believe that the addition of the following paragraph highlights the goal of our study and also bring in the idea that cis acting elements can be studied using PInT.

      It now reads:

      “We designed PInT specifically to isolate expanded repeats tracts from other potential locus-specific cis elements. This is helpful to identify factors that would affect instability and/or gene expression across several diseases. Moreover, both HDAC3 and DNMT1 were found to impact repeat instability at different loci, including at reporter genes [31,33,36,37,45]. These observations highlight that cis-acting elements from disease loci are not required by chromatin modifiers to affect repeat instability. A potential application of PInT includes cloning in specific cis elements, including CTCF binding sites and CpG islands, next to the repeat tract and evaluate their effects on instability with or without targeting. In fact, PInT can be used to clone any sequence of interest near the targeting site and can be applied for a wide array of applications, beyond the study of expanded CAG/CTG repeats.”

      My impression was that there is a lot of data but none of it makes sense.

      The focus of the manuscript is not entirely clear: it starts with monitoring the effect of epigenetics on repeat instability and gene activity, then it shifts to the mechanism by which HDAC5 functions, and ends with the allele-specific effect of HDAC5 on gene expression. I lost my train of thought.

      We have now improved the transitions in this new version of this manuscript. Specifically, at the core of this manuscript is the development of PInT, which is highly versatile and allowed us to study multiple aspects of expanded CAG/CTG repeat biology. We hope that it is now clearer.

      **Other concerns:**

      (1)the modest increase in methylation levels following DNMT1 recruitment (10%, reaching a total of 20% at the most) prevents from drawing any conclusions regarding the effect of methylation on stability or expression.

      As mentioned in the response to reviewer 1 above, although 10% to 20% of CpG methylation are associated with changes in gene expression in a variety of settings, we now point out that one reason for the lack of effect in cis is that the de novo activity of DNMT1 is too weak to produce an effect.

      (2)The effect of protein targeting on GFP levels should be better defined at the RNA/protein level. Does it act by blocking transcription? alternative splicing? or alters steady state levels?

      Although the exact mechanism remains unclear, this goes beyond the current scope of this study. All these possibilities remain possible as we pointed out in the discussion.

      (3)Fig 5: the scale is different for A vs. B and C. Also, better to compare the effect of targeting on equal sized expansions (either 91, 89 or 58 repeats).

      We have fixed the scale on the figures.

      Unfortunately, it is not possible to have the same repeat sizes for all the cell lines because by their very nature, repeats are unstable. We have added a note relating to this in the methods.

      It reads: “Notably, it is not possible to obtain several stable lines with the exact same repeat size as they are, by their nature, highly unstable. This is why we have lines with different repeat sizes. Furthermore, the sizes can change over time and upon thawing.”

      (4)Add asterix for significance in all figures.

      This has now been done.

      (5)Figure 6: show raw data rather than normalized.

      We have now added representative flow cytometry profiles for each construct as a new supplementary figure (S5).

      (6)Figure 7: there is a notable difference in GFP expression levels in untreated wild type control (16 CAG repeats) between A vs. B. Why?

      Fig. 7a shows PYL targeting only, whereas 7b shows the GFP expression upon PYL-HDAC5 targeting. The values for PYL-HDAC5 targeting are lower because targeting it, unlike targeting PYL alone, silences the reporter.

      (7)Avoid redundancy. No need to show schematic representations so many times.

      We believe that the schematics make it clearer for the reader.

      Reviewer #2 (Significance (Required)):

      REFEREES CROSS-COMMENTING

      I totally agree with the Reviewer #1 that the PinT targeting system is a potent experimental tool to study the function of specific chromatin binding proteins. However, the significance of the flanking regions is discounted.

      We hope it is now clear that we are not discounting the potential significance of flanking regions and that rather we have designed the system to avoid their potentially complicating effects.

      The fact that the recruitment of HDAC5 has resulted in a significant reduction in acetylated histones provides evidence for that "the targeted proteins can interact normally with partner proteins to form functional complexes". Still, I agree with that the activity of DNMT1 needs to be better established, considering the minor increase in DNA methylation levels.

      We will be using ChIP against interacting proteins of DNMT1 and HDAC5 to address this issue.

      The request for a positive control for repeat instability is totally correct.

      We will be adding this in the revised manuscript.

      It is difficult to discuss the missing effect of HDAC5 on contractions or the unexpected effect of HDAC3 on gene silencing bearing in mind the limits of the experimental system.

      There is no expectation for the effect of HDAC5 on contractions as this has not been studied in any system yet. However, we believe that there is no contractions not because of HDAC5 per se but rather because of the shorter repeat size this line has (see comment to reviewer 1 above). We have now addressed the “unexpected effect” of HDAC3 by citing a number of studies finding a similar evolutionary conserved effect (see comment to Reviewer 1 above).

      I also agree with the statement that "this manuscript settled a central question for both HDAC5 and DNMT1 and their involvement in CAG/CTG repeat instability", is not supported by the data.

      We have now rephrased our conclusions. In this particular case, we changed ‘settled’ to ‘addressed’. We have also rephrased this in the results headings.

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      Reply to the reviewers

      Reviewer #1

      Summary

      The authors present well written work on the evolution of proteome size and complexity, and the corresponding changes in chaperone proteins. Interestingly, they find chaperone copy numbers increase linearly with proteome size, despite the increasing 'complexity' of, in particular, post-LECA genomes. They suggest that to address the rise in complexity, organisms express chaperones at higher levels and an expanding network of co-chaperones has evolved across the tree of life.

      Major comments

      Comment-1. Summary reads strangely relative to the rest of the manuscript, and lists facts in a way that makes the purpose of the study confusing. I think most readers will dislike the characterisation of evolution as a progress from simple to complex, and the authors' might want to avoid this language throughout the manuscript- bacteria and archaea have also been evolving over this period of times, and have not become more 'complex'? Similarly the authors should reconsider their figure legend titles. As a specific example, 'in the course of evolution' should become 'across the tree of life'.

      Response

      Thank you for these crucial suggestions. We agree with the reviewer, and with Reviewer 2 (see below) that bacteria and archaea have also been evolving since their emergence, so basically, we (humans) and the simplest archaea have the same evolutionary origin. However, we all agree that the simplest archaea/bacteria are far more similar to LUCA than we are. That said, we accept the criticism that putting our analysis in the context of evolutionary time is an over-interpretation given that we have not examined the protein/proteome phylogeny (in relation to proteome complexity; for chaperones we have). We have thus reformulated the figures and text, to a comparison across the Tree of life, rather than a time-dependent evolutionary process. Specifically: as a first step, we revised the Figures to rename the X-axis as “Order of divergence”, rather than “Divergence time (million years)” in the previous version. In the revised main text we emphasized the fact that the branch lengths of the Tree of Life represent the relative order of divergence of the different clades, rather than time. All instances of ‘in the course of evolution’ has been replaced by ‘across the Tree of Life’.

      Secondly, we revised the main text to emphasize on prokaryote vs. eukaryote comparison, rather than comparing organisms that diverged at different time-points. Within bacterial and archaeal domains, proteomes do not seem to expand against the order of divergence (as the reviewer argued, bacteria and archaea have not become more complex, also see Comment-5).

      Thirdly, the word ‘complexity’ has been omitted from the manuscript. The section “The expansion of proteome complexity” now reads as “Proteome expansion by de novo innovations”. In the previous version, increasing complexity in fact implied a torrent of de novo innovations that impose a larger burden on the chaperone machinery. Instead of ‘complexity’, the latter is clearly stated in the revised manuscript.

      In the spirit of these changes, the title of the revised manuscript, figure legend titles, and related section titles have been edited as follows.

      Submitted version

      Revised version

      Paper title. On the evolution of chaperones and co-chaperones and the exponential expansion of proteome complexity

      On the evolution of chaperones and co-chaperones and the expansion of proteomes across the Tree of Life

      Section title. A Tree of Life analysis of the expansion of proteome complexity and chaperones

      A Tree of Life analysis of the expansion of proteomes and chaperones

      Section title. The expansion of proteome size

      The expansion of proteome size across the Tree of Life

      Section title. The expansion of proteome complexity

      Proteome expansion by de novo innovations

      Figure 1 legend title. Expansion of proteome size

      Expansion of proteome size across the Tree of Life

      Figure 2 legend title. Expansion of proteome complexity

      Expansion of proteomes by de novo innovations

      Further, changes have been made in the Summary and in the main text to exclude any impression that proteomes/organisms have become more complex with time. Rather we emphasized prokaryote versus eukaryote comparison.

      Comment-2. I think the manuscript would be improved if the authors significantly shortened the discussion of genome size evolution- this is fairly well understood, and could be covered briefly, especially as the main focus of the manuscript is on the evolution of chaperone and co-chaperone repertoire. They could also make clearer quantitative links between protein complexity and the evolution of chaperones and co-chaperones- perhaps this should be in the discussion? The authors might also consider referencing 'The evolution of genome complexity', which could be relevant to this manuscript and might make the work of broader interest.

      Response

      We thank the reviewer for this suggestion. The main focus of our paper is indeed the evolution of chaperones and co-chaperones but within the context of the expansion of proteomes. Having this focus in place, the discussion on proteome size evolution (section: The expansion of proteome size across the Tree of Life) has been revised and shortened to emphasize more on prokaryote versus eukaryotic comparison.

      The suggestion to provide “clearer quantitative links between protein complexity and the evolution of chaperones and co-chaperones” is indeed very useful and we authors sincerely thank the reviewer. To address this suggestion we revised Figure 4 to quantitatively compare the expansion of proteomes and that of chaperones, under one roof. This Figure compares proteome parameters that supposedly demands more chaperone action in all three domains of life and simultaneously summarizes the expansion of the chaperone machinery lacking de novo innovations.

      The first paragraph of the Discussion section has been revised accordingly that walks the reader through the revised Figure 4 and finally introduces to the dichotomy it implies.

      We did not understand the last comment “The authors might also consider referencing 'The evolution of genome complexity', which could be relevant to this manuscript and might make the work of broader interest.” We’d be glad to address it upon further clarification.

      Comment-3. The authors state 'protein trees were generated and compared with ToL to account for gene loss and transfer events'. The methodology for this procedure is not given in the manuscript. The authors should back up this point, and make it clear this is why they reconstruct the trees. Currently it is not convincing to me that the authors have found HGT given the considerable phylogenetic uncertainty in the basal events in the tree of life. I also expect the tree of a single protein to be potentially lack information due to the short sequence considered and possible lack of power. The authors need to consider whether the data is really of high enough quality to assess this.

      Response

      Thank you for this suggestion. For the various chaperone families, we manually compared the protein trees with the Tree of Life. This is clearly stated in the revised Methods section (see Page 25, Lines 31-32). We agree, however, that the identifying HGT, and in general, trees of single domains that are highly diverged, are tricky. We did our best to address these caveats. Specifically:

      We re-evaluated our work in the light of a recent study (PMID: 32316034). This paper discussed the phylogenetic uncertainties associated with molecular dating and re-evaluated the assignment of several protein families to LUCA. A careful analysis revealed that the reviewer is indeed right, meaning many of the HGT events shown in the previous version Figure 3B was indistinguishable from the phylogenetic uncertainties.

      Accordingly, we revised the section “The core-chaperones emerged in early-diverging prokaryotes”. We removed the previous version Figure 3B, along with all instances of HGT events mentioned in the main text, except one (archaea to Firmicute HGT of HSP60, which is well-supported by the data and was also detected previously). Dating the emergence of chaperone families was also re-evaluated. Though the major conclusions were not altered, we discussed the phylogenetic uncertainties associated with our work and the overall confidence of each dating analysis. We believe these discussions would be very useful to the readers.

      Finally, we note that most of our key assignments (points of emergence, and major HGT events) are in agreement with previous works. Specifically: the emergence of HSP20 and HSP60 to LUCA (Sousa et al., 2016; Weiss et al., 2016) and HSP60 being horizontally transferred from archaea to Firmicute (Techtmann and Robb, 2010) and HSP20 being horizontally transferred between bacterial clades and between bacteria and archaea (Kriehuber et al., 2010).

      Comment-4. Methods- the authors could consider taking an alternative source of LUCA proteins, rather than those found in 'Nanoarchaeota and Aquificae': it's possible these are not representative of LUCA, and it seems a somewhat arbitrary choice- the authors could consider using one of the available curated sets, such as that generated by Ranea et al. (2006).

      Response

      The reviewer is right that a more robust LUCA set could be used. However, given that the revised manuscript focuses on comparison across the ToL, and foremost on prokaryote versus eukaryote comparison, we don’t think that refining this set is important. Foremost, this set was used for one purpose only, for determining changes in domain length. And, the set of 38 X-groups used for this analysis are in fact, the ones present in all organisms across the ToL. Hence, we kept the original analysis, while mentioning that these 38 X-groups are conserved across the ToL, and removed the argument for LUCA assignment. See Page 5, Line 22.

      Comment-5. The patterns observed might only hold because of differences in the taxa that diverged pre and post LECA? The authors might consider subgroup analyses to ensure this is not the case. The authors could also consider using methods that take phylogeny into account.

      Response

      The reviewer is right that within prokaryotic domains proteomes do not seem to expand. For example, excluding a few early-diverging prokaryotes and parasites, proteome size in bacteria and archaea varies within 2000-3000 proteins per proteome. Only when pre-LECA and post-LECA organisms are compared, significant differences are observed. We thank the reviewer for this suggestion. We revised the main text to focus on prokaryote versus eukaryote comparison. This re-focusing does not change any of our major conclusions, but rather puts our analysis in the right context (see Comment 1).

      Minor comments

      Comment-6. 'Life's habitability has also expanded from its 10 specific niche of emergence-likely deep-sea hydrothermal vents, to highly variable and extreme 11 ranges of temperature, pressure, exposure to high UV-light, dehydration and free oxygen.' This is not really correct, as bacteria and archaea are found worldwide, and in the most extreme environments.

      Response

      Thank you for this suggestion. We removed the above-mentioned sentence.

      Comment-7. 'We reconciled the topology of our tree'- on first read this was not clear, I did not realise the authors were only building trees for subsets of the data- time tree is the best source for the overall topology. The phrase 'manually curated and adjusted' is used in the methods. This language is much too vague, and not a clear explanation of the steps taken.

      Response

      We apology for this confusion. The overall topology of our Tree of Life is indeed taken for TimeTree. We edited the text in Page 4, Line 4 to clarify this issue.

      The obtained tree topology was manually curated and adjusted to depict eukaryotes stemming from Asgard archaea and Alphaproteobacteria, by an endosymbiosis event. This is clearly mentioned in the Methods section (see Page 22, Lines 24-28).





























      Reviewer #2

      Summary

      Rebeaud and colleagues analyze evolution of chaperones compared to the evolution of whole proteome complexity across the entire tree of life. Their principal conclusions are well captured in the following quote from the Discussion:

      "Comparison of the expansion of proteome complexity versus that of core-chaperones presents a dichotomy-a linear expansion of core-chaperones supported an exponential expansion of proteome complexity. We propose that this dichotomy was reconciled by two features that comprise the hallmark of chaperones: the generalist nature of core-chaperones, and their ability to act in a cooperative mode alongside co-chaperones as an integrated network. Indeed, in contrast to core chaperones, there exist a consistent trend of evolutionary expansion of co-chaperones."

      Major comments

      Comment-1. The general theme of the evolution of proteome management is of obvious interest. Unfortunately, the entire analysis is shaky and fails to convincingly ascertain the authors' conclusions. There are many issues. Throughout the manuscript, the authors discuss 'expansion' of the proteome in bacteria, archaea and eukaryotes, creating the impression of a consistent evolutionary trend. No such trend actually exists if one considers the means or medians of proteome sizes within each of the three domains of life (there is a transition to greater complexity in eukaryotes). The maximum complexity, certainly, increases with time which can be attributed to the 'drunkard's walk' effect. This hardly qualifies as 'expansion'.

      Response

      The reviewer is right that within prokaryotes proteomes do not seem to significantly expand. Reviewer-1 raised a similar concern that prokaryotes and eukaryotes have been evolving for the same period of time and have not expanded significantly. We understand the misconception instated by the earlier version and we thank the reviewers for pointing it out. Accordingly, we revised the main text to clarify these issues, as described in the following.

      Firstly, the main text was revised to emphasize on prokaryote versus eukaryote comparison. The reviewer agrees that compared to prokaryotes, “there is a transition to greater complexity in eukaryotes”. This re-focusing does not change any of our major conclusions, but rather provides a systematic comparison that is adequately supported by data.

      Secondly, we revised the Figures to rename the X-axis as “Order of divergence”, rather than “Divergence time (million years)” in the previous version. We emphasized the fact that the X-axis actually represent the relative order of divergence of the different clades, rather than absolute dates. This emphasis certainly does not create the impression of a consistent evolutionary trend. Instead, combined with the revised main text, it depicts that only when pre-LECA and post-LECA organisms are compared, clear trends of proteome expansion is observed.

      Comment-2. The authors further claim a 'linear' expansion of the chaperone set and 'exponential' expansion of the total proteome size. These are precise mathematical terms and, as such, require fitting to the respective functions. No such thing in this manuscript. Even apart from that shortcoming, the explanation of both 'linear' and 'exponential' are quite confusing. Thus, when explaining the 'linearity' of chaperone evolution, the authors refer to the lack of major innovation among the chaperones. This is correct in itself but has nothing to do with linearity. Apart from the aforementioned conceptual problems, the estimation of the 'exponential' growth of the proteome are naive, inconsistent and inaccurate.

      Response

      Our uses of ‘linear expansion’ versus ‘exponential expansion’ may have been confusing although we have defined quite clearly what we mean by that (i.e., that it is not the mathematical sense). The statement regarding “the lack of major innovation among the chaperones” was made in this context/definition and was consistent with it.

      Nonetheless, to avoid confusion, we revised the main text by excluding the ‘linear expansion’ and ‘exponential expansion’ terms. We simply stated that a torrent of de novo innovations has occurred during the expansion of proteomes from prokaryotes to eukaryotes. In contrast, the evolutionary history of core-chaperones lacks such major innovations. Accordingly, the title of the revised manuscript, figure legend titles, and related section titles have been edited as follows.

      Submitted version

      Revised version

      Paper title. On the evolution of chaperones and co-chaperones and the exponential expansion of proteome complexity

      On the evolution of chaperones and co-chaperones and the expansion of proteomes across the Tree of Life

      Section title. A Tree of Life analysis of the expansion of proteome complexity and chaperones

      A Tree of Life analysis of the expansion of proteomes and chaperones

      Section title. The expansion of proteome complexity

      Proteome expansion by de novo innovations

      Figure 1 legend title. Expansion of proteome size

      Expansion of proteome size across the Tree of Life

      Figure 2 legend title. Expansion of proteome complexity

      Expansion of proteomes by de novo innovations

      Comment-3. As the base point for the expansion estimates for archaea and eukaryotes, the authors take parasitic forms. Even leaving aside the highly dubious claims that these organisms belong to the clades that diverged first from the respective ancestors, parasites are not an appropriate choice for such estimates because they certainly are products of reductive evolution. For bacteria, inconsistently, the authors choose a free-living form from a dubious ancient clade, and not even the one with the smallest genome. All taken together, this robs the expansion estimates of any substantial meaning.

      Response

      This point is overall valid. Although we adamantly reject the insinuation of “dubious claims that these organisms belong to the clades that diverged first from the respective ancestors” – firstly, we did not make any claims to this end, but took the ToL constructed by others (Hedges et al., 2015); second, that these claims are dubious need to backup by counter-evidence/data and with all due respect, neither were provided by the reviewer. However, what is of concern is that in a symbiont/parasite chaperones of the host may have a key role, and thus the comparison to free-living organisms could be misleading. To address this concern we excluded the obligatory endosymbiont Nanoarchaeum equitans and the parasitic organisms from the expansion estimates and such discussions are now limited to free-living organisms only. Further, as described in response to Comment-1, the revised manuscript focuses on prokaryote versus eukaryote comparison.

      Note that phylogenetic analysis often assigns parasitic and symbiotic organisms that have experienced reductive evolution as the earliest diverging clades of their corresponding kingdoms of life. Examples include Nanoarchaeum equitans, an obligate symbiont, assigned as the earliest diverging archaea (Hedges et al., 2015; Huber et al., 2002; Waters et al., 2003), and parasitic Excavate assigned as one of the earliest diverging eukaryotes (Burki et al., 2020; Simpson et al., 2002). In accordance with these studies, these parasitic and symbiotic organisms were included in our analysis. We acknowledged this fact in the Methods section (see Page 22, Lines 9-16).

      Comment-4. The authors do make a salient and I think essentially correct observation: chaperones typically comprise about 0.3% of the proteins in any organism. As such, this presents no dichotomy in evolutionary trends to be explained. Surely, as examined and discussed in the paper, eukaryotes also show significant increases in the size and domain content of the encoded proteins, suggesting the possibility that might need more chaperones. However, if this is the explanandum, rather than the number of proteins in the proteome as such, it should be clearly stated. Furthermore, it is quite natural to assume that this increase in protein complexity without a commensurate increase in the chaperone diversity, is enabled by higher expression of the chaperones as suggested in the Discussion of this paper. I doubt there is any big surprise here and even much need for an extended discussion let alone a special publication.

      Response

      As emphasized, and shown, eukaryotes have not only larger proteomes in terms of the number of proteins or protein size. They have a higher content of proteins that are prone to misfolding. This is shown explicitly, in Figure 2 (namely, multidomain proteins, repeat, beta-rich proteins, etc’) and is reiterated in a summary figure (suggested by Reviewer 1). Further, in response to Reviewer-3’s suggestion, we showed that eukaryotes feature much higher proportions of aggregation-prone proteins per proteome than prokaryotes (Figure 2E).

      To further clarify, we revised Figure 4 to quantitatively compare the expansion of proteomes and that of chaperones, under one roof. This Figure compares proteome parameters that supposedly demands more chaperone action in all three domains of life and simultaneously summarizes the expansion of the chaperone machinery lacking de novo innovations.

      In addition, the first paragraph of this Discussions section is revised to state that from prokaryotes to eukaryotes, proteomes have expanded by duplication-divergence as well as by innovations (de novo emergence of new folds). Thus, it’s not about the size only (a challenge that a proportion expansion of chaperone genes would resolve, i.e., the 0.3%) but about proteome composition changing in a way that demands more and more chaperone action.

      We also agree with the assertion that “it is quite natural to assume that this increase in protein complexity without a commensurate increase in the chaperone diversity, is enabled by higher expression of the chaperones”. However, we belong to a group of scientists for whom natural assumptions are insufficient, and think that supporting evidence is of importance.

      Reviewer’s significance statement

      As such, in the opinion of this reviewer, there is no substantial advance over the existing knowledge in this paper. Should the authors wish to revise, they would need to develop robust methodology to measure proteome expansion. That would involve starting from reconstructed ancestors rather than any extant forms (let alone parasites). I doubt that such analysis, non-trivial in itself, reveals an strong, consistent trends other than the well known increase in complexity in eukaryotes.

      Response

      We agree that to assert evolutionary, time-dependent trends one needs to analyze phylogenies and reconstructed ancestors, but still think that a comparison of proteome and chaperone contents along the Tree of Life is meaningful. We thus respectfully, yet adamantly disagree with “no substantial advance over the existing knowledge”. We strongly believe, as does Reviewer-3, that the results and the model presented in this paper are “fascinating to consider and… will stimulate a good deal of important discussion…”.

      Reviewer #3

      Summary

      The manuscript by Rebeaud et al describes phylogenetic analyses of proteome and chaperone complexity. The authors analyzed species across the tree of life to predict the proteome and chaperone properties of ancestors spanning to the last universal common ancestor. Their analyses indicate that many proteome properties increased in complexity over evolutionary time including: average protein length, the number of multi-domain proteins, the size of the proteome, the number of repeat proteins, and the number of beta-superfold proteins that are known to be difficult to fold. Their analyses also indicate an expansion in chaperone families that corresponds to the increase in proteome complexity. Based on their analyses, the authors propose a model where early life relied on a limited number of chaperones (Hsp20 and Hsp60) and that as proteome complexity evolved, so did chaperone complexity. Core chaperones including Hsp90, Hsp70, and Hsp100 evolved relatively early, and later chaperone evolution was driven by the appearance and alterations of co-chaperones and auxiliary factors as well as by increases in the protein abundance of chaperones.

      Major concerns

      Comment-1. This work is appropriately based on phylogenetic inferences, but as such, the limitations and uncertainties of phylogenetic inferences need to be discussed. This in no way takes away from the work, quite the opposite, it would make it richer by encouraging broader interpretations where justified and clear understanding of where support for the model is strongest. Posterior probabilities need to be discussed and the range of properties that a likely ancestor might have based on the data should be discussed. How this impacts the conclusions and models should be discussed. Throughout the manuscript, the authors present most-likely ancestral models (as I understood it), what are the next most likely models? How much power is there to distinguish one model from another? It would be very helpful to have a section describing the limitations and uncertainties of the phylogenetic analyses and how these relate to the main findings and conclusions.

      Response

      We thank the reviewer for this suggestion. Reviewer-1 raised a similar suggestion (see Comment-3). The phylogenetic analysis in our paper included dating the emergence of core- and co-chaperone families, and attempt to infer major their HGT events, foremost in relation to the origin of eukaryotic chaperones. To highlight the uncertainties of phylogenetic inferences we re-evaluated our work in the light of a recent study (PMID: 32316034) that carefully analyzed the uncertainties associated with the assignment of several protein families to LUCA.

      Ideally, for a protein family to be assigned to LUCA, there must be a single split of bacterial and archaeal domains at the root of the protein tree with strong bootstrap support, and the inter-domain branches would be longer than the intra-domain branches (PMID: 32316034). In the revised main text we discussed that only the HSP60 protein tree satisfies this criterion. HSP20 protein tree depicts a clear single split of bacterial and archaeal domains at the root, albeit with weak bootstrap support, and inter-domain branch lengths are smaller than intra-domain branch-lengths. We discussed that this is indeed the case of phylogenetic uncertainty, which means the sequence of this small, single-domain chaperone lacks the information to make reliable inference at the basal events in the ToL.

      In addition, the HGT events discussed in the previous version appear to be indistinguishable from phylogenetic uncertainties and we removed all instances of HGT events mentioned in the main text as well as Figure 3B. Only one HGT event – HSP60 being horizontally transferred from archaea to Firmicute, which is well-supported by the data is kept in the revised main text. We believe these discussions would be very useful to the readers.

      Finally, we note that most of our key assignments (points of emergence, and major HGT events) are in agreement with previous works. Specifically: the emergence of HSP20 and HSP60 to LUCA (Sousa et al., 2016; Weiss et al., 2016) and HSP60 being horizontally transferred from archaea to Firmicute (Techtmann and Robb, 2010) and HSP20 being horizontally transferred between bacterial clades and between bacteria and archaea (Kriehuber et al., 2010).

      Comment-2. General features that impact foldability, including contact order, should be discussed and what features can be searched for in genomes that relate to these - e.g. beta-rich proteins.

      Response

      Thanks for this valuable idea! Contact order, and other predictors of problematic folding are highly relevant but their analysis is structure-based and hence inapplicable on the proteome (sequence) scale. We did, hwoever, estimate the proportion of aggregation-prone proteins in the proteome. These proteins were identified by CamSol method that assigns poorly soluble regions from sequence data. Indeed, some of these predicted ‘poorly soluble segments’ refer to the hydrophobic core of the respective folded state instead of ‘true’ aggregation hotspots. With this unavoidable potential caveat, it appears that compared to prokaryotes, aggregation-prone proteins in the proteome have become nearly 6-fold more frequent in Chordates.

      Following changes were made to accommodate this new analysis:

      Figure 2 is revised to include a new panel (panel-E) that shows the expansion of aggregation-prone proteins in the proteome across the Tree of Life. The same result is summarized in the summary Figure 4.

      A new paragraph entitled “Proteins predicted as aggregation-prone became ~6-fold more frequent in the proteome” is added to the Results section, which describes the principle and the main results (see Page 7, Lines 14-28).

      The methodology is included in the Methods section, in a paragraph entitled “Predicted proportion of aggregation-prone proteins in the proteome”, see Page 24 Lines 17-27. For each representative organism, the percent of aggregation-prone proteins in proteome data are provided as Data S10.

      This analysis is also included in the revised Abstract: “Proteins prone to misfolding and aggregation, such as repeat and beta-rich proteins, proliferated ~600-fold, and accordingly, proteins predicted as aggregation-prone became 6-fold more frequent in mammalian compared to bacterial proteomes.” See Page 2, Lines 7-9.

      Comment-3. "Core" chaperones needs to be defined.

      Response

      Thank you for this suggestion. We restructured Page 3 Lines 19-23 in the Introduction to clearly explain this aspect. The current text is quoted below.

      “Chaperones can be broadly divided into core- and co-chaperones. Core-chaperones can function on their own, and include ATPases HSP60, HSP70, HSP100, and HSP90 and the ATP-independent HSP20. The basal protein holding, unfolding, and refolding activities of the core-chaperones are facilitated and modulated by a range of co-chaperones such as J-domain proteins (Caplan, 2003; Duncan et al., 2015; Schopf et al., 2017).”

      Minor concerns and thoughts

      Comment-4. This manuscript stimulated me to think about the dynamics between chaperone evolution and proteome evolution. The ability to tolerate proteins that need chaperones seems linked to major evolutionary innovations. Once you have these innovations though, you are addicted to the chaperones - and an expansion of the number of sub-optimal proteins. These ideas seem like they would be valuable to include in the discussion of this work. More generally, it would be wonderful to have a discussion of future directions that this work may spark.

      Response

      This is indeed a fascinating question or set of questions, that we have also become intrigued about following this work, We introduced a short section, though more of an ‘appetizer’ than a detailed discussion, as we know almost nothing about the co-evolution of new proteins and chaperones.

      Reviewer’s significance statement

      This manuscript provides a fascinating glimpse back in time of a fundamental interplay - between chaperone evolution/addiction and proteome evolution. I am not an expert in phylogenetic analyses so I cannot judge the details of the analyses. As an expert in molecular evolution and chaperones, I found the approach and model fascinating to consider and I believe it will stimulate a good deal of important discussion in these fields. I have one major concern that I feel ought to be addressed in the manuscript and a number of points that I would encourage the authors to consider. I am sure that these can be readily addressed and I look forward to seeing this work published and the further discussion and ideas that it may stimulate.

      Response

      Thank you!

    1. “moral education”

      <br>

      Analytic notes:

      The primacy of moral education in the design of the French primary school system is evidenced in the writings of Francois Guizot, the French Minister of Education who during the July Monarchy drafted the 1833 Loi that established, for the first time, a centrally-controlled system of primary schools, with a centralized curriculum and primary school teachers trained and recruited according to central government directives. The centrality of moral education as a motivation to centralize the control and expand the provision of primary schooling in France is also discussed by Weber (1976). The 1833 Loi is especially important because it was followed by the fastest expansion of primary schooling observed in French history (Grew and Harrigan 1991).

      That the Prussian primary school system focused mainly on moral and religious education was perceived to be true among those who admired and those who criticized Prussian schools. Among the admirers was Horace Mann, a U.S. education reformer who played a key role in the development of a system of common schools in Massachusetts during the 1830s and 40s. Mann devoted two of the twelve Annual Reports he wrote as Secretary of the Massachusetts Board of Education entirely to the importance of moral education. He highlighted Prussia as an example of an education system that shapes moral values. The Prussian emphasis on moral and religious education noticed by Mann continued to prevail at the beginning of the 20th century according to Thomas Alexander, a professor at the George Peabody College of Teachers who spent between 1912 and 1913 studying Prussian schools directly as the basis for his book The Prussian Elementary Schools, published in 1919.

      Source excerpts:

      In an essay discussing the perceived benefits of the 1833 Loi, Guizot (1860) writes: “The state must provide primary education to all families and give it to those who cannot afford it; and in this he does more for the moral life of peoples than he can do for their material condition. This is the true principle on this point, and it was the one adopted by my bill.” (p. 63-64) “Primary education is not a panacea which cures all the moral maladies of the people . . . ; it is a salutary or harmful power according to whether it is well or badly directed and contained within its limits or pushed out of its mission. . . . The law of June 28, 1833, has received various modifications . . .; but all its principles, all its essential dispositions, have remained upright and in force. Founded by this law, primary education is now, among us, a public institution and an acquired fact. There remains, surely, much to be done for the good government of the schools, to make it dominant in their bosom the influences of religion and order, of faith and law, which make both the dignity and the safety of a people.” (p.86)

      And continues: “If it is true that the attachment of the citizens to the fundamental laws of the state and to the sovereign who is its leader, is the most energetic power and surest safeguard of society, if it is true that there where this feeling has existed, it has produced miracles, and that its absence has brought about the greatest evils, it is the duty as well as the interest of the government to favor and direct its development. But this feeling can only arise from the concordance of public doctrines and national habits with political institutions, the nature and principles of government. . . . When men have learned from childhood to understand the fundamental laws of the country and to respect its sovereign, the sovereign and the laws become a kind of property which is dear to them, and they do not refuse the obligations that it imposes upon them: when the government has taken care to propagate, in favor of national education, in the relations of religion, morality, politics, etc., the doctrines which suit his nature and his direction, these doctrines soon acquire a power against which come the failures of liberty of mind and all seditious attempts. Thus the public mind is formed, thus a true patriotism is maintained, thus fortifying and consolidating societies and thrones.” (Guizot 1860, 86).

      Weber (1976, 329): “Official reports coupled poor education with rude, brutal ways. Where schooling did not take hold, ‘ways are coarse, characters are violent, excitable, and hotheaded, troubles and brawls are frequent.’ The school was supposed to improve manners and customs, and soothe the savage breast. The polite forms it inculcated ‘softened the savagery and harshness natural of peasants.’ Improved behavior and morality would be attributed to the effects of schooling.”

      Mann (1844, 22): “If Prussia can pervert the benign influence of education to the support of arbitrary power, we surely can employ them for the support and perpetuation of republican institutions… If a moral power over the understandings and affections of the people may be turned to evil, may it not also be employed for good?”

      Alexander (1919, v): “A careful study of the Prussian school system will convince any unbiased reader that the Prussian citizen cannot be free to do and act for himself; that the Prussian is to a large measure enslaved through the medium of his school; that his learning, instead of making him his own master, forges the chain by which he is held in servitude; that the whole scheme of Prussian elementary education is shaped with the express purpose of making ninety-five out of every hundred citizens subservient to the ruling house and to the state.”

      In a later chapter that focuses on the curriculum and teaching methods, Alexander refers to the influential ideas of Johann Friedrich Herbart, a German philosopher, psychologist and educator, author of The Moral or Ethical Revelation of the World: The Chief Aim of Education (1804), and Prussian commissioner of education. Alexander (1919, 258) writes that “With Herbart and his followers, the aim and end of instruction was the formation of moral and religious character. It was not Herbart’s intention merely to furnish the child with a definite number of concepts by the form and content of the instruction and subject matter, but rather to shape thereby the will and directly to affect the moral nature and attitude of the child.”

      In the conclusion, Alexander (1919, 540) writes: “Chief among the avowed aims of the Volksschule is the formation of moral and religious character. There is no doubt that the reason religion is made one of the chief subjects of instruction in the elementary schools is to teach the lesson of obedience to authority which is the basis of the German state.” And continues, “There are then leaders and followers. The leaders think and do; the followers merely do. The schools are organized for the express purpose of producing just these types . . . They educate the individual for the state; we [Americans] make the state for the individual. The lesson to learn here is this. The German sets definitely his national aims. Those in authority shape every resource to reach that goal. The schools were molded to meet the needs of the state.” (Alexander 1919, 538-39).

      Full citations:

      Alexander, Thomas. 1919. The Prussian elementary schools. New York: The Macmillan company.

      Grew, Raymond, and Patrick J. Harrigan. 1991. School, state, and society: the growth of elementary schooling in nineteenth-century France: a quantitative analysis. Ann Arbor: University of Michigan Press.

      Guizot, Francois. 1860. Memoires pour servir a l'histoire de mon temps, T.3. Paris: Michel Levy freres.

      Mann, Horace. 1844. Seventh Annual Report on the Board of Education. Boston.

      Weber, Eugen. 1976. Peasants into Frenchmen: The Modernization of Rural France, 1870-1914. Stanford, California: Stanford University Press.

    1. Reviewer #2:

      The paper compares musicians' behavior and ERP responses to those of non-musicians with the following statement in the abstract:

      "these better performances could be due to an improved ability to process sensory information, as opposed to an improved ability to learn sequence statistics. Unfortunately, these very different explanations make similar predictions on the performances averaged over multiple trials. To solve this controversy, we developed a Bayesian model and recorded electroencephalography (EEG) to study trial-by-trial responses."

      The authors claim:

      "This higher performance is explained in the Bayesian model by parameters governing SL, as opposed to parameters governing sensory information processing. " This is correct - but meaningless - the experiment does not challenge sensory noise since the 3 sounds used are so distinct that sensory noise is zero in the two groups. Given that basic design - this phrasing is not only too strong, it is in proper.

      My understanding is that are two actual observations in the paper:

      1) Musicians' learning of second order markov statistics is better than that of non-musicians based on parameter fitting of a Bayesian model of their behavior in answering explicit questions regarding which sound (of 3 very distinct options) should come next.

      2) ERP measures - specifically P300 of musicians, is more sensitive to this statistics as evident by its magnitude with respect to predictability/surprise of the sound based on serial statistics. These claims are interesting BUT - I am not convinced by the claim of specificity. I think the data (and previous studies) suggest that musicians do better with sound related judgments - with all respects.

      I am not convinced that the model adds information since it explains the data as a good as single accuracy numbers (or did I miss something?). So I am not convinced that this trial by trial analysis adds information.

      With respect to the specific model parameters:

      Sensory noise is zero - the sounds are quite distinct. This is not an observation - this is how the experiment was designed. The authors admit that (indeed - any study that focused on sensory discrimination found an advantage in musicians) - but then state specificity, particularly in the abstract.

      Regarding rate of decay - I wonder if this is relevant to overall performance when asked only up to 2nd order serial statistics. It may be sufficient for the task. The relevance of this parameter should be clarified.

      Thus the lack of group difference in these parameters probably tells about the experiment rather than the groups.

      Similarly, musicians' ERP responses are larger. But the early difference is not addressed at all. Is the earlier response sensitive to simpler stat - but in a similar way in both populations? Can't be - since they have a different magnitude. The authors base their analysis on (MEG analysis) in their 2019 paper. I tried to do the exact comparison, and wasn't sure about the mapping to components - please clarify the exact similarity.

      Thus - overall - I am not sure that the model analysis provides new conceptual insights.

    2. Preprint Review

      This preprint was reviewed using eLife’s Preprint Review service, which provides public peer reviews of manuscripts posted on bioRxiv for the benefit of the authors, readers, potential readers, and others interested in our assessment of the work. This review applies only to version 1 of the manuscript.

      Summary:

      This work constitutes an innovative and timely combination of modelling, behaviour and EEG to understand potential differences in SL abilities between musicians and non-musicians. However, as detailed below, we have many concerns regarding the modelling, experimental design and interpretation of the results.

      Our major concerns are summarized here (and further elaborated in the individual reviews below):

      1) Modelling: please report the accuracy of the model and whether this differs between groups.

      2) You should analyse the interaction in Fig. 1B and report whether or not it is significant.

      3) Relatedly, there appears to be an inconsistency between the behavioural results and the modelling. In the behavioural data you report a main effects of musicianship and of sequence complexity. Modelling of this data suggests that whilst the K for musicians is higher than non musicians it is substantially above 1 for both. If anything this should predict larger differences between groups in larger K than smaller K which is different from what is seen behaviourally. A similar inconsistency is present between the behavioural results and the results in figure 4 (see below). This requires careful consideration.

      4) Can you do more to convince the reader that the model is performing well? Is the fit good, how does it vary across participants? Does rate of memory decay affect performance at all? Can you show good versus poor performers within the same group - do parameters also vary there?

      5) It is important that you address the issues related to participants being aware of the stimulus construction. Are there any consequences of the fact that participants were aware of the probabilistic nature of the sequences and the differences between the two sequence types? This seems to be an important divergence from other SL studies which could impact on the behavioural and neural effects observed and should, therefore, be discussed.

      6) The EEG data are recorded whilst participants are performing the behavioural prediction task. Though probe trials occurred rarely, it is conceivable that participants were making an active judgement for each sequence item. There is therefore a concern that the measured EEG data would reflect this aspect (active task performance) rather than automatic SL. This makes conclusions about "neural statistical learning" (e.g. as in the title) difficult to make.

      7) In the results section the authors consider various differences between the musician and non-musician groups that could lead to differences in performance. One aspect that does not seem to be considered is that of attention, or task engagement. Is it possible that the musician participants were simply more engaged/less bored by the task? The EEG data (figure 3) are consistent with this interpretation showing overall substantially larger responses in the musicians relative to the non musicians.

      8) In general, we think the model has been constructed with due care and attention and we like the separation of parameters related to statistical learning (model order and selection noise) and more general aspects of perception and cognition (sensory noise and memory decay). We think the difficulties arise in the relationship between the model and the experiment. Specifically, the sensory noise model parameter reveals very little in the analysis of this data because the sounds were so readily distinguishable, which appears to have been a deliberate choice in the experimental design, somewhat confusingly. The present stimulus set is therefore not suitable for distinguishing differences in sensory processing vs. SL between groups. We suggest that the authors could simply remove this parameter from the analysis and the paper would be clearer as a result. This would involve re-modelling and you will also have to reshape the way the experiment is motivated.

      9) We have some questions about how the EEG data are analysed. In particular, the large amplitude difference between groups should be quantified, discussed and interpreted. We would also like to see stronger justification and discussion of why these differences are not affecting the main conclusions. We note that the authors provide R2 results in supp materials but we feel that a better approach may involve normalizing the responses before modelling. Higher SNR in the musician group may lead to stronger correlations. One way around this is to normalize the data (e.g. based on the P1 response) before computing the correlations.

      10) You should perform the appropriate statistical analysis to support the claims associated with Figure 4. You seek to conclude that the two groups have similar responses to surprise in simple statistical contexts (K=0) with divergence occurring for more complex statistical structure. However, you do not provide statistics to support this claim. It is not enough to show no significant difference between groups for K=0, but significant differences for K=1, 2. You need to demonstrate an interaction between group and model order. Additionally, it was also not quite clear how modelling was performed here. We understand that you take surprise values from the model fitted to each participant but with the order fixed at 0, 1 or 2. This may mean that the other parameters might no longer be optimal in the context of the new fixed K values, depending on how different these were from the fitted values for each participant, which might plausibly differ for the musicians and non-musicians. To address this, Can you supplement the existing analysis with an analysis in which the K parameters are fixed at 0, 1 and 2, and the other parameters are re-optimised in the context of these fixed parameter values. Please also provide information about how well each individual data were fit, and whether there was a significant difference between musicians and non musicians. In general, we think the authors should present the result in figure 4 more cautiously and also flesh out the interpretation in more detail in relation to the literature along with a consideration of other potential interpretations. A small related point is that the term hierarchy is strongly related to this interpretation and we would prefer a more neutral term such as 'model order'.

      11) The paper would benefit from a careful discussion of exactly what information, on top of that revealed with behaviour, is added by EEG and the significance of this in the context of the existing literature on expectation related ERP components.

    1. cultural forms that continue to resonate powerfully as part of what Raymond Williams calls a "selective tradition" (115).14

      Williams says that the selective tradition chooses cultural work-art/literature- to highlight and obscure based on the dominant ideology. Work that maintains the dominant ideology-capitalism- is generally highlighted and canonized, while work that is truly subversive is hidden and obscured. This is a somewhat unconscious process. So when we think about why we study Marlowe, and why he has been canonized we have to consider that the reason the culture keeps selecting him may be because he maintains the dominant ideological structure. This reminds me of Bartel and all of those critics from our first readings claiming that Marlowe is truly subversive. While he may have been for his own time, perhaps he isn't for ours and instead upholds our current culture of capitalist globalization.

    1. generate 20 to 30 assumptions, true or false, that you may be making about it. Then pick several of these assumptions and use them as thought starters and idea triggers to generate new ideas.

      I find this useful because a lot of times we make assumptions that may not be true--yet we think of them as fixed. that's what an assumption is. So to find new ideas, you gotta question what you think is "fixed" Always go to the consumer, for example, don't assume anything about the consumer.

    1. Do patients and surrogates have the moral right to insist on life-sustaining treatment even after permanent loss of consciousness? I tend to think not, even though stopping treatment would offend many people on religious or moral grounds. (Note that we already permit physicians to declare death by whole-brain criteria even when family members object.) But in order to limit distress to patients and surrogates who won’t accept the higher-brain criteria of personhood, it may be more compassionate not to mandate withdrawal of feeding tubes as a strict policy, but rather to educate people about the nature of PVS and the futility of treatment.

      This is a really interesting topic and raises some further questions for me, such as the role of insurance and how realistically possible it is for someone to afford this?

    Annotators

    1. Booker T Washington's gradualistic perspective on how newly freed slaves were to adapt to life seems to be appropriate for the times considering how turbulent they were, but it also seems to enable and continue the story and the image of Blacks as less than or average compared to their fellow citizens. I can see why he may have believed in this approach to integration of former slaves into society, but I think it may have set a dangerous precedent also in delaying the atonement and reckoning of the stain of slavery on our country's history by minimizing the fact that we enslaved humans. Frankly, I think its dehumanizing.

    2. Our greatest danger is that in the great leap from slavery to freedom we may overlook the fact that the masses of us are to live by the productions of our hands, and fail to keep in mind that we shall prosper in proportion as we learn to dignify and glorify common labour and put brains and skill into the common occupations of life; shall prosper in proportion as we learn to draw the line between the superficial and the substantial, the ornamental gewgaws [sic] of life and the useful.

      Washington and DuBois were newly freed slaves who took upon this burden of carrying and trying to mentally free other African Americans. Unfortunately, these two men were only viewed as property. Though these two leaders had different proposals as to how African Americans could escape their current situation. The two men were not going to give up until their black community "made it out". During Booker T. Washington's childhood he experienced slavery first hand, this is when he began to realize the importance of education. He later attended Hampton Institute where he soon felt the effects of segregation in the educational system between black and whites. This experience resulted in him becoming an educator himself. He then advocated that African American stood up and fought for change, which included their rights to vote in order to gain some type of security for themselves and the generations to come. W.E.B. DuBois had a completely different upbringing as he was free and not have to experience the unforeseen circumstances of those in the southern states. DuBois later became the first African American to receive his doctorate from Harvard University. The biggest disagree between these two leaders is that DuBois did not think that uneducated blacks needed to vote, which I think is outrageous.That is honestly the largest thing that bothered me throughout this learning. Even though there were plenty of differences there was one thing that they agreed on which was violence against blacks including lynching. This was a great read and refresher about these two phenomenal African American leaders.

    3. Our greatest danger is that in the great leap from slavery to freedom we may overlook the fact that the masses of us are to live by the productions of our hands, and fail to keep in mind that we shall prosper in proportion as we learn to dignify and glorify common labour and put brains and skill into the common occupations of life

      I really like this part of Mr. Washington's speech. I think it speaks volumes by drawing attention to the fact that yes, many's history was rooted in slavery, yet the skills that they possessed as a result are valuable and should be exalted instead of looked down upon. Hard labor is honest, needed, and valued work but during this time it was exceptional to have a dignified leader bring attention to it, and it's importance.

    1. Just what is relevant and what is not is a matter of judgment of the system-builder, but we think of such things as states of war or peace, degrees of hostility or friendliness, alliance or enmity, arms bud­gets, geographic extent, friendly or hostile communications, and so on

      So some people see a scenario as relevant/irrelevant, but for others it might be a matter of life and death. For example, 2001 Afghan invasion by America may be seen as relevant by America's strategic policy-makers but it cost thousands of innocent civilian life.

    1. Here the direct application of an automatic approach to the act makes it clear that not only is this not the old craft of painting, but it is perhaps bordering on ritual itself, which happens to use paint as one of its materials. (The European Surrealists may have used automatism as an ingredient, but we can hardly say they really practiced it wholeheartedly. In fact, only the writers among them—and only in a few instances—enjoyed any success in this way. In retrospect, most of the Surrealist painters appear to have derived from a psychology book or from each other: the empty vistas, the basic naturalism, the sexual fantasies, the bleak surfaces so characteristic of this period have impressed most American artists as a collection of unconvincing cliches. Hardly automatic, at that. And, more than the others associated with the Surrealists, such real talents as Picasso, K.lee, and Miro belong to the stricter discipline of Cubism; perhaps this is why their work appears to us, paradoxically, more free. Surrealism attracted Pollock as an attitude rather than as a collection of artistic examples.)

      When I think of surreal artwork I think of visions, not only dreams, but maybe a vision we have while looking at the clouds. We envision a transformation and it feels like a message from our unconscious. Or a guided meditation, I've worked with Shamans and done meditation with spiritual gurus and masters, and the visions you can have while practicing these I guess you could say ritualistic practices are profound. After they happen you wonder what they meant, the vision or the video type imagery comes to you and you just observe it, and then after when you come back to yourself you wonder what it meant and attach meaning to it. I find my own surreal art ideas in these practices. I really relate to what Kaprow is saying here, how Pollock's work was an attitude, and a ritual, not just a abstraction to be interpreted. Do you think Pollocks ritualistic practice of drip painting came from his unconscious or a higher source, and that unknowingly Pollack's work was was made with intention from this source?

    1. Nancy Lee sometimes forgot she was colored herself. She liked her classmates and her school.

      As a reader, these two sentences caught me off guard. This is in large part because of the time period this story takes place. Racism and segregation are very, heavily present, and many people may think this part is small and simple; however, to say that Nancy forgot she was colored, gives the audience a huge understanding on good she had it. Compared to other African-Americans during this era, to like being at school was huge, but most colored students couldn't go to the same school as whites, so that alone sets this story into a new perspective for me. Overall, Nancy was a fortunate student in a time, such as we are in now: uncertainty.

    1. Thus, Confucius meditated upon water; and the Confucian Xunzi later attempted to systematize the relationship between water’s various forms and people’s moral qualities. This assumption of a correspondence between the principles which inform both water and human conduct was not limited to the Confucians; it was generally assumed in all early philosophical texts. Nor was the imagery the provenance of any particular school. For example, water which moves forward without force, giving life to everything, is described in Xunzi as ‘wuwei’ (without action) or (doing nothing) a term that is particularly associated with Daoism.

      CONTEXT: Shuen-fu Lin addresses "the sage", the person with the highest spiritual attainment who was first emulated and thought of in the Wei-Jin movement, following the Han Dynasty. The sage allows the innate tendencies and has all five of the human emotions addressed in the passage, but "...does not act, complies, and does not implement. He eliminates what leads things astray and gets rid of what confuses them." The sage is addressed as exhibiting qualities of both the Daoist way of life and the Confucianist way. The sage is like the image of water that is an unattainable, sage-like, presence and moral conduct, desired by both Daoist and Confucianist beliefs. "Gentlemen" look at water in awe, gazing upon the perfection of its inaction and lack of effort in attaining its intellect, beauty, and respect. The water has of "ziran", or perhaps, is "ziran" that humans are able to express communion with nature and nonpurposive action. This word is also described as spontaneously existing and being "so oneself" -nothing acting behind them. Water does not decide or dwell for too long, it just exists in movement and in detachment which I think human beings desire greatly.

      Cai, Zongqi. Chinese Aesthetics: The Ordering of Literature, the Arts, and the Universe in the Six Dynasties. Honolulu: University of Hawaii Press, 2004.

      RELATE: In 'The Experience of Nature' by Rachel and Stephen Kaplan, human action and thought is addressed as influenced by our setting/environment whether the setting require immediate responsive action or the response take place in a slower, observational method varies. The authors write, "People are particularly aware of information that is visual, that concerns what they see. That does not mean that people interpret the information in visual terms exclusively; rather, visual stimuli are effective in conjuring associated information. The sight of water provides information about potential opportunities which may or may not be visual in themselves" (Kaplan, 4). Reverie from observation that allows self reflection, thought free from distraction, and intuitive action is typically included in our broader categorization of landscape qualities when we discuss as landscape architects. Human reaction to landscape is so much bigger than the texture, color, or even kinesthetic feeling within the place and can be thought of as artwork in addition- prompting development of thought even subconsciously within the the one experiencing.

      Kaplan, Rachel, and Stephen Kaplan. The Experience of Nature: A Psychological Perspective. Cambridge: Cambridge University Press, 1989.

    1. For if a person is imagined lying back with outstretch arms and feet within a circle whose center is at the navel, the fingers and toes will trace the circumference of this circle as they move about. But to whatever extent a circular scheme may be present in the body, a square design may also be discerned there. For if we measure from the soles of the feet to the crown of the head, and this measurement is compared with that of the outstretched hands, one discovers that this breadth equals the height, just as in areas which have been squared off by use of the set square.

      This particular section is interesting because shape is something we often see as being separate to the human body, but Vitruvius is encouraging his audience to see shape as something which in fact the human body operates within. Our limbs can fit within squares and circles and I think Vitruvius must have been fascinated by the way the body had a composition in the same that architecture did. I know others above have commented on the fact that Vitruvius does not seem to take account of the fact that the human body can have many imperfections, and this is a really important point to take further.

    1. Thus, we argue that enhancingpeople’s happiness levels may indeed be a wor-thy scientific goal, especially after their basicphysical and security needs are met.

      I wonder is there any research on whether happiness is achievable if some of your basic needs aren't met? If not, does that mean individuals who may be homeless, in poverty or alone aren't able to achieve happiness? What about people who live in less modernized places who don't view what we view as necessities? Makes me think that the way we separate our needs from our wants is completely subjective.

    1. Do you think the world is going to be a better place next year? In the next decade? Can we end hunger, achieve gender equality, halt climate change, all in the next 15 years?

      I personally believe the wild will be a better place in the future though not any time soon. In todays present situation we as a society are still struggling with a number of issues. We are still facing pollution that is killing our aquatic life, social and racial issues, and even a pandemic! The governments world may say we can, but they need to take the right actions to insure so. Hopefully in the upcoming years we can dissolve a number of these problems thus making thew world a better place for future generations.

    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)): **Summary:** This interesting study by Putker et al. showed that circadian rhythmicity persists in several typical circadian assay systems lacking Cry, including Cry knockout mouse behavior and gene expression in Cry knockout fibroblasts. They further demonstrated weak but significant circadian rhythmicity in Cry- and Per- knockout cells. Cry- (and potentially Per-)-independent oscillations are temperature compensated, and CKId/e still has a role in the period regulation of Cry-independent oscillations. **Major comments:** 1) The authors propose that the essential role of mammalian Cryptochrome is to bring the robust oscillation. As the authors analyze in many parts, the robustness of oscillation can be validated by the (relative) amplitude and phase/period variation, both of which should be affected significantly by the method for cell synchronization. Unfortunately, the method for synchronization is not adequately written in this version of supplementary information. This reviewer has no objection to the "iterative refinement of the synchronization protocol" but at least the correspondence between which methods were used in which experiments needs to be clearly explained. The detailed method may be found in the thesis of Dr. Wong, but the methods used in this manuscript need to be detailed within this manuscript.

      We thank the reviewer for recognising the importance of different synchronisation protocols. In experiments where bioluminescent CKO rhythms were observed, different synchronisation protocols resulted in similar results when comparing WT with CKO cells. The different synchronisation methods used in each experiment are now specified in the supplementary methods.

      2) The authors revealed that CKO mice have apparent behavioral rhythmicity under the condition of LL>DD. This is an intriguing finding. However, it should be carefully evaluated whether this rhythmicity (16 hr cycle) is the direct consequence of circadian rhythmicity observed in CKO and CPKO cells (24 hr cycle) because the period length is much different. Is it possible to induce the 16 hr periodicity in CKO mice behavior by 16 hr-L:16 hr-D cycle? Would it be a plausible another possibility that the 16 hr rhythmicity is the mice version of internal desynchronization or another type of methamphetamine-induced-oscillation/food-entrainable-oscillattion?

      The reviewer makes an excellent suggestion. As described in the manuscript text (page 13), CKO mice have already been shown to entrain to restricted feeding cycles (Iijima et al., 2005) and we therefore assessed whether CKO rhythms would entrain to a 16h day as suggested. Whilst CKO (but not WT) mice showed 16h behavioural rhythms during entrainment, they were arrhythmic under constant darkness thereafter (Revised Figure S2A). CKO cellular rhythms show reduced robustness under constant conditions ex vivo, and our other work has revealed that CRY-deficiency renders cells much more susceptible to stress (Wong et al, 2020, BioRxiv). The parsimonious explanation, therefore, is that whilst the cellular timing mechanism remains functional when CRY is absent, the amplitude of cellular clock outputs is severely attenuated (as we showed previously in Hoyle et al., Sci Trans Med, 2017) in a fashion that impairs the fidelity of intercellular synchronisation under most conditions in vivo, as well as the molecular mechanisms of entrainment to light-dark cycles.

      With respect to the apparent discrepancy between mean periods of CKO cultured cells (~21h), SCN (~19h) and mice (~17h). This is also observed in WT cells (~26h), SCN (~25h) and mice (~24h), simply with a smaller effect size and longer intrinsic period.

      We believe this difference in effect size can adequately be explained by differences in oscillator coupling, combined with the reduced robustness of CKO timekeeping. In Figure 1F we show that the range of rhythmic periods expressed by cultured CKO fibroblasts (14-30h) is much greater than for their WT counterparts (range of 22-26h), or that which is observed when cellular oscillators are coupled in CKO SCN (19h). Thus period of CKO oscillations is demonstrably more plastic (less robust) than WT, and with a cell-intrinsic tendency towards shorter period which is revealed more clearly when oscillators are coupled.

      In vivo there is more oscillator coupling in the intact SCN than in an isolated slice, from which communication with the caudal and rostral hypothalamus has been removed. Thus it seems plausible that increased coupling in vivo, combined with positive feedback via behavioural cycles of feeding and locomotor activity, resonate with a common frequency which is shorter than in isolated tissue.

      Critically, for both WT and CKO mice/SCN, the circadian period lies within the range of periods observed in isolated fibroblasts. To communicate this rather nuanced point we have inserted the following text into the supplementary discussion:

      “Circadian timekeeping is a cellular phenomenon. Co-ordinated ~24h rhythms in behaviour and physiology are observed in multi-cellular mammals under non-stressed conditions when individual cellular rhythms are synchronised and amplified by appropriate extrinsic and intrinsic timing cues. In light of short period (~16.5h) locomotor rhythms observed in CKO mice after transition from constant light to constant dark, but failure to entrain to 12h:12h light:dark cycles, it seemed plausible that either CKO mice might entrain to an short 8h:8h light:dark (16h day) or else have a general deficiency to entrainment by light:dark cycles. The data in Figure S2 supports the latter possibility, in that neither WT nor CKO mice stably entrained to 16h cycles whereas WT but not CKO mice entrained to 24h days. The bioluminescence oscillations observed in CKO cells conform to the long-established definition of a circadian rhythm (temperature-compensated ~24h period of oscillation with appropriate phase-response to relevant environmental stimuli). Whereas the locomotor rhythms observed in CKO mice under quite specific environmental conditions correlates with both the cellular and SCN data to suggest the persistence of capacity to maintain behavioural rhythms close to the circadian range, but which is masked under most circumstances. We suggest that in vivo the (pathophysiological) stress of CRY-deficiency is epistatic to the expression of daily rhythms in locomotor activity following standard entrainment by light:dark cycles and thus, whilst not arrhythmic, also cannot be described as circadian in the strictest sense.”

      3) The authors proposed that CKId/e at least in part is the component of cytoscillator (Fig. 5D), and turnover control of PER (likely to be controlled by CKId/e) may be an interaction point between cytoscillator and canonical circadian TTFL (Fig. 4). Strictly speaking, this model is not directly supported by the experimental setting of the current manuscript. The contribution of CKId/e is evaluated in the presence of PER by monitoring the canonical TTFL output (i.e. PER2::LUC); thus it is not clear whether the kinase determines the period of cytoscillator. It would be valuable to ask whether the PF and CHIR have the period-lengthening effect on the Nrd1:LUC in the CPKO cell.

      Another excellent suggestion, thanks. The experiment, showing similar results in CKO and CPKO cells, was performed and is now reported in Revised Figure S5D. The text was amended as follows: “We found that inhibition of CK1d/e and GSK3-α/β had the same effect on circadian period in CKO cells, CPKO cells, and WT controls (Figure 5A, B, S5A, B, D).”

      Moreover, our data are further supported by findings in RBCs, where CK1 inhibition affects circadian period in a similar manner as in WT and CKO cells (Beale et al, JBR 2019).

      **Minor comments:**

      4) The authors argue that the CKO cells' rhythmicity is entrained by the temperature cycle (Fig. 2C). Because the data of CKO cell only shows one peak after the release of constant temperature phase, it is difficult to conclude whether the cell is entrained or just respond to the final temperature shift.

      We agree with the reviewer and have replaced the original figure with another recording that includes an extra circadian cycle in free-running conditions (Revised Figure 2C).

      5) It would be useful for readers to provide information on the known phenotype of TIMELESS knockout flies; TIM is widely accepted as an essential component of the circadian clock in flies; are there any studies showing the presence of circadian rhythmicity in Tim-knockout flies (even if it is an oscillation seen in limited conditions, such as the neonatal SCN rhythm in mammalian Cry knockout)?

      The reviewer is correct that TIM is widely accepted as an essential component of the circadian clock in flies. Using more sensitive modern techniques however, ~50% of classic Tim01 mutant flies exhibit significant behavioural rhythms in the circadian range under constant darkness, as reported:

      https://opus.bibliothek.uni-wuerzburg.de/frontdoor/index/index/year/2015/docId/11914

      For this reason we employed a full gene knockout of the Timeless gene (Lamaze et al., Sci Rep, 2017), where the majority of flies are behaviourally arrhythmic under constant conditions following standard entrainment by light cycles and therefore represents a more appropriate model for CRY-deficient cells.

      We have revised the legend of Figure S2 to include the following:

      “N.B. The generation of Timout flies is reported in Lamaze et al, Sci Rep, 2017. Similar to CRY-deficient mice, whole gene Timeless knockout flies are characterised as being behaviourally arrhythmic under constant darkness following entrainment by light:dark cycles: https://opus.bibliothek.uni-wuerzburg.de/frontdoor/index/index/year/2015/docId/11914”

      5) Figure 3C shows that the amount of PER2::LUC mRNA changes ~2 fold between time = 0 hr and 24 hr in the CKO cell. This amplitude is similar to that observed in WT cell although the peak phase is different. Does the PER2::LUC mRNA level show the oscillation in CKO cells?

      No, we think we have shown convincingly this is not the case. We argue the data in figure 3C show that: (a) there is no circadian variation in mRNA PER2::LUC expression (mRNA levels increase but no trough is observed) and (b) that the temporal relationship between protein and mRNA as observed in WT is broken; i.e. the CRY-independent circadian variation in protein levels cannot be “driven by” changes in transcript levels. Similar results were obtained using transcriptional reporters Per2:LUC and Cry1:LUC (Figure S3E and F). Moreover, our findings are also in line with previous reports, such as Nangle et al. (2014, eLife) and Ode et al. (Mol Cell, 2017).

      6) Figure 3D: the authors discuss the amplitude and variation (whether the signal is noisier or not) of reporter luciferase expression between different cell lines. However, a huge difference in the luciferase signal can be observed even in the detrended bioluminescence plot. This reviewer concerns that some of the phenotypes of CKO and CPKO MEF reflect the lower transfection efficiency of the reporter gene, not the nature of circadian oscillators of these cell lines.

      As reported in the methods, these are stable cell lines rather than transiently transfected cells. The detrended luciferase data presented here do not actually reflect raw levels of luciferase protein expression, but rather reflect the amount of deviation from the 24 hour average. To make it easier to compare expression levels of Per2:LUC and Nr1d1:LUC between the different cell lines we have added figure S3H, presenting the average raw bioluminescence levels over 24 hours (after 24 hours of recovery from media change; ie from 24-48 hours). Using these data one can appreciate that expression levels of the Per2 reporter are never lower in CRY KO cells when compared to WT. We hope these data can take away the reviewer’s concerns about expression levels causing the differences observed.

      Reviewer #1 (Significance (Required)): Although Cryptochrome (Cry) has been considered a central component of the mammalian circadian clock, several studies have shown that circadian rhythms are maintained in the absence of Cry, including in the neonate SCN and red blood cells. Thus, although the need for Cry as a circadian oscillator has been debated, its essential role as a circadian oscillator remains established, at least in the cell-autonomous clock driven by the TTFL. This study provides additional evidence that the circadian rhythmicity can persist in the absence of Cry. More general context, the presence of a non-TTFL circadian oscillator has been one of the major topics in the field of circadian clocks except for the cyanobacteria. In mammals, the authors’ and other groups lead the finding of circadian oscillation in the absence of canonical TTFL by showing the redox cycle in red blood cells (O’Neil, Nature 2011). The presence of circadian oscillation in the absence of Bmal1 is also reported recently(Ray, Science 2020). Bmal1(-CLOCK), CRY, and PER compose the core mechanism of canonical circadian TTFL; thus, this manuscript put another layer of evidence for the non-TTFL circadian oscillation in mammals. Overall, the manuscript reports several surprising results that will receive considerable attention from the circadian community. This reviewer has expertise in the field of mammalian circadian clocks, including genomics, biochemistry, and mice's behavior analysis.

      Reviewer #2 (Evidence, reproducibility and clarity (Required)): In the canonical model of the mammalian circadian system, transcription factors, BMAL1/CLOCK, drive transcription of Cry and Per genes and CRY and PER proteins repress the BMAL1/CLOCK activity to close the feedback loop in a circadian cycle. The dominant opinion was that CRY1 and CRY2 are essential repressors of the mammalian circadian system. However, this was challenged by persistent bioluminescence rhythms observed in SCN slices derived from Cry-null mice (Maywood et al., 2011 PNAS) and then by persistent behavior rhythms shown by the Cry1 and Cry2 double knockout mice if they are synchronized under constant light prior to free running in the dark (Ono et al., 2013 PLOS One). In the manuscript, the authors first confirmed behavioral and molecular rhythms in the Cry1/Cry2- deficient mice and then provided evidence to suggest the rhythms of Per2:LUC and Nr1d1:LUC in CKOs are generated from the cytoplasmic oscillator instead of the well-studied transcription and translation feedback loop: Constant Per2 transcription driven by BMAL1/CLOCK plus rhythmic degradation of the PER protein result in a rhythmic PER2 level in the absence of both Cry1 and Cry2, which suggests a connection between the classic transcription- and translation-based negative feedback loops and non-canonical oscillators. **Major points:** Line 38-39, "Challenging this interpretation, however, we find evidence for persistent circadian rhythms in mouse behavior and cellular PER2 levels when CRY is absent." The rhythmic behavioral phenotype of cry1 and cry2 double knockout mice was first documented by Ono et al., 2013 PLOS ONE, in which eight cry1 and cry2 double knockout mice after synchronization in the light displayed circadian periods with different lengths and qualities. The paper reported two period lengths from the Cry mutant mice: "An eye-fitted regression line revealed that the mean shorter period was 22.86+/-0.4 h (n= 8) and the mean longer period was 24.66+/-0.2 h (n =9). The difference of two periods was statistically significant (p, 0.01).", either of which is quite different from the ~16.5 hr period in Figure 1B of the manuscript. A brief discussion on the period difference between studies will be helpful for readers to understand. Period information from the individual mouse should be calculated and shown since big period variations exist among CKO mice (Ono et al., 2013 PLOS One).

      Thanks for this suggestion. The mice used by Ono et al were raised from birth in constant light, whereas we used mice that were weaned and raised in normal LD cycles before being subject to constant light then constant dark as adults. Instead of the somewhat subjective fitting of regression lines by eye performed by Ono et al, our analysis was performed using the periodogram analysis routine of ClockLab 6.0 with a significance threshold for rhythmicity of p=0.0001. We have now repeated this experiment with 10 adult CKO mice (male and female), and found no evidence for two period lengths in that the second most significant period was consistently double that of the first. As the reviewer suggests, there is a much broader distribution of CKO mouse periods compared with WT, as we also found in cultured cells and SCN. These new data are now reported in revised Figure S1B & C. We have also included a statement about how our study differs from Ono et al in the supplementary discussion.

      The behavioral phenotype of Cry-null mice and luminescence from their SCNs are robustly rhythmic while fibroblasts derived from these mice only produce rhythms with very low amplitudes compared with those in WT, which may reflect the difference between the SCN’s rhythm and peripheral clocks. The behavioral phenotype is supposed to be controlled mainly by SCN. However, most molecular analyses in the work were done with MEF and lung fibroblasts. These tissues may not be the best representative of the behavioral phenotype of the CKO mice.

      Behavioural rhythms of CKO mice are significantly less robust than WT, with mean amplitude less than 50% of WT controls (Figures 1A & B, revised S1B. Furthermore, as reported, 40% of CKO SCN slices exhibited PER2::LUC rhythms, compared with 100% of WT SCN slices (as also observed by Maywood et al., PNAS, 2013), and therefore are also less robust by the definition used in this manuscript.

      As now discussed in the revised supplementary discussion:

      Circadian timekeeping is a cellular phenomenon. Co-ordinated ~24h rhythms in behaviour and physiology are observed in multi-cellular mammals under non-stressed conditions when individual cellular rhythms are synchronised and amplified by appropriate extrinsic and intrinsic timing cues.”

      The objective of this study was to understand the fundamental determinants that allow mammalian cells to generate a circadian rhythm, which we find does not include an essential role for CRY genes/proteins. Thus the cell is the appropriate level of biological abstraction at which to investigate the phenomenon, whereas the SCN and behavioural recordings simply serve to illustrate the competence of CRY-independent timing mechanisms to co-ordinate biological rhythms at higher levels of biological scale which are manifest under some conditions. To reiterate, the behavioural data supports the cellular observations, not the converse.

      Stronger evidence is needed to fully exclude the possibility that in CKO cells, the rhythm is not generated by PERs' compensation for the loss of Crys to repress BMAL1 and CLOCK. Since the rhythms of Per:LUC or Nr1d1:LUC (Figures 3D and S3E) are much weaker than those in WT, molecular analyses might not be sensitive enough to reflect the changes across a circadian cycle in the CKOs if the TTFL still occurs. CLOCKΔ19 mutant mice have a ~4 hr longer period than WT (Antoch et al., 1997 Cell; King et al., 1997 Cell). CLOCKΔ19; CKO cells or mice should be very helpful to address the question. Periods of Per:LUC and Nr1d1:LUC from the CLOCKΔ19; CKO should be similar to those in the CKO alone if the transcription feedback does not contribute to their oscillations.

      We agree this would be an interesting experiment, however the data in this manuscript and Wong et al. (BioRxiv, 2020), whilst not disputing the existence of the TTFL, strongly suggest that it fulfils a different function to that which is currently accepted and is not the mechanism that ultimately confers circadian periodicity upon mammalian cells. CLOCKΔ19 is an antimorphic gain-of-function mutation with many pleiotropic effects. Therefore, if the TTFL is not the basis of circadian timekeeping in mammalian cells, it follows that the CLOCKΔ19 mutation may not elicit its effects on circadian rhythms through delaying the timing of transcriptional activation, as was proposed. As such, whether or not CLOCKΔ19 alters circadian period of CKO cells/mice would not allow the two models to be distinguished in the way that the reviewer envisions.

      Secondly, we cannot detect any interaction between PER2 and BMAL1 in the absence of CRY using an extremely sensitive assay.

      Thirdly, very strong biochemical evidence suggests that PER has no repressive function in the absence of CRY (Chiou et al., 2016; Kume et al., 1999; Ode et al., 2017; Sato et al., 2006).

      Finally, in several figures particularly 3C and 4A, we show that PER2 peaks at the same time CKO and WT cells, but in CKO cells this is not accompanied by a coincident peak in the mRNA. Thus, even if PER were able to repress BMAL1/CLOCK without CRY, rhythms in PER2 protein level could not be explained by some residual PER/BMAL1-dependent TTFL mechanism.

      To address the reviewer’s concern however, we have employed mouse red blood cells which offer unambiguous insight into the causal determinants of circadian timing, as we can be absolutely confident that there is no transcriptional contribution to cellular timekeeping. Briefly, we took fibroblasts and RBCs from WT, short period Tau/Tau and long period Afh/Afh mutant mice. The basis of the circadian phenotype of these mutations is quite well established as occurring through the post-translational regulation of PER and CRY proteins respectively, and result in short and long period PER2::LUC rhythms compared with WT fibroblasts. RBCs do not express PER or CRY proteins, and commensurately no genotype-dependent differences of RBC circadian period were observed (Beale et al, 2020, in submission). In contrast, RBC circadian rhythms are sensitive to pharmacological inhibition of casein kinase 1 (Beale et al., JBR, 2019).

      Lines 51-52, "PER/CRY-mediated negative feedback is dispensable for mammalian circadian timekeeping" and lines 310-311, "We found that transcriptional feedback in the canonical TTFL clock model is dispensable for cell-autonomous circadian timekeeping in animal and cellular models." The authors have not excluded the possibility that the rhythmic behaviors of the CKO mice are derived from the PERs' compensation for the role of Crys in the feedback loop of the circadian clock in the SCN. In the fibroblasts, only two genes, Per2 and Nr1d1, have been studied in the work, which cannot be simply expanded to the thousands of circadian controlled genes. Also amplitudes of PER2:LUC and NR1D1:LUC in the CKOs are much lower than those in WT and no evidence has been provided to show that their weak rhythms are biologically relevant.

      The definition of a circadian rhythm (Pittendrigh, 1960) does not mention biological relevance or stipulate any lower threshold for amplitude. As now stated in the revised text (page 6):

      PER2::LUC rhythms in CKO cells were temperature compensated (Figure 2A, B) and entrained to 12h:12h 32°C:37°C temperature cycles in the same phase as WT controls (Figures 2C), and thus conform to the classic definition of a circadian rhythm (Pittendrigh, 1960) – which does not stipulate any lower threshold for amplitude or robustness.

      We make no claims about biological relevance or amplitude in this manuscript, which are addressed in our related manuscript (Wong et al., BioRxiv, 2020). In this related manuscript, we explicitly address whether CRY is necessary for mammalian cells to maintain a circadian rhythm in the abundance of clock-controlled proteins and find that it is not. Indeed, twice as many rhythmically abundant proteins are observed in CKO cells than WT controls, which suggests that, if anything, CRY functions to suppress rhythms in protein abundance rather than to generate them.

      We observe circadian rhythms in the activity of two different bioluminescent reporters, which have already been extensively characterised. The mouse and SCN data in figure 1 are correlative, and simply show that previous published observations are reproducible. PER2::LUC oscillations are not accompanied by Per2 mRNA oscillations. This, together with the absence of a BMAL1-PER2::LUC complex strongly argues against a model where PER2 oscillations are driven by residual (PER2-driven) transcriptional oscillations.

      We therefore concede the reviewer’s point that we “cannot exclude rhythmic behaviors of the CKO mice are derived from the PERs' compensation for the role of Crys in the feedback loop of the circadian clock in the SCN”. The reviewer will agree however, that there exists very strong biochemical evidence suggests that PER has no repressive function in the absence of CRY (Chiou et al., 2016; Kume et al., 1999; Ode et al., 2017; Sato et al., 2006); that there exists no experimental evidence to suggest that PERs can fulfil this function in the absence of CRY in any mammalian cellular context; and finally that our observations are not consistent with the canonical model for the generation of circadian rhythms in mammals.

      We have therefore amended the text to focus on CRY specifically, as follows:

      PER/CRY-mediated negative feedback is dispensable for mammalian circadian timekeeping

      Page 12. “We found that CRY-mediated transcriptional feedback in the canonical TTFL clock model is dispensable for cell-autonomous circadian timekeeping in cellular models. Whilst we cannot exclude the possibility that in the SCN, but not fibroblasts, PER alone may be competent to effect transcriptional feedback repression in the absence of CRY, we are not aware of any evidence that would render this possibility biochemically feasible.”

      **Minor points:** Lines 66-67, "...(Dunlap, 1999; Reppert and Weaver, 2002; Takahashi, 2016)." to "... (reviewed in Dunlap, 1999; Reppert and Weaver, 2002; Takahashi, 2016)."

      Thanks, changed as requested.

      Line 70, "...((Liu et al., 2008..." to "...(Liu et al., 2008..."

      Thanks, changed as requested.

      Lines 174-175, "Considering recent reports that transcriptional feedback repression is not absolutely required for circadian rhythms in the activity of FRQ...". Larrondo et al., 2015 paper says "however, in such ∆fwd-1 cells, the amount of FRQ still oscillated, the result of cyclic transcription of frq and reinitiation of FRQ synthesis." The point of the paper is "we unveiled an unexpected uncoupling between negative element half-life and circadian period determination." instead of "...transcriptional feedback repression is not absolutely required for circadian rhythms in the activity of FRQ,"

      This is a good point which, following discussion with Profs Dunlap and Larrondo, we have revised into “no obligate relationship between clock protein turnover and circadian regulation of its activity” – a more accurate summary of their findings.

      Lines 249-252, "CKO cells exhibit no rhythm in Per2 mRNA (Figure 3C, D), nor do they show a rhythm in global translational rate (Figure S4A, B), nor did we observe any interaction between BMAL1 and S6K/eIF4 as occurs in WT cells (Lipton et al, 2015) (Figure S4C)." In figures 3D and S3E, in CKO and CPKO cells the Per2:LUC data without fitting look better than that of Nr1d1:LUC. But the Nr1d1:LUC rhythm became clear after fitting the raw data. So to better visualize the low amplitude rhythm, if any, of Per2:LUC and compare with Nr1d1:LUC, fitted the Per2:LUC data in CKOs and CPKOs in Figure 3D and S3E should be shown as what has been done to Nr1d1:LUC.

      Thanks, these data can be found in Figure S3F. The detrended Per2:Luc CKO and CPKO bioluminescence traces were better fit by the null hypothesis (straight line) than a damped sine wave (p>0.05) and so were not significantly rhythmic by the criteria used in this manuscript.

      Lines 258-259, "much less than the half-life of luciferase expressed in fibroblasts under a constitutive promoter" In figure S4D, the y-axis of the PER2::LUC is ~800 while the y-axis of the SV40::LUC is ~600000. The over-expressed LUC by the SV40 promoter might saturate the degradation system in the cell so the comparison is not fair. A weaker promoter with the level similar to Per2 should be used to make the comparison.

      Thank you for this suggestion. In our experience, the SV40 promoter is actually a rather weak promoter compared with CMV, and faithfully facilitates the constitutive (non-rhythmic) expression of heterologous proteins such as Luciferase (Feeney et al., JBR, 2016). It has been shown previously that constitutive over-expression of heterologous proteins such as GFP or even CRY1 does not affect circadian rhythms in fibroblast cells (e.g. Chen et al., Mol Cell, 2009). To address the reviewer’s reasonable concern however, multiple stable SV40:Luc fibroblast lines were generated by puromycin selection, grown to confluence in 96-well plates, then treated with 25 μg/mL CHX at the beginning of the recording. Random genomic integration of SV40:Luc leads to a broad range of different levels of luciferase expression, evident from the broad range of initial luciferase activities. For each line the decline in luciferase activity was fit with a simple one-phase exponential decay curve (R2≥0.98) to derive the half-life of luciferase in each cell line. There was no significant relationship between the level of luciferase expression and luciferase stability (straight line vs. horizontal line fit p-value = 0.82). Therefore constitutive expression of SV40:Luc in fibroblasts does affect the cellular protein degradation machinery within the range of expression used for our half-life measurements. These new data are reported in Revised Figure S3H.

      Line 430, "sigma" to "Sigma".

      Changed

      In figure S2, the classification of rhythms in Drosophila is not clear since even the "Robustly rhythmic" ones have high background noise. Detrending or fitting the data might be able to improve the quality of the rhythms prior to classification.

      These are noisy data as they come from freely behaving flies. The mean data was shown in Figure S3A and individual examples in S3B, and look very similar to previous bioluminescence fly recordings of XLG-LUC flies in papers from the Stanewsky lab who have published extensively using this model. The classifications arose from double-blinded analysis of the bioluminescence traces by several individuals, but we agree that this was not clearly communicated in our original submission. In Revised figure S2 we now present the mean bioluminescence traces, with and without damped sine wave vs. straight line fitting, as suggested, which is more consistent with the mammalian cellular data presented elsewhere.

      In figure S3B, the original blots for Per2 including Input and IP should be shown.

      The original blots for BMAL1 are shown in figure S3I. PER2::LUC levels were assessed by measuring bioluminescence levels present on the anti-bmal1-beads, as described in the figure 3B legend.

      Supplemental information Line 44, "...(reviewed in (Lakin-Thomas,..." to "...(reviewed in Lakin-Thomas,..."

      Changed

      Line 188, "Period CDS", the full name of CDS should be provided the first time it appearances.

      Changed to “coding sequence”.

      Reviewer #2 (Significance (Required)): The work suggests a link between the TTFL and non-canonical oscillators, which should be interesting to the circadian field.

      Reviewer #3 (Evidence, reproducibility and clarity (Required)): **Summary:** The paper "CRYPTOCHROMES confer robustness, not rhythmicity, to circadian timekeeping" by Putker et al. answers the question of whether or not the rhythmic abundance of clock proteins is a prerequisite for circadian timekeeping. They addressed this by monitoring PER2::LUC rhythms in WT and CRY KO (CKO) cells. CRY forms a complex with PER, which in turn represses the ability of CLOCK/BMAL1 to drive the expression of clock-controlled genes, including PER and CRY. Consistent with previous observations, the authors found residual PER2::LUC rhythms in CKO SCN slices, fibroblasts and in a functional analogue KO of CRY in Drosophila, even in the absence of rhythmic Per2 transcription due to the loss of CRY as a negative regulator of the oscillation. They have shown that these rhythms, in the absence of CRY, follow the formal definition of circadian rhythms. They attributed these residual PER2::LUC rhythms to the maintenance of oscillation in PER2::LUC stability independent of CRY, by testing the decay kinetics of luciferase activity when translation is inhibited. Moreover, they implicated the kinases CK1d/e and GSK3 to be involved in regulating PER2::LUC post-translational rhythms through kinase inhibitor studies. They concluded that CRY is not necessary for maintaining PER2::LUC rhythms, but plays an important role in reinforcing high-amplitude rhythms when coupled to a proposed "ctyoscillator" likely composed of CK1d/e and GSK3. **Major comments:** The authors have shown sufficient data that under different testing conditions (mice locomotor activity, SCN preps or fibroblasts), behavioral rhythms and PER2::LUC rhythms are still observed in the CRY KO (CKO) cells, contrary to a previous study (Liu et al., 2007). They also indicated limitations to some of the.experimental work. However, there are some parts of the paper that need clarification to support their conclusions. 1.In Fig. 1A, the x-axes of the actograms for WT and CKO are different. While they mentioned this in the figure legend, and described the axis transformation in Fig. S1A, they need a justification statement about why they did this in the results.

      Thanks, we have included the following sentence in the results section as requested:

      Figure 1 representative actograms are plotted as a function of endogenous tau (**t) to allow the periodic organisation of rest-activity cycles to be readily discerned; 24h-plotted actograms are shown in Figure S1A and S2A

      2.In an attempt to show conservation of their proposed role for CRY, they tested the model system Drosophila melanogaster where TIMELESS serves as the functional analogue of CRY. While they showed in the figures and described in the text that rhythms still persisted with lower relative amplitude in the TIMELESS-deficient flies, they did not describe any period differences between WT and mutant. Showing the period quantification in Supp. Fig. S2 using the robustly rhythmic datasets, and describing this data in the text, will strengthen their claim.

      These analyses are now reported in revised Figure S2 as requested. As described in our response to reviewer 2, the “robustly rhythmic” flies were scored as such through double-blinded analysis by several individuals. We hope the reviewer will appreciate our concern that exclusion of the majority of TIMELESS-deficient flies that were not robustly rhythmic might skew their apparent period by unconscious bias towards favouring traces that most clearly resemble robustly rhythmic WT controls. To avoid any potential bias we therefore included all flies of both genotypes in the analysis of circadian period for the revised figure, as suggested by our other reviewers.

      In Fig. S2B, there is no clear distinction between the representative datasets shown for poorly rhythmic and arrhythmic, i.e. they all appear arrhythmic, without an indicated statistical test. The authors could present better representative data to better reflect the categories.

      As described above, we now show the grouped mean with and without fitting for all flies of both genotypes. The statistical test for rhythmicity and analysis of circadian period is now the same as was performed for the cellular data presented elsewhere.

      3.In Fig. 2A, the authors note the lack of rhythmicity in the CKO fibroblasts in the 1st three days at 37oC. How are the conditions here different from fibroblasts in Fig. 1E, where rhythms are seen during the 1st three days in CKO fibroblasts?

      As discussed in the manuscript, PER2::LUC rhythms in CKO cells and SCN are observed stochastically between recordings i.e. if one dish in a recording showed rhythms, all dishes showed rhythms and vice versa. The media change that occurred after 3 days in Fig 2A, in this case, was sufficient to initiate clear rhythms of PER2::LUC in all experimental replicates. In other experiments, media change did not have this effect. Herculean efforts by multiple lab members over many years, including the PI, have been unable to delineate the basis of this variability – which is discussed at length in the thesis of Dr. David Wong https://www.repository.cam.ac.uk/handle/1810/300610. As such, we clearly state in the discussion:

      We were unable to identify all of the variables that contribute to the apparent stochasticity of CKO PER2::LUC oscillations, and so cannot distinguish whether this variability arises from reduced fidelity of PER2::LUC as a circadian reporter or impaired timing function in CKO cells. In consequence, we restricted our study to those recordings in which clear bioluminescence rhythms were observed, enabling the interrogation of TTFL-independent cellular timekeeping.”

      1. The authors claimed in the results section- "in contrast and as expected, Per2 mRNA in WT cells varied in phase with co-recorded PER2::LUC oscillations." but Fig. 3C does not show this expected lag between mRNA and protein levels. This needs to be explained

      No lag is expected in vitro. A lag between PER protein levels and Per mRNA does occur in vivo and is very likely to attributable to daily rhythms in feeding (Crosby et al, Cell, 2019), where increased insulin signalling elicits an increase in PER protein production 4-6h after E-box and GRE-stimulated increase in Per transcription.

      When luciferin is saturating intracellularly, PER2::LUC activity correlates most closely with the amount of PER2::LUC protein that was translated during the preceding 1-2h, rather than the total amount of PER2, due to the enzymatic inactivation of the luciferase protein (Feeney et al, JBR, 2016). Consistent with many previous observations, under constant conditions, the rate of nascent PER protein synthesis is largely determined by the level of Per2 mRNA, and thus more similar phases are observed between protein and mRNA in vitro than in vivo.

      We have inserted an additional citation of Feeney et al at this point in the text to make this clear.

      5.In Figs. 5A-B, the PER2::LUC periods in the CKO untreated cells seem to vary significantly between A, B, and C. While this could be due to the high variability in the rhythms that were previously described by the authors, the average periods here seem to be longer than the one reported in Fig. 1F. Are there specific condition differences?

      There are no specific condition differences. As reported in Figure S1B, D & E, the range of CKO cellular periods is simply much broader than for WT cells. Over several dozen experiments the average period was significantly shorter, but the period variance is an equally striking feature of rhythms in these cells which we take as evidence for their lack of robustness.

      *Would additional experiments be essential to support the claims of the paper?*

      1. There is sufficient experimental data to support the major claims; however some suggested experiments are listed below.

        a. If CKO exhibits residual rhythms in PER::LUC, it would be interesting to know how CRY overexpression influences PER2::LUC rhythms, or point to previous reference papers which may have already shown such effects. The prediction would be PER2::LUC levels will still be rhythmic when CRY is overexpressed. What would be the extent of "robustness" conferred by CRY on PER2::LUC rhythms based on CRY KO and overexpression studies?

      These experiments have largely already been performed (see Chen et al., Mol Cell; Nangle et al., eLife, 2014; Fan et al., Curr Biol, 2007; Edwards et al., PNAS, 2016) and are cited in this manuscript. As suggested, PER2 rhythms remain intact under CRY1 over-expression, though are clearly perturbed, but their robustness was not investigated in any detail. We hope to be able to address this important question in our subsequent work

      The authors found that CK1d/e and GSK3 contribute to CRY-independent PER2 oscillations by showing that addition of kinase inhibitors affect the PER2::LUC period lengths in WT and CKO in the same manner. It would be interesting to know if a) PER2::LUC stability and b) PER2 phosphorylation status, is affected in WT and CKO in the presence of the inhibitors, or point to previous reference papers which may already have shown such effects.

      As the reviewer points out, PER2 stability is already reported to be regulated via phosphorylation by GSK3 and CK1. We have made explicit reference to this in the revised manuscript as follows:

      In contemporary models of the mammalian cellular clockwork CRY proteins are essential for rhythmic PER protein production, however, the stability and activity of PER proteins are also regulated post-translationally (Lee et al., 2009; Philpott et al., 2020; Iitaka et al, 2005).”

      *Are the data and the methods presented in such a way that they can be reproduced?*

      1. The protocol for the inhibitor treatments are not in the main or supplemental methods.

      In the main text methods, section luciferase recordings we state: “For pharmacological perturbation experiments (unless stated otherwise in the text) cells were changed into drug-containing air medium from the start of the recording. Mock-treatments were carried out with DMSO or ethanol as appropriate.”

      *Are the experiments adequately replicated and statistical analysis adequate?*

      1. All experiments had the sufficient number of technical and biological replicates to make valid statistical analyses. For Fig. S2, the authors used RAIN to assess rhythmicity in WT and mutant flies, but it is not clear whether the different categories (rhythmic, poorly rhythmic, and arrhythmic) were based on amplitude differences alone, or a combination of amplitude and p-values as determined by RAIN.

      As reported above, we have revised the analysis of the fly data to be consistent with the cellular data reported elsewhere in the manuscript.

      **Minor comments:** *1. Are prior studies referenced appropriately?* Authors may wish to include Fan et al., 2007, Current Biology which demonstrated that cycling of CRY1, CRY2, and BMAL1 is not necessary for circadian-clock function in fibroblasts.

      Apologies for the omission of citation to this excellent paper. Now referenced in the introduction.

      *2. Are the text and figures clear and accurate?* Figures were clear and illustrated well. See minor comments on text below:

      1. Other minor comments

      Main Text: p3, line 62; p12, line l32: It doesn't seem necessary or appropriate to cite the dictionary for the definition of robust.

      Thanks for this suggestion. During preparation of the manuscript we found that there was some disagreement between authors as to the meaning of robustness in a circadian context. We therefore feel it most necessary to define clearly what we mean by the use of this word to avoid any potential ambiguity.

      p4, line l87: "~20 h" rhythms instead of "~20h-hour" p3, line 70; p5, line 121; p14, line 380; p16, line 416 and p18, line 458: Close parentheses have been doubled in parenthetical references. p14, line 363: "crassa" instead of "Crassa" p17, line 430: "Sigma" instead of "sigma" p18, lines 464 and 483; p20, line 521: put a space between numerical values and units, to be consistent with other entries p19, line 488: "luciferase" instead of Luciferase p20, line 512: "Cell Signaling" instead of "cell signalling" p20, line 526: "single" instead of "Single"

      We thank the reviewer for his/her thoroughness, all of the above have been changed.

      Main figures: Fig. 2 p37, line 921: close parenthesis was doubled on "red"

      This was actually correct.

      Fig. 4 p41, line 989: "0.1 mM" instead of "0.1 mM" for consistency throughout text Supplementary text: line 171: "30 mM HEPES" instead of "30mM HEPES" line 184: "Cell Signaling" instead of "cell signalling" Supplementary figures: Fig. S2A "Drosophila melanogaster" instead of "Drosophila Melanogaster"

      All of the above have been changed.

      Reviewer #3 (Significance (Required)): This paper revisits the previously proposed idea that rhythmic expression of central TTFL components is not essential for circadian timekeeping to persist. However, this paper does not add a significant advance in the understanding of the underlying reasons behind sustained clock protein rhythmicity like PER in the absence of CRY, since such mechanisms in functional analogs have been shown in other systems, like Neurospora (Larrondo et al., 2015). However, this paper does clarify some issues in the field, such as discrepancies between behavioral and cellular rhythms observed in CKO mice, leading future researchers to examine closely the conditions of their CKO rhythmic assays before making conclusions pertaining to rhythmicity. The identification of the kinases as components of the proposed cytosolic oscillator (cytoscillator) needs further validation, but this is perhaps beyond the scope of the paper. The data provides incremental evidence for the existence of a cytoscillator, but opens up opportunities to identify other players, like phosphatases, to establish the connection between the central TTFL and the proposed cytoscillator.

    1. For the current generation, games may represent the bestway of tapping that sense of engagement with learning

      I wrote a facebook post about this last year that I think is relevant here.

      "It occurs to me that one of my core beliefs as a teacher is in the value of games and game like activities for learning.

      Some recent insights: I watched a student struggle with exchanging different values of money and after asking him a few questions found out that board games were not big at home. I might be about to give him my spare monopoly board if his mom is okay with it, and ask if she'll play with him as his homework: games build numeracy.

      Today I had to cover for someone and we played Swap! which is like uno but with some twists. It was a social skills class, so this was a pretty appropriate activity. Not only did were the kids good sports about it, the game requires everyone to pay attention to everyone else's moves in order to succeed, and it was fun to watch them really try to do that.

      The Payday game has helped kids understand additive inverses and adding positive and negative integers. We're still figuring out how to expand the metaphor to subtraction but we can add a die or a coin and do that too.

      We swapped out the traditional dice in shut the box for a d4 and a d12 and changed the rules so that you can do any operation you want with the two numbers you roll. Kids are excited to play.

      Kids who refuse other class activities will come up and play a math game, and will even try to learn the concept built into the game if it helps them win the game.

      (Additionally) When my class at BAMS invented the game we called Duels, we made the rules work so that everybody had to have the right answer because you never knew who was being called on. This led to kids explaining things to each other and working together. (Ask me about the rules of Duels sometime. It's an ideal game if you are popsicle stick user)."

    1. Reviewer #1:

      This paper describes the impact of outliers in normative cortical thickness (CT) measurements when examining those suffering from autism spectrum disorder (ASD). The authors used the ABIDE sample and binned subjects by age, and assessed outliers as a function of a "w-score" which they estimated across CT parcellations across the entire cortex. They then demonstrate that cortical thickness differences that can ascribed to ASD can essentially be attributed to a small number of outliers within the sample. They also demonstrate that this w-score may be sensitive to clinical variables as well.

      Overall, it is unclear to me what the exact goal of the work is: To describe the anatomy of ASD better? To subtype? Or is there another "take-home" message of this paper? I would imagine that the case-control differences in most neurodevelopmental disorders with high heterogeneity and high variability would demonstrate a similar kind of trend. And thus, at the end of the day, I am not sure how much this technique advanced our understanding of ASD.

      Issues and Questions:

      1) It is unclear from the methods how the authors deal with motion and image quality. Recent work by Pardoe and Bedford demonstrate the importance of dealing with this issue, particularly in the context of the ABIDE sample. This would likely have a significant impact on any of the results. It's unclear if the use of the Euler index at the extremes of the distribution of the dataset being used is sufficient. How did the authors come up with their Euler number cut-off?

      2) The W-score could use a much better explanation. It is not clear to me as to what it is and how this should be interpreted. The lack of information regarding the number of age-bins used also makes interpreting these findings confusing in my mind.

      3) The authors report that, "The median number of brain regions per subject with a significant p-value was 1 (out of 308), indicating that the w-score provides a robust measure of atypicality." I guess this could be true, but given the variation in normative ageing and development, I suspect this would also be true of a large number of TD children. That being the case, would it be worth doing a permutation test to determine the threshold of how man "atypical" areas one could expect by chance?

      4) The authors note "Unfortunately, despite a significant female subgroup, the age-wise binning greatly reduced the number of bins with enough data-points in the female group." I understand that this could indeed be a problem. However, I think it would be good for the authors to provide more details. Potentially a histogram to demonstrate the issue. My feeling is that with sex difference with respect to ASD, the more information that could be provided the better. Overall, it is unclear to me as to how useful a sex-specific analysis may be in this particular context given the sample sizes available in ABIDE.

      5) Results, page 8: "Because we also had computed w-scores from our normative age-modelling approach, we identified specific 'statistical outlier' patients for each individual region with w-scores > 2 standard deviations from typical norms and excluded them from the case-control analysis."

      I'm not sure I agree with the premise of this statement. First, it is hard to know without seeing all of the data, but based on Fig 1, it seems that there are ASD individuals that fall on both sides of this distribution. So if there are effect sizes that can be gleaned, this would be in spite of the variability. Second, it would be paramount to determine how many people are outliers-by-region. This, in and of itself, would be useful information. If a significant proportion of individuals can be identified as outliers, this suggests that variability is the norm rather than an exception. I'm skeptical as to whether you get interesting information from removing these individuals from analyses.

      6) Result, page 9: "While the normative modelling approach can be sensitive to different pathology." I don't think you're capturing anything interesting about pathology with this method, especially as it pertains to CT values.

      7) Result, page 9-10: I'm still confused by this notion of atypicality. Presumably this suggests that 5-10% of all ASDs are more than 2SDs from a normative distribution. But is this at both tails of the distribution? There are significant interpretational issues with this. thus, it is imperative on the authors to do a better job of describing these distributions.

      8) Part of the rationale of this paper is that using the w-score is far more robust than using simple CT values. I'm sure that residualized CT values could have been used for any of these analyses. If that were to be done how would this change the results?

      Minor comments and suggestions on presentation:

      1) While this paper has some merits, I found it hard to read. There is not a clear delineation between the methods and the results, and some methodological considerations are written into the results section and vice-versa.

      2) In the introduction, the authors use the word "deviance" to describe what appears more to me like age-related variation and heterogeneity in ASD. Deviance may be too strong a term and easily mis-interpretable. I would suggest replacing it with something a bit more like variation. Also, the work at the institution of the main author (for example by Baron-Cohen and authors) really champions the use of terms like "neurotypical" rather normally developing. I think, in general, the authors may want to take their cues from this type of language.

      3) This passage in the Introduction need of references. The work by Hong (in Boris Bernhardt's group), Bedford (in Mallar Chakravarty's group), Schuetze (in Signe Bray's group), and Meng-Chuan Lai all come to mind.

      "Even within mesoscopic levels of analysis such as examining brain endophenotypes, heterogeneity is the rule rather than the exception (Ecker, 2017). At the level of structural brain variation, neuroimaging studies have identified various neuroanatomical features that might help identify individuals with autism or reveal elements of a common underlying biology (Ecker, 2017). However, the vast neuroimaging literature is also considerably inconsistent, with reports of hypo- or hyper-connectivity, cortical thinning versus increased grey or white matter, brain overgrowth, arrested growth, etc., leaving stunted progress towards understanding mechanisms driving cortical pathophysiology in ASD."

      4) I found the Discussion missed the mark. It was mostly written as a rehash of the results, with no real biological interpretation. There is not a sufficient examination of the relationship of these findings to other important papers (Kundrakpham, Bedford, Hong, Ecker, Hyde, Lange, etc...).

      5) Figure 3 - The colour bars should be labelled.

    1. Just because something is available instantly to vision does not mean that it is available instantly to consciousness. Or, in slightly more general terms: access is not synonymous with learning. What turns access into learning is time and strategic patience.

      I think that this is really applicable to multi-tasking, even with the mundane, because we typically want to keep as many senses busy when possible (watching Netflix while exercising, eating while studying, etc.). We may be viewing something but not necessarily taking it in, as we technically cannot multitask but only switch between tasks. So in this way, it enforces the idea that multitasking doesn't allow for deep thinking or absorption. It's focus and time that allows you to actually gain something from a task, so the 'access' they talk about that may occur during multitasking is truly ineffective in the long-run. It's worth questioning why we allow ourselves to be indulged in habits that prove to be distracting and harmful to our focus.

  2. classroom.google.com classroom.google.com
    1. page 22 "Early anthropologists of language, like Sapir and Whorf , took this insight to its logical extreme when they argued that we are all, as it were locked into our cultural perspectives or 'mind-sets' and that language is the best clue we have to that conceptual universe. This observation, when applied to all human cultures, lies at the root of what, today, we may think of cultural or linguistic relativism."

    1. The use of the phrase‘especially a woman’in the earlier mentioned Supreme Court rul-ing is case and point; not only is it patronizing but it implies an imperative of modesty forwomen. The purpose is to suggest that women deserve extra protection, but by linking ofprotections to gender presentation, it creates a scenario where a court may blame a victimif it deems she has not appropriately performed femininity. In other words, recognition ofthe right to bodily integrity is made contingent upon one’s conformity to social norms,particularly with regards to gender

      This is a very powerful idea. I think by default, society as a whole tends to have a lot of sympathy for women who had been sexually abused, until we find out they had been drinking or dancing or wearing skintight clothing or even wearing limited clothing. We don't get to pick and choose when its not okay for someone to be sexually abused; it is NEVER okay to be sexually abused.

    1. According to numerous research studies, when adults hear a statement twice, they are more likely to think it is true compared with when they have heard it only once. Multiple theoretical explanations exist for this illusory-truth effect. However, none of the current theories fully explains how or why people begin to use repetition as a cue for truth. In this preregistered study, we investigated those developmental origins in twenty-four 5-year-olds, twenty-four 10-year-olds, and 32 adults. If the link between repetition and truth is learned implicitly, then even 5-year-olds should show the effect. Alternatively, realizing this connection may require metacognition and intentional reflection, skills acquired later in development. Repetition increased truth judgments for all three age groups, and prior knowledge did not protect participants from the effects of repetition. These results suggest that the illusory-truth effect is a universal effect learned at a young age.
    1. Perchance he for whom this bell1 tolls may be so ill as that he knows not it tolls for him; and perchance I may think myself so much better than I am, as that they who are about me and see my state may have caused it to toll for me, and I know not that.

      Disasters may come silently, and we may not be prepared at all, but we must face it and find a way to solve it. Just like the COVID-19 pandemic this year, in January and February, no one could have imagined that the pandemic would become so severe in the next six months.

    2. when one man dies, one chapter is not torn out of the book, but translated into a better language; and every chapter must be so translated. God employs several translators; some pieces are translated by age, some by sickness, some by war, some by justice;

      I don't believe in Catholicism, but it is obvious that such as Buddhism, Islam, Catholicism, or religious ideas are closely related to our spiritual world no matter how they are used, and can even affect our survival during a pandemic. In our world, there are too many things we cannot explain with science and logic, there are too many things we can’t stop, like the author said, diseases, wars, and so on. Faced with these disasters, we may feel helpless and insignificant. Your religious faith could be your strongest backing. If God really exists who love us unconditionally and accept us, Whether you are guilty or not. People will feel very at ease in their hearts and have the courage to face the hardships of life, and even to face death. I don't think there is heaven or hell after death, but religion can teach those who believe it not to fear death and live with confidence and happiness. Imagine if you live in the era of the Black Death, your friends and relatives around you are dying every day, perhaps only the church bells can give you some consolation. Make you believe that, no matter what, you are not alone and the pain will always pass. This might not be a way to make life happier now.

    1. Equally though, some types of pornography, for some viewers, can be hugely empowering. It may reflect our identities and experiences, help us explore our sexuality, help us exercise sexual agency and bodily autonomy,

      I think pornography also has its pros, such as empowering both men and women. It helps us understand things that we like or dislike. It helps people explore their identities.

    1. I think in the United States where we have organic solidarity in the sense we are very complex and heterogeneous, individuals' moral preferences should not play any major role in determining criminal punishment. It is, in my opinion, unfair to allow for one person's moral views impact the punishment when myself or another may have a completely different moral viewpoint on the issue.

      I agree, the United States as a whole may be considered "organic" by Durkheim's conception of solidarity. However, within the United States, there are countless communities of "mechanical solidarity," such as small towns, local governments, schools, etc.. Regarding criminal justice or (perhaps more fitting) criminal injustice, there are federal standards which apply to the entire nation, which for some smaller communities may be too forgiving or too restricting (in this sense, federal laws may protect people from possibly harsher local laws). I understand your concern around the few creating policy for the many (and I completely agree!), but state and local governments (which are more "mechanical") possess power in creating local policies and in implementing policies.

    1. It seems that only the third angle of representation need be aperson: we can represent stones with dabs of paint or letters or sounds, but we can represent things only to people.

      I do not think this is necessarily true. Take a religious sacrament for example. A person may perform a ritual or wear a certain article of clothing as a symbol, or representation, of a feeling they have towards deity. Deity is not a person, yet it is the someone/something in which something/someone is being represented. I do not think this distinction really impacts the author's argument, but rather begs the question as to why the author felt it necessary to make this statement at all when it could be challenged and in no way really supports his overall point.

    1. This manuscript is in revision at eLife

      The decision letter after peer review, sent to the authors on June 12, 2020, follows.

      Summary

      All three reviewers agree that the research question under study, the requirement of the cross-talk between two important developmental signaling pathways - retinoic acid and the NO - for amphioxus pharynx development, is in principle interesting and could be suitable for publication in eLife.

      However, at present there are major open concerns especially on the lack of statistical analyses, quality of data presentation and inconsistencies with previously published work, that need to be addressed. Although it is the current policy of eLife to avoid additional experiments in revisions as much as possible, this is unfortunately likely impossible to fulfil with the current manuscript in order to bring it to a level that matches the standards of eLife. However, we think that in many cases an improvement of analyses and data presentation will likely already significantly improve the manuscript.

      1) The presented study is a follow-up on a previous paper by the same lab (Annona et al 2017 ; DOI:10.1038/s41598-017-08157-w). When comparing the work of this previous study with the current manuscript two major discrepancies are apparent:

      In Annona et al the two drugs were used to inhibit NOS production: L-NAME and TRIM, while only one inhibitor was used in the present study. Furthermore, there appear to be discrepancies concerning the developmental time windows during which chemical disruption of NO signaling is effective described in the two publications. This needs to be clarified.

      The timing of NosA,B,C expression, the suggested regulation of NosA and B by retinoic acid (RA) and the detected presumptive RARE regulatory elements in the genome don't match. More specifically, NosA,B,C expression at 24 hours (or around this time point) was investigated by Annona et al, 2017. Based on these data, NosA is not expressed during development, whereas NosB and NosC are expressed. In the submitted manuscript, the authors show that NosA and NosB are upregulated upon RA treatment, whereas NosC shows no changes in expression. They therefore suggest that RA regulates NosA and NosB transcription. Since only NosB is expressed during the relevant timepoints at early development, the transcription of this gene could be under the regulation of RA. However, when the authors look into the retinoic acid response elements (RARE) in the genomic region of NosB, they only find a DR3, which is not the typical RARE. They find DR1 and DR5 (apart from DR3's), which are more typical RARE's, in the genomic region of NosA, but as mentioned this gene is not expressed during development. This makes the hypothesis of a direct regulation of NosA and NosB by RA during normal development unconvincing. Can the author dissolve these apparent discrepancies?

      2) The authors study the open chomatin structure at 8, 15, 36 and 60 hours, thus time points, which do not overlap with the drug treatment period (24-30 hours). They need to analyze the genome architecture at this time period.

      3) The previous work by Annona et al 2017 et al shows that a major peak at NO levels occurs later than the chosen treatment window. How do NO levels during the time window of the experiment compare with other studies, i.e. is there evidence these are relevant levels? This is particularly noteworthy, as there is no control experiment showing that TRIM incubation affects NO levels or NO signaling during the incubation period (e.g. DAF-FM-DA staining or by NO quantification). It is therefore not possible to estimate the specificity of the resulting phenotypes.

      We thus request from the authors to provide ISH patterns of all the Nos genes, as well as NO localisation from at least 2 timepoints (e.g. start and end of window) of the TRIM application window.

      4) One overarching critique is that the general description of the figures and hence also the phenotypes are of poor quality. An improvement of this point will already majorly improve the entire manuscript.

      Fig.1A: Indicate developmental stages (N2, N4, T1, T2, T3, L0) together with the hours-post-fertilization (hpf) to facilitate the understanding of the treatment period with respect to the development of amphioxus.

      Fig.1B: Outline pharyngeal region e.g. with thin, dashed white lines in longitudinal and cross-sections and indicate relevant anatomical structures (club-shaped gland, endostyle, gill slits) e.g. with an arrow. Is the endostyle positioned more ventrally in TRIM treated larva?

      Figure 1C: why are Cyp26.3, Rdh11/12.18 and Crabp shown in triplicates?

      Fig.1B: The 'digital sectioning' method using confocal imaging and reconstruction of nuclear stainings is not suited to characterize the phenotype. Due to the loss of signal in deeper regions, morphological structures (e.g. differences in pharyngeal and gill slit morphology, endostyl, club-shaped glands) are impossible to recognize.

      Fig.3B: the heads of these amphioxus should be annotated to indicate key structures for non-amphioxus specialists. Ideally the images should be higher magnification and resolution as well, as the morphology is currently not very clear.

      Fig.3A and B: Furthermore, the morphological differences between 'altered', 'partially recovered' and 'recovered' is unclear. Fig.3B does not help understanding changes as the pictures are too small to recognize any morphological details without staining, and no structures are indicated. It is also unclear how animals scored as 'altered', 'partially recovered' and 'recovered' differ in their morphological structures. And does 'recovered' mean that these embryos show an initial phenotype that then 'recovers' during development, or do they show a completely normal development?

      5) Missing statistics/statistical information: Lines 85-89 (Fig.1): Where is the evidence that there is reduction in pharynx length? Where is the evidence for a smaller first gill slit? Measurements with a decent sample size and a basic statistical test must be provided.

      The description of ISH pictures in Fig.2A lacks any quantification and thus any information on the penetrance of the respective phenotypes are (as in Fig 3C). The lack of any 'negative control genes' (the large set of genes that, based on the RNASeq dataset, should not be affected) make it difficult to judge how specific changes in AP axis and RA pathway genes are.

      How did the authors obtain the qRT-PCR calculations? They need to clarify how they obtained the Fold changes shown in the histograms .e.g. by showing the maths behind the result when marking the cells in the excel sheet. The raw data for rpl32 is missing for Crabp in Figure 2B. The qPCR results in Fig.2B-E lack significance tests.

      6) The RNA-Seq study needs improvements: The PCA (Fig.S1C) shows no concordance among control samples or treated samples. Also, the histogram shows a clustering of replicates, and NOT of 'treated' and 'control' samples. This casts doubts on the quality and validity of the RNASeq dataset. These doubts are not removed by the current validation experiments, as these experiments tested only significantly upregulated genes by RNA-Seq, while downregulated and non-significant genes as 'controls' are missing. These additional controls are necessary to assess the validity of the RNA-Seq data.

      7) More information about the details of the ATACseq and ChIPseq data used, as well as the general RA responsive elements prediction is required.

      For example, in what amphioxus samples (and treatments if any) are these ATACseq and ChIPseq signals seen? There is some detail provided in the Methods section, but something is odd here and perhaps needs some further explanation. Since the two relevant Nos genes are supposedly not active during development then why do they have ATACseq and ChIPseq signals from embryo and larval samples? Why should these two Nos genes have apparently active regulatory elements focused on RAREs when the genes are not normally expressed under the control of RA, but only become active when exogenous RA is applied? We may well have missed something in the logic here, but this merely shows that the current level of explanation is insufficient.

      The analysis of RA responsive elements lacks statistical analysis and depth. It is left unclear how many RAREs would be expected by chance on a 52kb resp. 25kb locus. In addition, the authors include all ATAC-Seq peaks from stages ranging between 8h and 60hpf, while the window of RA responsiveness has been tightly restricted to the 24h-30hpf window. Also, as NosC expression levels stay constant upon RA incubation, it would be crucial to know if the NosC locus lacks any open RARE sites (as would be expected).

      The authors use NHR-SCAN tool to predict putative direct repeats binding sites in the genomic sequence of NosA and NosB. Which consensus sequence does the program follow? It appears that it does not follow the consensus sequence for typical RARE ((A/G)G(G/T)TCA), since the sequence for DR1 deviate from this sequence? DR1, DR2 and DR5 are the commonly described binding RARE's for the RAR/RXR heterodimers. Further, DR8 has been described as retinoic acid dependent regulation of gene transcription through RAR/RXR (Moutier et al., 2012). The authors need to provide clarification which are the most commonly used RARE's of the DR's detected.

      Please also mention if RAREs fall within an intron in the genomic regions of the Nos genes, since the transcriptional regulation through RARE is often associated to introns.

      8) Information on the concentration dependency of compounds used in the rescue experiment is lacking. Please explain why the BMS009 concentration used here (10exp-6 M) is 10x higher than the highest concentration used in the original publication on amphioxus pharynx development (Escriva et al., Development 2002).

      9) A summary drawing of the regulatory loop between NO and RA would be informative, also indicating the known target genes (from this study).

    1. This manuscript is in revision at eLife

      The decision letter after peer review, sent to the authors on May 25, 2020, follows.

      Summary

      This work addresses the key question how the herpesvirus HSV-1 reactivates from latency in neurons and shows that neuronal excitability plays a major role for controlling latency and reactivation. How hyperexcitability might influence the behavior of a latent, neurotrophic virus was previously unknown, and the authors show that neuronal hyperexcitability induces HSV reactivation in a DLK/JNK-dependent manner. In additon, the authors identify the cytokine IL-1b as a stimulus that triggers HSV reactivation in neurons, dependent on neuronal excitability, which is also a novel finding and of great interest for the field.

      Essential Revisions

      The reviewers all agree that your work about the potential link between IL1b, neuronal hyperexcitability, and HSV-1 reactivation is very interesting. However, we think that the three experiments listed below would be needed to substantiate the conclusion regarding the link between these three elements.

      1) To make sure that the results obtained with the inhibitors are not off-target effects, experiments with KO cells or siRNA knockdowns of DLK/JNK would strengthen the manuscript. Can you please specify in the manuscript the specific targets of the three inhibitors that were used - if they have discrete mechanisms of action, off-target effects may actually not be a problem. However, KO or knockdown experiments would validate the inhibitor results by an independent method and should be doable in these cultures.

      2) To substantiate the very interesting finding with IL1b, an experiment with a neutralizing IL1b antibody should be performed to unequivocally show that IL1b induces reactivation (this would exclude that impurities in the cytokine batch such as LPS activate the cells).

      3) To unequivocally show that IL1b induces reactivation through increasing neuronal hyperexcitability, calcium flux, which is induced by neuronal hyperexcitability, should be measured. A simple method to do this would be the use of Fura-2 AM or similar dyes. An advantage of this approach is that it could be measured what percentage of neurons are excited upon IL1b treatment and this could be correlated with the percentage of neurons that reactivate. This could also be performed in the presence of IL1b neutralizing antibodies to confirm that this cytokine induces neuronal hyperexcitability and HSV-1 reactivation.

      These three additional experiments would make the report more robust and elegantly correlate hyperexcitability of neurons with HSV-1 reactivation.

    1. It’s true that studies have found that readers given text on a screen do worse on recall and comprehension tests than readers given the same text on paper.

      Like I have previously said I find that I understand something so much more having it physically in my hands. But I never knew that studies have backed this up. I think this may be a problem we face in the future as digital books are becoming more and more common while many times physical books are not chosen.

    2. 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. But it is at the strategic level where the effects of this erosion of military ethics may be the most dangerous. To take one important example, Wong and Gerras were frequently told that the readiness assessments of partner forces in Iraq and Afghanistan were an example of ethical deception. These critically important assessments rated the ability of Iraqi and Afghan forces to fight on their own, without U.S. assistance. Yet these ratings usually depended more on the U.S. rotational unit deployment cycle than on the actual capabilities of those partner forces. In other words, partner units received low ratings when a new U.S. unit arrived, better ratings over time, and high ratings right before that unit left – only to plummet once again when a new unit arrived. This rollercoaster annual cycle would almost seem comical were it not for the fact that U.S. strategy in both Iraq and Afghanistan rested heavily on turning over the battlefield to these very same local national forces.

      Personally, I think this article is pretty absurd, there findings are no where near method based. They could not have possibly analyzed the fullest extent of research on the strategic or operational level. The tactical level explanation I thought was pretty standard which is typical coming from intellects. On this operational, but more so strategic interpretation, when I look at it they point to the U.S. military as carving out weak assessments for their allies and adversaries. I mean, I am sorry but the Afghan and Iraqi forces are just objectively horrible at their military tactics. If you read the book "Armies of Sand" by Kenneth Pollack we see that the Arab militaries are just inferior and that is nothing against them but they just do not have a superior command structure to the United States military. We are not in an ethical dilemma as most realist would state, I am more on the American Exceptionalism argument of this debate so this study is subpar and lacking.

    1. Thespecies that we rely on for food and shelter are a tiny proportion of total biodiversity, and most humans live in— and rely on — areas of only moderate biodiversity, not the Amazon or the Congo Basin

      But we also need to think about the porpotion of biodiveristy that depends on our tiny proportion of species that we rely on for food and shelter While we may not actively use areas of high biodiverity such as the amazon or congo for consturctuing high rise buildings and homes, we are instead still having a huge impact on the env through deforestation and industrialized agriultracal processes.

    1. I think we could freshen up the way we express central beliefs borne of experience about adolescent literacy with some selective appropriation. Such an orientation might play strongly with the general public and allow literacy scholars and practitioners to have a louder voice on literacy policy issues; it might also empower our literacy policy advisories while allowing us to lean deeper into the constructed and highly political science of reading. Given the ongoing and impressive advances in the life sciences, their productive and challenging impact on the social sciences and humanities, and their probable if belated impact on education theory, research, and practice, biological and ecological idioms and narratives may be an interesting and useful way to reframe adolescent literacy development.

      Topic: The author focuses on the relation between science and adolescent literacy. The influences that science has brought can advance the and contribute towards literacy, and education as a whole. Science had accomplished many achievements and I think the author wants to achieve the same thing for literacy. The author suggests a goal for science research to enhance and pave the way for reading in order to reframe its development.

    1. The view that literacy always involves social and cultural elements[

      I think this is important to understand when we consider varying levels of literacy among different groups of people. For example, if we were living in the mid-20th century, literacy discrepancies would be very great between states which have a long history of public education such as Massachusetts and others such as Tennessee which were only then fully adopting public education. These kind of discrepancies may seem bad to us today, but we must always consider the cultural context, Tennessee being an agricultural state primarily while New England was steeped in an educational tradition dating back to the Puritans with an economy of the kind necessitating such emphasis on education.

    1. How dreadful is the state of those that are daily and hourly in danger of this great wrath, and infinite misery! But this is the dismal case of every soul in this congregation, that has not been born again, however moral and strict, sober and religious they may otherwise be. Oh that you would consider it, whether you be young or old. There is reason to think, that there are many in this congregation now hearing this discourse, that will actually be the subjects of this very misery to all eternity. 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! How might all the rest of the congregation lift up a lamentable and bitter cry over him! But alas! instead of one, how many is it likely will remember this discourse in hell? And it would be a wonder if some that are now present, should not be in hell in a very short time, before this year is out. And it would be no wonder if some person that now sits here in some seat of this meeting house in health, and quiet and secure, should be there before tomorrow morning. Those of you that finally continue in a natural condition, that shall keep out of hell longest, will be there in a little time! your damnation don’t slumber; it will come swiftly, and in all probability very suddenly upon many of you. You have reason to wonder, that you are not already in hell. ‘Tis doubtless the case of some that heretofore you have seen and known, that never deserved hell more than you, and that heretofore appeared as likely to have been now alive as you: their case is past all hope; they are crying in extreme misery and perfect despair; but here you are in the land of the living, and in the house of God, and have an opportunity to obtain salvation. What would not those poor damned, hopeless souls give for one day’s such opportunity as you now enjoy!

      Jonathan Edwards is preaching to his congregation in a very vivid manner how they need to reflected on their lives and ways of living. He is trying to explain to them that giving yourself to God is the only way into salvation. Jonathan Edwards explains that if you are not born again and repent for your sins you will be going to hell. He explains that God's forgiveness of sins is the only reason they are not in hell at this moment. He explained that turning to God now and not later needs to be done before it is to late.

    2. How dreadful is the state of those that are daily and hourly in danger of this great wrath, and infinite misery! But this is the dismal case of every soul in this congregation, that has not been born again, however moral and strict, sober and religious they may otherwise be. Oh that you would consider it, whether you be young or old. There is reason to think, that there are many in this congregation now hearing this discourse, that will actually be the subjects of this very misery to all eternity. 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! How might all the rest of the congregation lift up a lamentable and bitter cry over him! But alas! instead of one, how many is it likely will remember this discourse in hell? And it would be a wonder if some that are now present, should not be in hell in a very short time, before this year is out. And it would be no wonder if some person that now sits here in some seat of this meeting house in health, and quiet and secure, should be there before tomorrow morning. Those of you that finally continue in a natural condition, that shall keep out of hell longest, will be there in a little time! your damnation don’t slumber; it will come swiftly, and in all probability very suddenly upon many of you. You have reason to wonder, that you are not already in hell. ‘Tis doubtless the case of some that heretofore you have seen and known, that never deserved hell more than you, and that heretofore appeared as likely to have been now alive as you: their case is past all hope; they are crying in extreme misery and perfect despair; but here you are in the land of the living, and in the house of God, and have an opportunity to obtain salvation. What would not those poor damned, hopeless souls give for one day’s such opportunity as you now enjoy!

      This is such a strong statement towards the congregation. Through this passage, the congregation could probably get a glimpse and feel it in their bones of the regret and eternal despair one might have to endure for eternity if they don't wake up from their slumbers and live a righteous life as God commands. Even the last sentence of those that have already been damned who would long for even a short moment and opportunity that the current people that are alive are currently enjoying really plants in fear of hell. Yet he goes on how many people still would be judged to hell.

    3. That world of misery, that lake of burning brimstone is extended abroad under you. There is the dreadful pit of the glowing flames of the wrath of God; there is hell’s wide gaping mouth open; and you have nothing to stand upon, nor anything to take hold of: there is nothing between you and hell but the air; ’tis only the power and mere pleasure of God that holds you up. You probably are not sensible of this; you find you are kept out of hell, but don’t see the hand of God in it, but look at other things, as the good state of your bodily constitution, your care of your own life, and the means you use for your own preservation. But indeed these things are nothing; if God should withdraw his hand, they would avail no more to keep you from falling, than the thin air to hold up a person that is suspended in it…. The God that holds you over the pit of hell, much as one holds a spider, or some loathsome insect, over the fire, abhors you, and is dreadfully provoked; his wrath towards you burns like fire; he looks upon you as worthy of nothing else, but to be cast into the fire; he is of purer eyes than to bear to have you in his sight; you are ten thousand times so abominable in his eyes as the most hateful venomous serpent is in ours. You have offended him infinitely more than ever a stubborn rebel did his prince: and yet ’tis nothing but his hand that holds you from falling into the fire every moment; ’tis to be ascribed to nothing else, that you did not go to hell the last night; that you was suffered to awake again in this world, after you closed your eyes to sleep: and there is no other reason to be given why you have not dropped into hell since you arose in the morning, but that God’s hand has held you up; there is no other reason to be given why you han’t gone to hell since you have sat here in the house of God, provoking his pure eyes by your sinful wicked manner of attending his solemn worship: yea, there is nothing else that is to be given as a reason why you don’t this very moment drop down into hell. O sinner! Consider the fearful danger you are in: ’tis a great furnace of wrath, a wide and bottomless pit, full of the fire of wrath, that you are held over in the hand of that God, whose wrath is provoked and incensed as much against you as against many of the damned in hell; you hang by a slender thread, with the flames of divine wrath flashing about it, and ready every moment to singe it, and burn it asunder; and you have no interest in any mediator, and nothing to lay hold of to save yourself, nothing to keep off the flames of wrath, nothing of your own, nothing that you ever have done, nothing that you can do, to induce God to spare you one moment… Consider this, you that are here present, that yet remain in an unregenerate state. That God will execute the fierceness of his anger, implies that he will inflict wrath without any pity… you will be a vessel of wrath fitted to destruction; and there will be no other use of this vessel but only to be filled full of wrath: God will be so far from pitying you when you cry to him, that ’tis said he will only laugh and mock (Proverbs 1:25-32)… How dreadful is the state of those that are daily and hourly in danger of this great wrath, and infinite misery! But this is the dismal case of every soul in this congregation, that has not been born again, however moral and strict, sober and religious they may otherwise be. Oh that you would consider it, whether you be young or old. There is reason to think, that there are many in this congregation now hearing this discourse, that will actually be the subjects of this very misery to all eternity. 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! How might all the rest of the congregation lift up a lamentable and bitter cry over him! But alas! instead of one, how many is it likely will remember this discourse in hell? And it would be a wonder if some that are now present, should not be in hell in a very short time, before this year is out. And it would be no wonder if some person that now sits here in some seat of this meeting house in health, and quiet and secure, should be there before tomorrow morning. Those of you that finally continue in a natural condition, that shall keep out of hell longest, will be there in a little time! your damnation don’t slumber; it will come swiftly, and in all probability very suddenly upon many of you. You have reason to wonder, that you are not already in hell. ‘Tis doubtless the case of some that heretofore you have seen and known, that never deserved hell more than you, and that heretofore appeared as likely to have been now alive as you: their case is past all hope; they are crying in extreme misery and perfect despair; but here you are in the land of the living, and in the house of God, and have an opportunity to obtain salvation. What would not those poor damned, hopeless souls give for one day’s such opportunity as you now enjoy! And now you have an extraordinary opportunity, a day wherein Christ has flung the door of mercy wide open, and stands in the door calling and crying with a loud voice to poor sinners; a day wherein many are flocking to him, and pressing into the kingdom of God; many are daily coming from the east, west, north and south; many that were very lately in the same miserable condition that you are in, are in now an happy state, with their hearts filled with love to him that has loved them and washed them from their sins in his own blood, and rejoicing in hope of the glory of God. How awful is it to be left behind at such a day! To see so many others feasting, while you are pining and perishing! To see so many rejoicing and singing for joy of heart, while you have cause to mourn for sorrow of heart, and howl for vexation of spirit! How can you rest one moment in such a condition? Are not your souls as precious as the souls of the people at Suffield,7 where they are flocking from day to day to Christ? … 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? And let everyone that is yet out of Christ, and hanging over the pit of hell, whether they be old men and women, or middle aged, or young people, or little children, now hearken to the loud calls of God’s Word and providence. This acceptable year of the Lord, that is a day of such great favor to some, will doubtless be a day of as remarkable vengeance to others… Therefore let everyone that is out of Christ, now awake and fly from the wrath to come. The wrath of almighty God is now undoubtedly hanging over great part of this congregation: let everyone fly out of Sodom. Haste and escape for your lives, look not behind you, escape to the mountain, lest you be consumed [Genesis 19:17].

      While I agree with the overall message, the actual delivery is much different than what we would encounter today. In today's society, we are encouraged to be politically correct and avoid offending others; sometimes, that is not the most effective. When matters are sugar-coated, there is room for people to find some margin to justify their wrong-doings. For example, how many times after you've done something wrong, i.e., frivolously spent money, told a small lie, etc. and later found yourself trying to justify it? Edwards is leaving little room for justification and explicitly details what will happen if people do not live by God's Word. Though fear was the prevailing tactic, the goal was to have as many conversions to Christianity as possible in a time where many were desensitized to the church and beginning to lean on their own understanding. A majority of the congregation was in tears during the sermon; this can be counted as a victory for Edwards. Their reactions show that members felt convicted, which in turn, may compel them to form or strengthen their relationship with God so that they may have salvation.

    4. 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.

    5. 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!

    6. 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. Reviewer #2:

      In this work cognitive assessment after isoflurane anaesthesia shows that several cognitive domains are impaired in speed of response and accuracy but dynamics of recovery are not the same for all domains. Specifically, tests related to executive functions recovered faster than others, against the authors' expectations.

      These results are important as they help to understand the dynamics of recovery of the cognitive systems after being challenged pharmacologically. The dynamics of a complex system (the brain) coming back to functioning in full is assessed both cognitively and neurally.

      I think this paper requires some clarifications, some more analyses and further discussion. One important result is the assessment of the dynamics of cognitive recovery after unconsciousness and its parallels with local and global complexity measures. As I was reading the paper I thought there would be a combined analyses to address the dependencies between complexity measured before, in unconsciousness and ROC to the behavioural outcomes. How does the level of complexity before even getting sedated or the complexity reached during unconsciousness influences the degree or speed of recovery? Please let me know if this sounds too post-hoc for you since it feels like an important and meaningful question to pose to the data for me.

      Am I correct in interpreting that you have calculated the LZC over the global topography? It would be important to clarify this point, differentiate from the other variant, and reflect that in the theoretical interpretation to avoid misunderstanding and subsequent unnecessary criticism. Two different variants of LZ complexity have been described: one that quantifies local, channel-wise complexity (LZS/LZSUM) and one that quantifies the complexity of the global topography of the scalp over time (LZC). These two variants appear to occasionally track different aspects of consciousness (Comsa 2018, thesis and Schartner et al., 2017). Specifically from Comsa's thesis "To compute the Lempel-Ziv complexity of EEG data, the concatenation of a signal consisting of channel values over time can be performed either channel-by-channel or observation-by-observation, where an observation consists of the values of all channels at a single point in time. The interpretation of the two complexity flavours is slightly different: the former case reflects the local, temporal signal diversity in individual channel values over time, whereas the latter captures the spatial diversity of the global landscape of neural activity. In some of the above studies, a different flavour appears to have worked best in different contexts: for example, the spatial variant in anaesthesia (Schartner et al., 2015), and the temporal variant in psychedelic states (Schartner et al., 2017). These different interpretations have not been thoroughly explored so far and it is not clear which variant best fits with the original theoretical framework that indicates neural information diversity as a key element for the emergence of consciousness".

      It would be a good idea to ask the question of no differences between cognitive scores before isoflurane and after several hours (three hours?), and compare to the control group in a statistically robust manner. If the aim is to claim full return-to-normal then a test to trust the no-difference would offer the answer. Please consider a statistical model that allows you to test the "return to normal" of cognitive capacities appropriately, maybe a Bayesian framework like the NLMM used but including some measure of the trust in the no-differences. It may be that the authors consider the CI values enough, in that case please express the results in terms of strength of these?

      I think a rerun of the stats asking for the effect size or bayes factor or any other parameter that would allow for an impression of the strength of the effect would go a long way in interpreting the results. Currently there seems to be a reliance on the p value (in the text), that does not reflect the strength of an effect.

      Further to this, supplementary material with the single subject dynamics of recovery would paint a true picture of the variance and variability of the results. We have gained great insight about the differential impact of sedatives in the last few years in the transition of consciousness. Here a couple of examples:

      https://www.pnas.org/content/110/12/E1142 https://www.sciencedirect.com/science/article/pii/S1053811920301142#bib68 and even one of our own https://journals.plos.org/ploscompbiol/article?id=10.1371/journal.pcbi.1004669

      In particular you might want to take a look at a recent reanalysis of our data of mild sedation by Bola and collaborators (https://www.biorxiv.org/content/10.1101/444281v2.full ) where they analyse the eeg using measures of diversity and complexity that are particularly relevant for the interpretation of your results.

      In the discussion there is the need for a section where the theoretical justification for the use of PE and LCZ. How is this better or complementary to power, connectivity and other measures used in EEG to discuss consciousness and sedation needs to be addressed so the readers get a more contextualised picture of why using these measures may shield better results, why they may be better for interpretation of the loss, maintenance and recovery of consciousness.

    1. Reviewer #2:

      Sherman et al seek to understand the basis of human time perception using a combination of psychophysics, computational modeling, and fMRI. This work builds on previously published work by the same group (Roseboom, Nature Communications 2019) showing that integrated changes in the state of (a) deep image classification network(s) during the presentation of movies predicted aspects of human timing reports. In that study, similar to what is shown in the current manuscript, timing biases were found in human behavior for different movie scene types, for example, city, natural scenes, or offices. Interestingly, similar biases were found in the timing estimates produced by their integrated deep network state change procedure. They interpret these findings as evidence that estimates of duration are derived from changes in the state of perceptual networks, in this case presumably those involved in visual perception. I find this previous work to be an important contribution toward understanding how the brain constructs information about a fundamental dimension of the environment for which there are no obvious sensors.

      In the current study, the authors repeat many of the steps contained in the previous publication, but in the context of humans estimating the duration of silent movies while positioned in an MRI scanner. They compute BOLD signals during movie viewing using a set of techniques I am not intimately familiar with because I do not use MR to assess brain activity in my own research, but which seem standard from what I can tell. They then treat the voxel by voxel BOLD measures similarly to the manner they did nodes in the deep network, and show that estimates derived from visual cortices may correlate with human biases and effects of scene type, but not those estimates derived from voxels in auditory or somatosensory cortices. While I have some technical questions, I find the work to be overall well reasoned and clearly presented. My major issue with the paper has to do with the fact that given their previous publication already showed that human behavior exhibits timing biases that correlate with the rate of change in visual scenes, and what we know about the localization of modality specific sensory function in cortex, it would be worrying if they could not derive time estimates from a measure of neural activity in visual cortex. It seems that the core hypothesis they are testing has to do with whether one can extract a measure of change in visual scenes from BOLD signals recorded in the visual cortex. Finding that one can indeed do so doesn't seem particularly surprising and thus represents a relatively incremental advance relative to what was known before. In terms of novelty, what we are left with then is the observation that the use of different metrics on BOLD changes per voxel to estimate elapsed time differ with respect to their ability to reproduce timing biases by scene type. However, clarification is needed regarding how they compute these metrics to fully assess the importance of these differences.

      The authors state that they compute Euclidian distance between voxel activations from TR to TR. However, it looks like they are computing the L1 norm of the differences, or the manhattan/city block distances. Which is it?

      Why should the sum of signed differences provide a different result? Is it that in the distance measurement, noise is accumulated in the measure over voxels whereas in the signed difference this noise is canceled out by averaging? Some amount of intuition would be helpful.

      Writing level comments:

      4) Regarding the framing and discussion of the experiments, I am not sure why the authors see their results as incompatible with and not complementary to some of the existing proposals for time encoding in the brain. For example, the impact of sensory change on responses in perceptual networks might very well have an influence on dynamics of downstream neural populations, potentially through neuromodulators, so I don't see the obvious incompatibility. This is not to say that the authors are not addressing an important problem, namely why does sensory change bias timing reports.

      For example, I think this statement is a bit inaccurate and unnecessary:

      "...This end-to-end account of time perception represents a significant advance over homuncular accounts that depend on "clocks" in the brain. "

      5) I wouldn't say their work represents an "end to end" account of time perception, and certainly not an end to end account of the behavior they are studying. What happens in more naturalistic situations where people are moving, and taking in other sensory modalities? How does this time perception information get transformed into the behavioral report of individuals, for example? The authors don't need to over-reach for the work to be interesting. The authors would also seem to be implying that the previously cited studies assume a specialized clock somewhere, where in fact Tsao et al and Soares et al at least are explicitly saying the opposite, and from my perspective the field views the idea of explicit "clocks" as a bit antiquated, and rather that timing is an emergent property of the functions that neural circuits are optimized to perform... an idea that seems compatible with the authors' work.

    1. Reviewer #3:

      This is the largest study of DNA methylation differences in the blood of controls and patients with psychosis, performed in a sample of 4,483 participants. As is predictable, the authors found significant differences in measures of blood cell proportions and smoking exposure in patients with psychosis compared with controls, and in patients with schizophrenia with clozapine treatment compared with other patients. They also detected differentially methylated positions in such comparisons. The authors have employed an appropriate methodology to search for schizophrenia- and psychosis- associated methylation changes, and the manuscript is interesting and well-written. However, I think a more extensive analysis may increase our insight about DNA methylation differences in schizophrenia, and is therefore necessary.

      1) An important question is whether the methylation differences are pre-existing the disorder or a consequence, an epiphenomenon of the disorder. The fact that the authors detect a higher number of DMPs when they exclude individuals with first episode psychosis from their analysis could suggest that the methylation differences are not present before the onset of the disorder. However, the authors have the resources and the ability to better answer this question. For example:

      1a) I think they should report in a separate section the results in the two samples of FEP individuals compared with age-matched controls. Can they identify any FEP-specific DMP?

      1b) Also, I think they could try to integrate their data with other blood methylation datasets, to see whether the DMPs associated with psychosis/schizophrenia have been associated with environmental risk factors associated with schizophrenia. For example, the authors could check the overlap of the DMPs with blood methylation changes associated with gestational age (PMID: 32114984; this work contains references to other studies that may be useful too). Data on methylation and cannabis or other environmental factors, if available, may be useful too.

      1c) The authors could also explore, in patients and controls, the relationship between age and methylation of the DMPs. An increase of the differences between patients and controls in older ages would suggest that the methylation differences are related to factors that are secondary to the disorders, while the presence of methylation differences at younger ages could suggest the opposite. Analyzing the interaction between methylation and age on case-control status could be an alternative way to answer this question.

      2) Sex is an important biological variable that the authors could analyze more extensively, considering that being male is a risk factor for schizophrenia, and is associated with a different epigenetic regulation. The authors have already the statistics to analyze whether the psychosis/schizophrenia-associated DMPs are also associated with sex. Moreover, they could analyze the interaction between methylation and sex on case-control status and/or perform analyses stratified by sex.

      3) The authors did not find association of schizophrenia with age acceleration. However, a recent study has performed a comprehensive analysis of 14 epigenetic clocks categorized according to what they were trained to predict: chronological age, mortality, mitotic divisions, or telomere length. I think it is relevant that the authors try to validate and perhaps extend the findings of Higgis-Chen and coll. ("Schizophrenia and Epigenetic Aging Biomarkers: Increased Mortality, Reduced Cancer Risk, and Unique Clozapine Effects", PMID: 32199607).

      4) Adjustment: I have not found any clear information about ethnicity/race. I assume the samples were mainly composed by white Caucasians. Did the authors perform any adjustment for ethnicity/race or population stratification? Also, were principal components of negative control probes included as covariates?

      5) Replication: was there any replication at the level of DMP in the data from Montano et al.? Also, if many DMPs are under genetic control, we should expect an overlap between DMPs in blood and brain of patients with schizophrenia. Have the authors analyzed such overlap?

      6) I think the authors should be more cautious in interpreting the clozapine data. They write: "Studies have also shown that higher neutrophil counts in schizophrenia patients correlate with a greater burden of positive symptoms (Núñez et al., 2019) suggesting that variations in the number of neutrophils is a potential marker of disease severity(Steiner et al., 2019). Our sub-analysis of treatment-resistant schizophrenia, which is associated with a higher number of positive symptoms (Bachmann et al., 2017), found that the increase in granulocytes was primary driven by those with the more severe phenotype, supporting this hypothesis." Actually, the fact that TRS cases are characterized by a significantly higher proportion of granulocytes could be related a "recruitment bias": because clozapine administration is associated with a risk of agranulocytosis, clozapine is usually not prescribed to patients with low number of granulocytes. I think this possibility needs to be mentioned, unless the authors can exclude it.

    1. The challenge was to think of behavioural implications of moving to a new, more shorter distance rule. Here is a short summary of points made, and questions generated that we do not have the research evidence for yet (perhaps a study on these would prove useful?)Issues raised:People may not accurately perceive distances (especially under different conditions)—if they underestimate, 2m has a buffer than 1m would notThe change from 2m to 1m could undermine compliance and rule-adherence (because the rule has changed)—especially if there are more changes made.1m is close to a (regular) socially-appropriate distance taking into consideration personal boundaries—as such, it could signal that everything is back to normal (but also, the distance varies depending on how close the contact is)The rule might be perceived as a 'normal' vs. 'not normal' conditionMedia discussion on this appears to be mostly based on the physical sciences—how far droplets can travel, and infection rates
    1. Black Joy & Accountability: As a future teacher, fighting for injustice may appear like a tall and overwhelming order. However, I am humbled and inspired to take part in the effort to create a better world. And I agree that there is joy to be gleaned from this effort. Analogously, teachers may be underpaid and undervalued and overworked, so I think one must find joy in the vocation until the educational survival complex is dismantled. So in the meantime, we must hold ourselves to a "level of accountability that focuses on justice, love, healing, and restoring humanity" (122).

    Annotators

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      Reply to the reviewers

      Reviewer 1

      __*Review 1 Summary:

      __In this manuscript, Borah et al showed that Heh2, a component of INM, can be co-purified with a specific subset of nucleoporins. They also found that disrupting interactions between Heh2 and NPC causes NPC clustering. Lastly, they showed that the knockout of Nup133, which does not physically interact with Heh2, causes the dissociation of Heh2 from NPCs. These findings led the authors to propose that Heh2 acts as a sensor of NPC assembly state. *

      __Reviewer 1 major comment 1:__ The authors claimed that Heh2 acts as a sensor of NPC assembly state, as evidenced by their finding that Heh2 fails to bind with NPCs in nup133 Δ cells (Fig2, Fig 5). However, there is a possibility that the association between Heh2 and NPCs is merely affected by the clustering of the NPCs (as the authors discussed) but not related to the structural integrity of NPC.

      • *

      Our Response: We agree that this is a possibility, however, we ask the reviewer to also consider that we artificially cluster NPCs using the anchor away system (Figure 3C) and this does not affect Heh2’s association with NPCs. Thus, clustering per se is insufficient to disrupt Heh2 binding to NPCs. We will also make changes in the text to make this point.

      • *

      Reviewer 1 major comment 2: In addition, their data showing that the Heh2-NPCs association is not easily disrupted by knocking out the individual components of the IRC (Fig. 5A and 5D), also disfavor the idea that Heh2 could sense NPC assembly state.

      Our Response: There are three considerations here. The first is that as this is the first evidence of any kind of “NPC assembly state” sensor, it is difficult to make any assumptions as to what specifically such a sensor would be monitoring. i.e. perhaps sensing only the ORC is what is functionally important. Second, for obvious reasons, we only tested non-essential IRC nups so by definition there is inherent functional redundancy that maintains NPC function and thus there may be no need to “sense” anything in the absence of these IRC nups. Further (and last), the IRC is essential for NPC assembly. Thus, without an IRC there is no NPC assembly state to sense.

      Reviewer 1 major comment 3: Since some nup knockout strains, other than nup133 Δ, are also known to show the NPC clustering (ex. nup159 (Gorsch JCB 1995) and nup120 (Aitchison JCB 1995; Heath JCB 1995)), it will be worth trying to monitor the localization of Heh2 and its interaction with nucleoporins (by Heh2-TAP) using these strains. While Nup159 is a member of the cytoplasmic complex, Nup120 is an ORC nucleoporin. Thus, biochemical and phenotypical analysis using these mutant cells will be useful to clarify if the striking phenotypes the authors found are specific to nup133 knockout strain (or ORC Nup knockouts) or could be commonly observed in the strains that show NPC clustering. Another interesting point is that Nup159 shows strong interaction with Heh2, even in nup133Δ cells. As the authors mentioned, Nup159-Heh2 interaction may not be sufficient for Heh2-NPC association, but it could be important for NPC clustering.

      Our Response: These are excellent points and we agree that there is a need to more thoroughly explore how NPC clustering driven by abrogating the function of other nups impacts Heh2’s association with NPCs. Thus, in a revised manuscript, we would examine Heh2’s association with NPCs in several additional genetic backgrounds where NPCs cluster.

      Reviewer 1 major comment 4: Figure 4C: Is it known that rapamycin treatment in this strain did not affect the protein levels of nucleoporins? Otherwise, the authors should confirm this by western blotting (at least some of them).

      Our Response: This is a good point and we will directly address this with Western blotting of some nups.

      Reviewer 1 major comment 5: Figure 5: The authors mentioned (line 256-257) that "in all cases the punctate, NPC-like distribution of Heh2-GFP was retained (Fig 5D)". However, nup107 KO strain seems to show more diminished punctate staining as compared with other strains. To clarify this, the authors should express mCherry tagged Nup as in Fig. 2 or Fig. 3.

      Our Response: Yes, we agree and in fact this observation is consistent with the fact that there is an ER-pool of Heh2 observed in this strain and we observe loss of nup interactions in the affinity purification. We will include a more thorough quantification of this in a revised manuscript and more directly address this in the text.

      **Minor comments:**

      Reviewer 1 minor comment 1: Figure 4A and 4B: The authors should show Scatter plot as in Fig. 2 and Fig. 3.

      • *

      We will include this in a revised manuscript.

      Reviewer 1 minor comment 2: Figure 5C: Explanations of the arrowheads is missing in the figure legend.

      Thank you for pointing this out, it will be fixed in a revised manuscript.

      Reviewer 1 minor comment 3: Figure 6: Is there any information as to where Heh2 (316-663) is localized in the cell?

      As this truncation lacks INM targeting sequences, it is found throughout the cortical ER. The determinants of Heh2 targeting (including truncations) has been extensively evaluated in King et al. 2006, Meinema et al., 2011 and Rempel et al. 2020. We will make this clearer in the revised manuscript.

      Reviewer 1 minor comment 4: Figure 6B: Nucleoporins should be marked with color circles as in Fig. 1 and Fig. 5.

      This will be done.

      Reviewer 2

      Borah et al. present a biochemical and cell biological examination of the inner nuclear membrane (INM) protein Heh2 and its putative interactions with the nuclear pore complex (NPC). The potential conceptual advance of this study is that Heh2 interacts with the NPC, while mutations believed to trigger NPC mis-assembly are shown to abolish interaction with Heh2, leading to the hypothesis that Heh2 is a sensor for NPC assembly states within the (INM). The conclusions would undoubtably be of broad interest to the nucleocytoplasmic transport field, but the evidence provided thus far is insufficient to build confidence and consequently this manuscript is premature for publication.

      Our Response: We thank the reviewer for recognizing the potential for a significant conceptual advance for the field but object to the notion that the work is “premature for publication”. This is a highly subjective statement that does not seem to meet the mission or purpose of the Review Commons platform. While it is possible that some of the conclusions drawn in our manuscript might not be fully supported by the data in its current form, there is a substantial body of work here that is certainly publishable.

      Reviewer 2 major comment 1: The TAP-tag Heh1/Heh2 pulldowns are the most significant experiment presented, and on face value provide compelling evidence that Heh2 interacts with the NPC. It is stated that mass spectroscopy (MS) was used to confirm the identities of the labeled bands yet there is no methods section, nor any MS data reported in the manuscript. Given the large number of unspecified proteins observed in these gels, and the single-step pulldown methodology used, knowledge of the contaminants present may aid in elucidating how Heh2 pulls down NPC components. Consequently, within the supplementary materials, the authors must indicate which regions of the gel were excised for MS analysis and provide a table listing all of the proteins that were detected for each sample, including the number of unique/expected peptides observed. Our Response: This was a major oversight on our part and a revised manuscript will contain all relevant details with regards to the MS analysis including a more detailed description of the excised bands and the quantification of spectra derived from these bands.

      Reviewer 2 major comment 2a: The representative micrographs provided across Figures 2, 3, 4, 5 and 6 are very noisy. Particularly in the case of the mCherry labeled nucleoporins, this is both unusual and unfortunate given this is used to infer colocalization of Heh2 with the NPC.

      Our Response: These micrographs are not unusual and are in fact of respectable quality. We agree that the apparent “noise” is unfortunate, but this is simply a reality of the yeast system. We remind the reviewer that there are only ~100 to ~200 NPCs per budding yeast nucleus, which is an order of magnitude smaller than a typical mammalian cell nucleus. Further, the copy number of yeast nups per NPC is half of the mammalian cell NPC. Further, budding yeast are spherical with a cell wall that is extremely effective at scattering light; they are also highly autofluorescent (particularly in the red channel). Lastly, unlike in mammalian cells, budding yeast NPCs are mobile on the nuclear envelope. Thus, co-localization is challenging (particularly with the long exposures required to obtain good images). This is why clustering of NPCs driven by nup133**∆ cells has provided one of the key assays in the field to assess whether a given protein associates with NPCs at the level of light microscopy.

      Reviewer 2 major comment 2b: As a result it is unclear whether this experiment can be used to differentiate between NPC colocalization vs. nuclear envelope colocalization.

      Our Response: The reviewer is correct. Co-localization between Heh2-GFP and any Nup-mCherry is insufficient to assess NPC association in WT cells. In fact, as we point out in Figure 3B, at best one can expect a correlation of r = 0.48 for two well established nups. Thus, to further support the conclusion that Heh2 associates with NPCs, we established the Nsp1-FRB NPC clustering assay (Figure 3).

      Reviewer 2 major comment 2c: The authors should include negative controls for an alternative NE membrane protein that doesn't bind the NPC, which would be expected to exhibit a reduced level of colocalization with NPC proteins when compared to Heh2. For example, Heh1 would be a suitable, given the clear-cut negative pulldown data and its prior usage as a negative control in Figure 4.

      • *

      Our Response: This is included in Figure 3D.

      Reviewer 2 major comment 3a. Figure 2. The rim staining for the Nup82-mCherry in the WT background is unusually punctate, bringing into question the viability of the cells imaged.

      Our Response: As the middle cell in the panel is undergoing cell division, these cells are clearly viable. All our imaging is performed on mid-log phase cultures.

      • *

      Reviewer 2 major comment 3b. Why has ScNup82, a cytoplasmic filament component, been selected for colocalization experiments when Heh2 is proposed to interact with the inner ring complex?

      Our Response: The resolution of a conventional light microscope is, at best, 200 nm in x, y. As NPCs are 100 nm in diameter, even two NPCs side-by-side cannot be resolved. The IRC is tens of nm away from the cytoplasmic filaments thus any nup is relevant for a co-localization analysis with a light microscope.

      Reviewer 2 major comment 3c: Additionally, the experiments shown in panels A and C are not directly comparable, ScNup82 is an asymmetric cytoplasmic nucleoporin, while SpNup107 is located in the Y-shaped Nup84 nucleoporin complex and present on both faces of the NPC. This experiment should be repeated with scNup84 to match panel C, additionally a viability dot spot assay and western blot analysis of the labeled proteins should be conducted.

      Our response: These are in fact directly comparable within the limits of resolution of light microscopy as described above. Viability assays are not required here as both nups are essential and perturbation to their function would lead to inviability.

      Reviewer 2 major comment 4: Figure 3, the authors use yeast strains where proteins are tagged with FRB and FKBP12 domains, which dimerize upon the addition of rapamycin inducing NPC clusters. The authors then observe the effect this has on Heh2 NPC colocalization. However, Rapamycin may also have an effect independent from the induced dimerization event. Negative controls should be performed in strains lacking the FRB and FKBP12 tagged proteins to demonstrate that Rapamycin doesn't modify Heh2 localization independently of NPC clustering.

      Our response: This is a good point and important control that we performed in prior studies, see Colombi et al., JCB, 2013. We will be more explicit in describing that this control has been done.

      Reviewer 2 major comment 5: Figure 4. The authors provide a qualitative description of the colocalization presented, while in all other instances they calculate a Pearson correlation coefficient. This is significant because Heh2 appears to be evenly distributed within the NE of the DMSO control (panel B). Given the presented hypothesis isn't colocalization expected with Nup192? As a minimum, a Pearson correlation coefficient analysis should be conducted and added to Figure 4.

      Our response: This will be included in a revised manuscript.

      Reviewer 2 major comment 6: Figure 4. Pom152-mCherry localizes at both the NE and strongly within the cytoplasm, which is unexpected given typical rim staining phenotypes observed previously for both Pom152-YFP and Pom152-GFP strains (Katta, ..., Jaspersen et al., Genetics (2015) & Upla, ..., Fernandez-Martinez et al., Structure (2017), respectively). Given the unusually weak rim staining observed throughout, viability assays of the strains listed in Table S1 and protein expression analysis of the tagged nucleoporins via western blot is necessary.

      Our response: This is not localization in the cytoplasm but is in fact autofluorescence from the yeast vacuole. We regret we were not more explicit in describing this and we will make the manuscript more accessible for the non yeast expert. In order to perform the Western blot analysis for all strains requested by the reviewer would require a battery of antibodies to the endogenous proteins to directly assess how tagging influences nup levels, which we do not have (nor does anyone else that we are aware of). This is also not standard practice in the field as it is an onerous and unnecessary burden.

      Reviewer 2 major comment 7:* Figure 5A. The TAP-tagged pulldowns from ∆Pom152 and ∆Nup133 strains appear to be from a different round of experiments than the previous deletion strains presented. Interestingly, there appears to be an additional band at approximately 250 kDa in both cases that is not present in any other experiments. This band could be a contaminant observed due to different experimental conditions, or a protein that exclusively binds to Heh2 in the ∆Pom152 and ∆Nup133 background. Either way the authors should identify this protein with MS to address this ambiguity.

      *

      Our response: We will include negative controls for these specific experiments to show that this is a non specific band.

      Reviewer 2 major comment 8: Figure 6B. Please label the nucleoporin bands in the TAP-tagged pulldowns.

      Our response: This will be done.

      Reviewer 2 major comment 9: Figure 6D. Please specify Heh2-GFP clustering in the y-axis.

      Our response: As this represents both Heh2-GFP and heh2-1-570-GFP, we will keep it as is to avoid confusion.

      Reviewer 2 major comment 10: *Under the results section titled 'Heh2 binds to specific nups in evolutionarily distant yeasts', the authors state that spHeh2 co-purifies with "several specific species". The meaning is unclear, this sentence should be rephrased and the specific species clearly described. **

      *

      Our response: Ok.

      Reviewer 2 major comment 11: Under the results section titled 'Heh2 fails to interact with NPCs lacking Nup133', the authors refer to a Pearson correlation coefficient of -0.03 as a clear anticorrelation. Instead state there was no correlation.

      Our response: Ok.

      Reviewer 2 major comment 12: In the discussion, the authors state that "clustering itself may sterically preclude an interaction with Heh2". The text should be expanded to explain this in more detail, it is not clear from the presented data why this would occur.

      Our response: Ok.

      Reviewer 2 comment on significance: the manuscript is premature for publication.

      Our Response: Such a statement has no relevance to this form of review as a decision as to whether a study is premature for publication should be made by journal editors, not reviewers. We would argue quite strongly that we have definitively shown that Heh2 binds to NPCs, that it does so in multiple evolutionarily distant yeasts and that this binding is functionally relevant. For example, we can specifically disrupt the association of Heh2 with NPCs with a specific domain deletion and observe a loss of function phenotype (e.g. NPC clustering). What all three reviewers agree on is that the concept of a “NPC assembly state sensor” needs additional data to be fully supported, although we note that this reviewer did not provide any suggestions for how we might achieve this goal. We further note that we added the qualifier “may” into the title of the work. Thus, we will therefore perform additional experiments as outlined in comments to Reviewer 1 to support this conclusion in order to introduce this as a new concept in the field.

      Reviewer Comment from Cross Commenting: It seems to me that all reviewers agree that the manuscript is premature for publication. The data thus far do not support the conclusion that Heh2 may be an NPC assembly sensor nor does it provide any mechanistic insight. Reading the comments of the other two reviewers makes me more negative, as it is care that the paper also lacks scientific rigor. The manuscript is a great starting point for a rigorous dissection but I do not see this paper to be a candidate for a broad impact journal.

      Our Response: The statement that this manuscript is premature for publication is an opinion and does not seem to reflect the sentiment of the other reviewers. It is also confounding that this reviewer suggests that this work lacks rigor. With the exception of the omission of the MS analysis (our fault), the data are of high quality and rigorously quantified. Our assertion of rigor and data quality is based on our collective team’s many decades-long history of publishing and reviewing papers at the highest levels in this field. Questions as to the quality of the data as stated by this reviewer (and only this reviewer) in fact address limitations of light microscopy and the yeast system more generally in this one respect.


      Reviewer 3

      Reviewer 3 Summary part a*: This is quite an interesting manuscript that explores the relationship between an INM protein, Heh2, and NPCs. It represents an extension of earlier work performed by this group in which it was shown that the HEH2 gene shares genetic interactions with the genes encoding various nucleoporins. Heh2 belongs to an intriguing family of conserved proteins that includes its orthologue, Heh1, as well as human MAN1 (LEMD3) and LEMD2, among others. Each of these proteins contains two transmembrane domains with the N- and C-terminal regions extending in to the nucleoplasm. The two TM domains are separated by a short lumenal loop.

      In this study, the authors show that a population of Heh2 is associated with Nups of the NPC inner ring complex. This was demonstrated initially in pulldown experiments. The authors go on to show that when NPCs are caused to aggregate, by physical tethering employing an FKBP/FRP system in combination with Rapamycin, Heh2, but not Heh1, colocalizes with the NPC clusters. *

      • *

      Our Response: Thank you to the reviewer for recognizing the value of this work.

      • *

      Reviewer 3 Summary_b. Although not stated explicitly in the manuscript, this would imply that there is a population of Heh2 that resides in the NPC membrane domain, with the remainder in the INM. As an idle question, is there any evidence for a similar localization of MAN1 or LEMD2 in mammals? I am guessing probably not.

      Our Response: We regret this was not made more clear but the idea that there is a pool of Heh2 at the POM and a pool at the INM is an important conclusion of the work and was stated in the results - we’ll re-emphasize in the revised discussion. As to whether MAN1 or LEMD2 has a similar NPC association, we hypothesize that MAN1 but not LEMD2 will indeed interact with NPCs in mammalian cells. This is based on considering that we show that both the budding and fission yeast orthologues of MAN1 share this association so unless it was lost in evolution, this is a likely outcome of future studies.

      Reviewer 3 Significance statement a: The complications arise when the authors show that an alternative method of NPC aggregation (although they did this first), involving Nup133 deletion, results in failure of Heh2 to co-aggregate. In other words, Nup133 is required for the association of Heh2 with NPCs. The issue here is that there is no evidence for an interaction between Heh2 and Nup133, and furthermore that loss of Nup133 (a Y complex component of the outer ring complex) leaves the inner ring complex intact.

      • *

      Our Response: We tested the nup133Δ background first as this is the standard approach for assessing NPC-association of a given protein so we felt this would be logical for a reader in the field. Further, while the disruption of Heh2’s binding by loss of Nup133 may be a complication, we prefer to see it as an opportunity for discovery. As described in our manuscript, we have chosen to interpret this result in the context of a new biological function/concept with Heh2 being a novel “NPC assembly state” sensor. While one could argue that we have not fully met this bar yet, we will perform additional experiments as outlined in our response to reviewer 1 to help support this compelling conclusion.

      • *

      Reviewer 3 Signfiicance statement b: What is clear, however, is that Heh2 seems to be required to inhibit NPC aggregation since Heh2 deficient cells exhibit NPC clusters. The association between Heh2 and IRC Nups resides in the C-terminal nucleoplasmic winged helix domain. The N-terminal domain, in contrast confers INM localization.

      • *

      Our Response: We agree.__*


      Reviewer 3 Signfiicance statement c I must admit, I am in two minds about this manuscript. The data clearly show that Heh2 is associated with IRC components and I agree with the authors that this protein may well have a role in NPC assembly quality control perhaps in the guise of a chaperone. However, I find it hard to come up with a convincing model for the effects of Nup133. On the one hand, one could make an argument that the data presented here is too preliminary and fails to provide a complete story. On the other hand, it does provide an intriguing foundation for future studies and I do feel positively disposed towards it. In short, I have no fundamental complaints about the science, I am just uncertain as to whether the study is ready for publication.

      Our Response: This statement nicely articulates the challenge with this manuscript as there are some solid findings (that Heh2 binds specifically to NPCs etc.) but also a provocative finding (that loss of Nup133 breaks Heh2’s interaction with NPCs despite not physically interacting). Thus, there is a decision to be made about whether there is value in introducing a novel concept to the field once additional data is provided in a revised manuscript.

      Reviewer 3 Cross commenting: I have no fundamental disagreements with either of the other two reviewers. The comment from Reviewer#2 summarises this quite neatly. While I have fewer concerns about the quality of the data as presented, I think we all agree that at best the study is preliminary. What the authors need to do is to construct a coherent model that will account for the observations described here and then to design experiments that will test this model. I'm not suggesting that they must have a complete story, but they do need to go beyond what is in the current manuscript.

      • *

      Our Response: We appreciate that the reviewer does not have any questions about the quality of our data, but we argue that we have in fact presented the most coherent interpretation of the data as it currently stands. As described above, we intend to attempt to solidify this model by performing experiments suggested by reviewer 1.



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      The typical domed appearance of a hydrocephalus-harboring skull is apparent as early as P4, as shown in a new side-by-side comparison of pups at that age (Fig. 1A). Though this is not stated in the MS

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      Referee #3

      Evidence, reproducibility and clarity

      This is quite an interesting manuscript that explores the relationship between an INM protein, Heh2, and NPCs. It represents an extension of earlier work performed by this group in which it was shown that the HEH2 gene shares genetic interactions with the genes encoding various nucleoporins. Heh2 belongs to an intriguing family of conserved proteins that includes its orthologue, Heh1, as well as human MAN1 (LEMD3) and LEMD2, among others. Each of these proteins contains two transmembrane domains with the N- and C-terminal regions extending in to the nucleoplasm. The two TM domains are separated by a short lumenal loop.

      In this study, the authors show that a population of Heh2 is associated with Nups of the NPC inner ring complex. This was demonstrated initially in pulldown experiments. The authors go on to show that when NPCs are caused to aggregate, by physical tethering employing an FKBP/FRP system in combination with Rapamycin, Heh2, but not Heh1, colocalizes with the NPC clusters. Although not stated explicitly in the manuscript, this would imply that there is a population of Heh2 that resides in the NPC membrane domain, with the remainder in the INM. As an idle question, is there any evidence for a similar localization of MAN1 or LEMD2 in mammals? I am guessing probably not.

      Significance

      The complications arise when the authors show that an alternative method of NPC aggregation (although they did this first), involving Nup133 deletion, results in failure of Heh2 to co-aggregate. In other words, Nup133 is required for the association of Heh2 with NPCs. The issue here is that there is no evidence for an interaction between Heh2 and Nup133, and furthermore that loss of Nup133 (a Y complex component of the outer ring complex) leaves the inner ring complex intact. What is clear, however, is that Heh2 seems to be required to inhibit NPC aggregation since Heh2 deficient cells exhibit NPC clusters. The association between Heh2 and IRC Nups resides in the C-terminal nucleoplasmic winged helix domain. The N-terminal domain, in contrast confers INM localization.

      I must admit, I am in two minds about this manuscript. The data clearly show that Heh2 is associated with IRC components and I agree with the authors that this protein may well have a role in NPC assembly quality control perhaps in the guise of a chaperone. However, I find it hard to come up with a convincing model for the effects of Nup133. On the one hand, one could make an argument that the data presented here is too preliminary and fails to provide a complete story. On the other hand, it does provide an intriguing foundation for future studies and I do feel positively disposed towards it. In short, I have no fundamental complaints about the science, I am just uncertain as to whether the study is ready for publication.

      REFEREES CROSS COMMENTING

      I have no fundamental disagreements with either of the other two reviewers. The comment from Reviewer#2 summarises this quite neatly. While I have fewer concerns about the quality of the data as presented, I think we all agree that at best the study is preliminary. What the authors need to do is to construct a coherent model that will account for the observations described here and then to design experiments that will test this model. I'm not suggesting that they must have a complete story, but they do need to go beyond what is in the current manuscript.

    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.

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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. 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. 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.

  3. 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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  4. 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

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    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.

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      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.

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      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.

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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:

      Summary:

      In this paper, the authors utilize CRISPR-Cas9 to generate two different DMD cell lines. The first is a DMD human myoblast cell line that lacks exon 52 within the dystrophin gene. The second is a DMD patient cell line that is missing miRNA binding sites within the regulatory regions of the utrophin gene, resulting in increased utrophin expression. Then, the authors proceeded to test antisense oligonucleotides and utrophin up-regulators in these cell lines.

      Overall opinion (expanded in more detail below).

      The paper suffers from the following weaknesses:

      1) The protocol used to generate the myoblast cell lines is rather inefficient and is not new.

      2) Many of the data figures are of low quality and are missing proper controls (detailed in points 5,7,10, 12, 13,14)

      Detailed critiques:

      1) The title needs to be changed. The method used by the authors is inefficient. The title should instead focus on the two cell lines generated.

      We appreciate the reviewer’s comments: thanks to them, we have realized the focus of the manuscript should be in the new models we described and less in the methodology used to create them.

      Originally, we wanted to share the problems we faced when applying new CRISPR/Cas9 edition techniques to myoblasts: our conversations with other researchers in the field confirmed that many were having similar problems. However, the reviewer is right in the fact that there are many ways around this problem. We do describe ours and we are working in a new version of the manuscript with additional data to characterize our new models further and where the method used to create them, although included, is not the main focus of the manuscript. In this new version we will change the title accordingly.

      2) Line 104: The authors declare that the efficiency of CRISPR/Cas9 is currently too low to provide therapeutic benefit for DMD in vivo. There are lots of papers that show efficient recovery of dystrophin in small and large animals following CRISPR/Cas9 therapy. The authors should cite them properly.

      Thank you for your appreciation. We have reviewed the literature again to include new evidences of efficient dystrophin recovery as well as other studies with lower efficiency.

      3) Figures 1, 2,3, and 4 can be merged into one figure.

      4) Figure 2A and 2B can be moved to supplementary.

      5) Figure 2C and 2D are not clear. Are the duplicates the same? Please invert the black and white colors of the blots.

      Thank you for your comments. We have inverted the colors of the blots and changed the marks used in figure 2C and 2D to clarify that duplicates are indeed the same sample, assayed in duplicates. We have also merged figures 1 and 4 and moved figures 2 and 3 to supplementary in this new version.

      6) Figure 3: In order to optimize the efficiency of myoblast transfection, the plasmids containing the Cas9 and the sgRNA should have different fluorophores (GFP and mCherry). This approach would increase the percentage of positive edited clones among the clones sorted.

      We think the reviewer may have misunderstood our methodology: we are not using a plasmid with the Cas9 and another with the sgRNA, we are using two plasmids, both containing Cas9 and each a different sgRNA. We did try to use two different plasmids, one expressing GFP and one expressing puromycin resistance, but we found out that single GFP positive cell selection plus puromycin selection was too inefficient. We could have tried with two different fluorophores, but we tested the tools we had in our hands first and were successful at obtaining enough clones to continue with their characterization, so we did so instead of a further optimization to our editing protocol.

      7) Figure 4A: In the text, the authors state that only 1 clone had the correct genomic edit, but from the PCR genotyping in this figure shows at least 2 positive clones (number 4 and 7).

      Thank you for your appreciation. As you said, we got two positive clones (as we also indicate in figure 3B) but we completed the full characterization of one of them (clone number 7= DMD-UTRN-Model). In the new version of the manuscript we explain this further.

      8) Figure 4C: The authors should address whether one or both copies of the UTRN gene was edited in their clones.

      Thank you for your comment. Both copies of the UTRN gene were edited in our clones. We have included this information both in the text and in the figure 4 legend.

      9) Figure 4 B and D: The authors should report the sequence below the electropherograms.

      Thank you for this correction, we have included the sequence under the electropherograms.

      10) Figure 5B: This western blot is of poor quality. Also, the authors should specify that the samples are differentiated myoblasts. Lastly, a standard protein should be included as a loading control.

      Thank you for your comment. Poor quality of dystrophin and utrophin western blots was the main reason to validate a new method in our laboratory to measure these proteins directly in cell culture (1) like an alternative to western blotting. Since then, the myoblot method has been routinely used by us and in collaboration with other groups and companies. We included the western blot as it is sometimes easier for those used to this technique to be able to assess a blot in which there is no dystrophin expression. As you pointed out, our samples were all differentiated myotubes, not myoblasts, and we have modified this accordingly. Thank you very much for pointing out this mistake

      On the other hand, as described in the methods, Revert TM 700 Total Protein Stain (Li-Cor) and alpha-actinin were included as standards in dystrophin and utrophin western blots, respectively.

      11) Figure 5E: We would like to see triplicates for the level of Utrophin expression.

      We thank the reviewer for his/her recommendation, but we do not consider western blotting a good quantitative technique, we have included western blots to show the expression/absence of protein at the same level. We have included many more replicates than needed to show at the level of utrophin by myoblots. We acknowledge that western blotting is the preferred method for some reviewers, so in the new version of our manuscript we clearly indicate the value we give to each technique, being myoblots our choice for quantification.

      12) Figure 6: A dystrophin western blot should be included to demonstrate protein recovery following antisense oligonucleotide treatment. Also, the RT-PCR data could be biased as you can have preferential amplification of shorter fragments.

      Thank you for your recommendation but as we have explained before, myoblots have been validated in our laboratory to replace western blot for accurate dystrophin quantification in cell culture.

      13) Figure 6A: Invert the black and white colors. The authors should also report the control sequences and sequences of the clones under the electropherograms.

      Thank you for your suggestion, we have inverted the colors and added the sequences under the electropherograms.

      14) Figure 6B: Control myoblasts should be included in figure 5C.

      Thank you for this correction, we will include control myoblasts in the new manuscript version.

      15) Figure S2A: Invert the black and white colors.

      Thank you for your suggestion, we have inverted the colors.

      Reviewer #2:

      The work from Soblechero-Martín et al reports the generation of a human DMD line deleted for exon 52 using CRISPR technology. In addition, the authors introduced a second mutation that leads to upregulation of utrophin, a protein similar to dystrophin, which has been considered as a therapeutic surrogate. The authors provide a careful description of the methodology used to generate the new cell line and have conducted meticulous evaluations to test the validity of the reagents.

      However, if the main purpose of this cell line is to perform drug or small molecule compound screenings, a single line might not be sufficient to draw robust conclusions. The generation of additional DMD lines in different genetic backgrounds using the reagents developed in this study will strengthen the work and will be of interest to the DMD field.

      Thank you for your appreciation. We think that a well characterized immortalized culture, like the one we describe is sufficient for compound screening, as described in other recently published studies (2), (3). About the other suggestion, we have indeed used our method to generate other cultures for collaborators, but they will be reported in their own publications, as they are interested in them as tools in their own research projects.

      Further, the future use of the edited DMD line with upregulated utrophin is unclear. The utrophin upregulation adds a complexity to this line that might complicate the assessment of screened compounds. In contrast, this line could be used to test if overexpression of utrophin generates myotubes that produce increased force compared to the control DMD line.

      We think we may have not explained our screening platform well enough. Our suggestion is to offer our newly generated culture ALONGSIDE the original unedited culture: the original is treated with potential drug candidates, while the new one may or may not be treated, if these drug candidates are thought to act by activating the edited region (see an example in the figure below). In this case, the new culture will be a reliable positive control to the effects that may be reported in the unedited cultures by the drug candidates. We will make this clear in the new version of the manuscript.

      Created with BioRender.com

      In summary, while there is support and enthusiasm for the techniques and methodological approach of the study, the future use of this single line might be dubious and could be strengthened if additional lines are generated.

      We share the reviewer’s enthusiasm for this approach, and we have included in the new version of the manuscript further characterization of this new cell culture that we think would demonstrate its usefulness better.

    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

  5. 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.
  6. 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.

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      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.*


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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 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.

  7. 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. At the point when your organization needs new ability, the initial step is to begin selecting exceptionally qualified possibility for the activity. At the point when you realize how to isolate the quality goods from the refuse, it’s simpler to locate the ideal contender for your organization. Sadly, this can now and then be actually quite difficult. By what method can your specialty prevail with regards to finding the most ideal contender for your organization’s vacant positions? Here’s your bit by bit control. You can push all the extraordinary sets of expectations you need, it despite everything won’t get your image the perfect applicant except if your area of expertise knows how to scout. While trusting that up-and-comers will apply is detached, scouting is a functioning procedure that gives your group a superior possibility of finding the ideal contender for your activity. 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.

    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 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.

    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 #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.

  8. 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.
  9. 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.

  10. 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.

  11. 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.
  12. inst-fs-iad-prod.inscloudgate.net inst-fs-iad-prod.inscloudgate.net
    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.