Total submissions: 8
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Gene |
RCV000085559 | SCV000577305 | pathogenic | not provided | 2023-07-11 | criteria provided, single submitter | clinical testing | In silico analysis, which includes protein predictors and evolutionary conservation, supports a deleterious effect; This variant is associated with the following publications: (PMID: 15192030, 23341817, 26720470, 31589614, 35156991, 33301772, 9973280, 29555955) |
Labcorp Genetics |
RCV000085559 | SCV001580409 | pathogenic | not provided | 2024-12-19 | criteria provided, single submitter | clinical testing | This sequence change replaces serine, which is neutral and polar, with leucine, which is neutral and non-polar, at codon 1071 of the ABCA4 protein (p.Ser1071Leu). This variant is present in population databases (rs61750065, gnomAD 0.006%). This missense change has been observed in individual(s) with ABCA4-related inherited retinal dystrophies (PMID: 9973280, 15192030, 26720470, 29555955; internal data). In at least one individual the data is consistent with being in trans (on the opposite chromosome) from a pathogenic variant. ClinVar contains an entry for this variant (Variation ID: 99208). Invitae Evidence Modeling of protein sequence and biophysical properties (such as structural, functional, and spatial information, amino acid conservation, physicochemical variation, residue mobility, and thermodynamic stability) indicates that this missense variant is expected to disrupt ABCA4 protein function with a positive predictive value of 95%. For these reasons, this variant has been classified as Pathogenic. |
DASA | RCV002255094 | SCV002526422 | pathogenic | ABCA4-related disorder | 2022-06-10 | criteria provided, single submitter | clinical testing | The c.3212C>T;p.(Ser1071Leu) missense change has been observed in affected individual(s) and ClinVar contains an entry for this variant (ClinVar ID: 99208; PMID: 29555955; PMID: 26720470) - PS4. The variant is located in a mutational hot spot and/or critical and well-established functional domain (AAA_21; ABC_tran) - PM1. The variant is present at low allele frequencies population databases (rs61750065– gnomAD 0.0001314%; ABraOM no frequency - http://abraom.ib.usp.br/) -PM2_supporting. The p.(Ser1071Leu) was detected in trans with a Pathogenic variant (PMID: 26720470) - PM3. Multiple lines of computational evidence support a deleterious effect on the gene or gene product - PP3. In summary, the currently available evidence indicates that the variant is Pathogenic |
Ophthalmic Genetics Group, |
RCV003324508 | SCV004030376 | pathogenic | Cone-rod dystrophy | 2023-07-24 | criteria provided, single submitter | research | Clinical significance based on ACMG v2.0 |
Women's Health and Genetics/Laboratory Corporation of America, |
RCV004689455 | SCV005185667 | pathogenic | Retinitis pigmentosa | 2024-05-30 | criteria provided, single submitter | clinical testing | Variant summary: ABCA4 c.3212C>T (p.Ser1071Leu) results in a non-conservative amino acid change located in the ABC transporter-like, ATP-binding domain (IPR003439) of the encoded protein sequence. Five of five in-silico tools predict a damaging effect of the variant on protein function. The variant allele was found at a frequency of 8e-06 in 251470 control chromosomes. c.3212C>T has been reported in the literature in multiple individuals affected with ABCA4-related inherited retinal dystrophies (examples: Lewis_1999, Stenirri_2004, Muller_2015, Birtel_2018, Peter_2023). These data indicate that the variant is very likely to be associated with disease. The following publications have been ascertained in the context of this evaluation (PMID: 29555955, 9973280, 26720470, 36909829, 15192030). ClinVar contains an entry for this variant (Variation ID: 99208). Based on the evidence outlined above, the variant was classified as pathogenic. |
Fulgent Genetics, |
RCV005025155 | SCV005657433 | pathogenic | Cone-rod dystrophy 3; Age related macular degeneration 2; Severe early-childhood-onset retinal dystrophy; Retinitis pigmentosa 19 | 2024-06-13 | criteria provided, single submitter | clinical testing | |
Retina International | RCV000085559 | SCV000117696 | not provided | not provided | no assertion provided | not provided | ||
Institute of Human Genetics, |
RCV004815102 | SCV005072203 | pathogenic | Retinal dystrophy | 2007-01-01 | no assertion criteria provided | clinical testing |