Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Labcorp Genetics |
RCV001899046 | SCV002173351 | likely pathogenic | not provided | 2024-12-16 | 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 158 of the WFS1 protein (p.Glu158Lys). This variant is present in population databases (rs567563179, gnomAD 0.006%). This missense change has been observed in individual(s) with clinical features of Wolfram syndrome (PMID: 19042979, 31658956, 33841295). ClinVar contains an entry for this variant (Variation ID: 1404588). 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 not expected to disrupt WFS1 protein function with a negative predictive value of 80%. Experimental studies have shown that this missense change affects WFS1 function (PMID: 34848728). In summary, the currently available evidence indicates that the variant is pathogenic, but additional data are needed to prove that conclusively. Therefore, this variant has been classified as Likely Pathogenic. |
Mendelics | RCV002246586 | SCV002516172 | uncertain significance | not specified | 2022-05-04 | criteria provided, single submitter | clinical testing | |
Fulgent Genetics, |
RCV005038478 | SCV005664681 | uncertain significance | Cataract 41; Wolfram syndrome 1; Autosomal dominant nonsyndromic hearing loss 6; Type 2 diabetes mellitus; Wolfram-like syndrome | 2024-01-22 | criteria provided, single submitter | clinical testing |