Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
3billion | RCV002283966 | SCV002573238 | uncertain significance | Autosomal dominant nonsyndromic hearing loss 20 | 2022-09-01 | criteria provided, single submitter | clinical testing | The variant is not observed in the gnomAD v2.1.1 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: 0.96; 3Cnet: 0.99). Same nucleotide change resulting in same amino acid change has been previously reported to be associated with ACTG1-related disorder (PMID: 23506231). However, the evidence of pathogenicity is insufficient at this time. Therefore, this variant is classified as uncertain significance according to the recommendation of ACMG/AMP guideline. |
Labcorp Genetics |
RCV005227568 | SCV005863405 | pathogenic | Autosomal dominant nonsyndromic hearing loss 20; Baraitser-winter syndrome 2 | 2024-12-05 | criteria provided, single submitter | clinical testing | This sequence change replaces methionine, which is neutral and non-polar, with threonine, which is neutral and polar, at codon 305 of the ACTG1 protein (p.Met305Thr). This variant is not present in population databases (gnomAD no frequency). This missense change has been observed in individual(s) with deafness (PMID: 23506231). It has also been observed to segregate with disease in related individuals. ClinVar contains an entry for this variant (Variation ID: 1705652). 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 ACTG1 protein function with a negative predictive value of 80%. For these reasons, this variant has been classified as Pathogenic. |