Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Mendelics | RCV002249139 | SCV002517377 | likely pathogenic | Hogue-Janssens syndrome 1 | 2022-05-04 | criteria provided, single submitter | clinical testing | |
Labcorp Genetics |
RCV003094013 | SCV003225759 | uncertain significance | not provided | 2022-08-22 | criteria provided, single submitter | clinical testing | This sequence change replaces aspartic acid, which is acidic and polar, with asparagine, which is neutral and polar, at codon 251 of the PPP2R5D protein (p.Asp251Asn). This variant is not present in population databases (gnomAD no frequency). This variant has not been reported in the literature in individuals affected with PPP2R5D-related conditions. ClinVar contains an entry for this variant (Variation ID: 1685412). Algorithms developed to predict the effect of missense changes on protein structure and function are either unavailable or do not agree on the potential impact of this missense change (SIFT: "Deleterious"; PolyPhen-2: "Probably Damaging"; Align-GVGD: "Class C15"). This variant disrupts the p.Asp251 amino acid residue in PPP2R5D. Other variant(s) that disrupt this residue have been determined to be pathogenic (PMID: 28867141, 32371413; Invitae). This suggests that this residue is clinically significant, and that variants that disrupt this residue are likely to be disease-causing. In summary, the available evidence is currently insufficient to determine the role of this variant in disease. Therefore, it has been classified as a Variant of Uncertain Significance. |
3billion | RCV002249139 | SCV005328832 | likely pathogenic | Hogue-Janssens syndrome 1 | 2024-09-20 | criteria provided, single submitter | clinical testing | The variant is not observed in the gnomAD v4.1.0 dataset. Missense changes are a common disease-causing mechanism. In silico tool predictions suggest damaging effect of the variant on gene or gene product [REVEL: 0.67 (>=0.6, sensitivity 0.68 and specificity 0.92); 3Cnet: 0.99 (>=0.6, sensitivity 0.72 and precision 0.9)]. Different missense changes at the same codon (p.Asp251Ala, p.Asp251His, p.Asp251Tyr, p.Asp251Val) have been reported as pathogenic/likely pathogenic with strong evidence (PMID: 28867141, 35586607, 36216457). |