Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Labcorp Genetics |
RCV000291561 | SCV001545595 | pathogenic | not provided | 2023-10-15 | criteria provided, single submitter | clinical testing | This sequence change replaces arginine, which is basic and polar, with glutamine, which is neutral and polar, at codon 2038 of the ABCA4 protein (p.Arg2038Gln). This variant is present in population databases (rs767729255, gnomAD 0.006%). This missense change has been observed in individual(s) with retinitis pigmentosa and/or Stargardt disease (PMID: 33261146). 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: 282260). Advanced modeling of protein sequence and biophysical properties (such as structural, functional, and spatial information, amino acid conservation, physicochemical variation, residue mobility, and thermodynamic stability) performed at Invitae indicates that this missense variant is expected to disrupt ABCA4 protein function. This variant disrupts the p.Arg2038 amino acid residue in ABCA4. Other variant(s) that disrupt this residue have been determined to be pathogenic (PMID: 15161829, 24763286, 29854428, 29925512). This suggests that this residue is clinically significant, and that variants that disrupt this residue are likely to be disease-causing. For these reasons, this variant has been classified as Pathogenic. |
3billion | RCV002250614 | SCV002521356 | likely pathogenic | Severe early-childhood-onset retinal dystrophy | 2022-05-22 | criteria provided, single submitter | clinical testing | The variant is observed at an extremely low frequency in the gnomAD v2.1.1 dataset (total allele frequency: 0.002%). In silico tool predictions suggest damaging effect of the variant on gene or gene product (REVEL: 0.89; 3Cnet: 0.85). Same nucleotide change resulting in same amino acid change has been previously reported to be associated with ABCA4 related disorder (PMID: 33261146). A different missense change at the same codon (p.Arg2038Trp) has been reported as pathogenic/likely pathogenic with strong evidence (ClinVar ID: VCV000099430). Therefore, this variant is classified as likely pathogenic according to the recommendation of ACMG/AMP guideline. |
Eurofins Ntd Llc |
RCV000291561 | SCV000333627 | uncertain significance | not provided | 2015-08-11 | flagged submission | clinical testing |