Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Dept. |
RCV001726695 | SCV001960991 | pathogenic | 3-Oxo-5 alpha-steroid delta 4-dehydrogenase deficiency | criteria provided, single submitter | clinical testing | ||
Labcorp Genetics |
RCV001726695 | SCV005697689 | pathogenic | 3-Oxo-5 alpha-steroid delta 4-dehydrogenase deficiency | 2024-09-06 | criteria provided, single submitter | clinical testing | This sequence change replaces glutamic acid, which is acidic and polar, with lysine, which is basic and polar, at codon 197 of the SRD5A2 protein (p.Glu197Lys). This variant is present in population databases (rs534671822, gnomAD 0.01%). This missense change has been observed in individual(s) with steroid-5 alpha-reductase deficiency (PMID: 30695888, 35700942, 36617173; internal data). ClinVar contains an entry for this variant (Variation ID: 1298365). 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 SRD5A2 protein function with a positive predictive value of 80%. Algorithms developed to predict the effect of sequence changes on RNA splicing suggest that this variant may disrupt the consensus splice site. This variant disrupts the p.Glu197 amino acid residue in SRD5A2. Other variant(s) that disrupt this residue have been determined to be pathogenic (PMID: 8262007, 10999800, 20019388; internal data). 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. |