7 Matching Annotations
  1. Last 7 days
    1. Mutations in the human retinal degeneration slow (RDS) gene can cause either retinitis pigmentosa or macular dystrophy

      PMID: 8485576

      Gene: PRPH2

      HGNC: 9942

      Disease: retinitis pigmentosa 7

      MONDO: 0011974

      InheritancePattern: semi-dominant

      Prevalence: The incidence of PRPH2-related cases among inherited retinal dystrophies is reported to range from 3.5% in North America to 10.3% in France (PMID: 36010202).

      Penetrance: The penetrance among biallelic cases appears to be complete, while the monoallelic cases exhibit incomplete penetrance (PMID: 36609934).

    1. mutation

      Case#: Meins_1993_Proband, male, age 58 years at time of report, national origin reported as German

      CasePresentingHPOs: Nyctalopia HP:0000662, Reduced visual acuity HP:0007663, Peripheral visual field loss HP:0007994, Central scotoma HP:0000603, Decreased light- and dark-adapted electroretinogram amplitude HP:0000654, Decreased Arden ratio of electrooculogram HP:0008179, Bone spicule pigmentation of the retina HP:0007737, Macular flecks HP:0011507

      CaseHPOFreeText: The proband has been diagnosed with retinitis pigmentosa, following a previous diagnosis of Stargardt's macular dystrophy almost 1 decade earlier. First onset of night vision loss was reportedly in his teens.

      CaseNotHPOs: N/A

      CaseNotHPOFreeText: N/A

      CasePreviousTesting: Previous testing in the proband is not described but genotyping appears to have been limited to the PRPH2 locus and did not include other candidate genes.

      CaseMethod1: single-strand conformational polymorphism (SSCP) analysis of PCR products from the 3 exons of PRPH2

      CaseMethod2: Sanger sequencing of PRPH2

      CaseGenotypingMethod: Previous testing in the proband is not described but genotyping appears to have been limited to the PRPH2 locus and did not include other candidate genes. Genotyping was performed by SSCP analysis of PCR products from the 3 exons of PRPH2 and Sanger sequencing of PRPH2.

      Variant: The proband harbors the NM_000322.5(PRPH2):c.136C>T (p.Arg46Ter) variant in the heterozygous state.

      ClinVar ID: 13179

      gnomAD: The NM_000322.5(PRPH2):c.136C>T (p.Arg46Ter) variant is found in gnomAD v4.0.0 at a frequency of 0.00004005 (3 alleles / 74906 total alleles, with 0 homozygotes) in the African / African-American population https://gnomad.broadinstitute.org/variant/6-42722199-G-A?dataset=gnomad_r4

      VariantEvidence: NM_000322.5(PRPH2):c.136C>T (p.Arg46Ter) is a nonsense variant in exon 1 of 3 that is predicted to trigger nonsense-mediated decay or at least to truncate the C-terminal 87% of the protein product.

      CaseAddInfo: The proband has a similarly affected father and grandfather who have not been genotyped.

      CasePMIDs: N/A

    1. separate families

      FamilyLabel: Kajiwara_1991_Family_6935, family national origin not reported

      Family#PresentingHPOs: Decreased light- and dark-adapted electroretinogram amplitude HP:0000654

      FamilyHPOFreeText: The patient and affected family members have been diagnosed with retinitis pigmentosa.

      Family#PresentingNotHPOs: N/A

      Family#PresentingNotHPOFreeText: N/A

      CountryOrigin: N/A

      Ethnicity: Not mentioned

      Race: Not mentioned

      Family#PreviousTesting: Previous testing consisted of screening of the RHO locus for disease-causing variants.

      Family#Method1: Single strand conformation polymorphism analysis of PCR-amplified cDNA products from the PRPH2 gene, followed by Sanger sequencing of the relevant PCR product to identify the variant

      Family#Method2: SSCP to genotype family members

      Family#GenotypingMethod: Previous testing of this family screening of the RHO locus for variants causing RP, with negative results. Genotyping started with single strand conformation polymorphism analysis of PCR-amplified products from the PRPH2 gene, followed by Sanger sequencing of the relevant PCR product to identify the variant. SSCP was subsequently performed to genotype family members.

      Affected: 3

      Unaffected: 5

      Segregations: Two segregations are reported in the family, with none inconsistent. The family is not reported to be consanguineous. The pedigree is shown in Figure 3c.

      PublishedLOD: N/A

      eLOD: 2.40

      AddSegregationInfo: N/A

      ProbandLabel: Kajiwara_1991_Family_6935_Proband

      Variant: The affected family members harbor the NM_000322.5(PRPH2):c.554T>C (p.Leu185Pro) variant in the heterozygous state.

      ClinVarID: 13165

      gnomAD: This variant was found in gnomAD v4.0.0 at a frequency of 0.00003587 (15 alleles / 418142 total alleles, with no homozygotes) in the European non-Finnish population https://gnomad.broadinstitute.org/variant/6-42721781-A-G?dataset=gnomad_r4

      FamilyAddInfo: The family has not been reported as consanguineous.

      FamilyPMIDs: N/A

    2. mutations of the human homologue of the rds gene

      FamilyLabel: Kajiwara_1991_Family_6948, family national origin not reported

      Family#PresentingHPOs: Decreased light- and dark-adapted electroretinogram amplitude HP:0000654

      FamilyHPOFreeText: The patient and affected family members have been diagnosed with retinitis pigmentosa.

      Family#PresentingNotHPOs: N/A

      Family#PresentingNotHPOFreeText: N/A

      CountryOrigin: N/A

      Ethnicity: Not mentioned

      Race: Not mentioned

      Family#PreviousTesting: Previous testing consisted of screening of the RHO locus for disease-causing variants.

      Family#Method1: Single strand conformation polymorphism analysis of PCR-amplified cDNA products from the PRPH2 gene, followed by Sanger sequencing of the relevant PCR product to identify the variant

      Family#Method2: SSCP to genotype family members

      Family#GenotypingMethod: Previous testing of this family screening of the RHO locus for variants causing RP, with negative results. Genotyping started with single strand conformation polymorphism analysis of PCR-amplified products from the PRPH2 gene, followed by Sanger sequencing of the relevant PCR product to identify the variant. SSCP was subsequently performed to genotype family members.

      Affected: 8

      Unaffected: 7

      Segregations: Eight segregations are reported in the family, with none inconsistent. The family is not reported to be consanguineous. The pedigree is shown in Figure 3b.

      PublishedLOD: N/A

      eLOD: 2.40

      AddSegregationInfo: N/A

      ProbandLabel: Kajiwara_1991_Family_6948_Proband

      Variant: The affected family members harbor the NM_000322.5(PRPH2):c.647C>T (p.Pro216Leu) variant in the heterozygous state.

      ClinVarID: 13164

      gnomAD: This variant was found in gnomAD v4.0.0 at a frequency of 0.000002856 (1 allele / 350108 total alleles, with no homozygotes) in the European non-Finnish population https://gnomad.broadinstitute.org/variant/6-42704546-G-A?dataset=gnomad_r4

      FamilyAddInfo: The family has not been reported as consanguineous.

      FamilyPMIDs: N/A

    1. This deletion segregates with the disease phenotype but is not present in unaffected controls, and suggests that mutant peripherin gives rise to retinitis pigmentosa.

      FamilyLabel: Farrar_1991_Family, family national origin reported as Irish

      Family#PresentingHPOs: Nyctalopia HP:0000662, Peripheral visual field loss HP:0007994, Undetectable dark-adapted electroretinogram HP:0030474, Abnormal light-adapted electroretinogram HP:0008275, Pigmentary retinopathy HP:0000580, Optic disc pallor HP:0000543, Retinal arteriolar constriction HP:0008043, Bone spicule pigmentation of the retina HP:0007737

      FamilyHPOFreeText: The patient and affected family members have been diagnosed with retinitis pigmentosa, with onset noted as late. Nyctalopia generally presented in the early third decade, while rod ERG responses showed reduced amplitudes in the teens.

      Family#PresentingNotHPOs: N/A

      Family#PresentingNotHPOFreeText: N/A

      CountryOrigin: Ireland

      Ethnicity: Not mentioned

      Race: Not mentioned

      Family#PreviousTesting: Previous testing did not occur in this family, although linkage mapping had previously identified PRPH2 as a candidate locus for variants causing RP.

      Family#Method1: Single strand conformation polymorphism analysis of PCR-amplified cDNA products from the PRPH2 gene, followed by Sanger sequencing of the relevant PCR product to identify the variant

      Family#Method2: allele-specific PCR to genotype family members

      Family#GenotypingMethod: Previous testing of this family did not include linkage mapping, although this technique had previously identified PRPH2 as a candidate locus for variants causing RP. Genotyping started with single strand conformation polymorphism analysis of PCR-amplified cDNA products from the PRPH2 gene, followed by Sanger sequencing of the relevant PCR product to identify the variant. Allele-specific PCR was subsequently performed to genotype family members.

      Affected: 9

      Unaffected: 6

      Segregations: Nine segregations are reported in the family, with none inconsistent. The family is not reported to be consanguineous. The pedigree is shown in Figure 2.

      PublishedLOD: N/A

      eLOD: 2.70

      AddSegregationInfo: N/A

      ProbandLabel: Farrar_1991_Proband

      Variant: The affected family members harbor the NM_000322.5:c.356_358del (p.Cys119del) variant in the heterozygous state.

      CAID: CA226222

      gnomAD: This variant was not found in gnomAD v2.1.1.

      FamilyAddInfo: The family has not been reported as consanguineous.

      FamilyPMIDs: N/A

    1. The map position of the rds gene on the 17th chromosome of the mouse

      PMID: 419532

      Gene: PRPH2

      HGNC: 9942

      Disease: retinitis pigmentosa 7

      MONDO: 0011974

      InheritancePattern: semi-dominant

      Prevalence: The incidence of PRPH2-related cases among inherited retinal dystrophies is reported to range from 3.5% in North America to 10.3% in France (PMID: 36010202).

      Penetrance: The penetrance among biallelic cases appears to be complete, while the monoallelic cases exhibit incomplete penetrance (PMID: 36609934).

    1. A 2 base pair deletion in the RDS gene associated with butterfly-shaped pigment dystrophy of the fovea

      PMID: 8251014

      Gene: PRPH2

      HGNC: 9942

      Disease: retinitis pigmentosa 7

      MONDO: 0011974

      InheritancePattern: semi-dominant

      Prevalence: The incidence of PRPH2-related cases among inherited retinal dystrophies is reported to range from 3.5% in North America to 10.3% in France (PMID: 36010202).

      Penetrance: The penetrance among biallelic cases appears to be complete, while the monoallelic cases exhibit incomplete penetrance (PMID: 36609934).