Total submissions: 4
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Institute of Human Genetics, |
RCV000148319 | SCV001440416 | pathogenic | Alternating hemiplegia of childhood 2 | 2019-01-01 | criteria provided, single submitter | clinical testing | |
3billion, |
RCV000148319 | SCV002012103 | pathogenic | Alternating hemiplegia of childhood 2 | 2021-10-02 | criteria provided, single submitter | clinical testing | Same nucleotide change resulting in same amino acid change has been previously reported as pathogenic/likely pathogenic with strong evidence (ClinVar ID: VCV000161138.2, PS1_S). The missense variant is located in a mutational hot spot and/or well-established functional domain in which established pathogenic variants have been reported (PM1). It is not observed in the gnomAD v2.1.1 dataset (PM2). A different missense change at the same codon (p.Asn773Ile) has been reported as pathogenic (VCV000161139.1, PM5). The variant was observed as assumed (i.e. paternity and maternity not confirmed) de novoo (3billion dataset, PM6). In silico tool predictions suggest damaging effect of the variant on gene or gene product (REVEL: 0.679, PP3). Therefore, this variant is classified as pathogenic according to the recommendation of ACMG/AMP guideline. |
Gene |
RCV002510791 | SCV002820658 | pathogenic | not provided | 2024-05-20 | criteria provided, single submitter | clinical testing | Not observed at significant frequency in large population cohorts (gnomAD); In silico analysis supports that this missense variant has a deleterious effect on protein structure/function; This variant is associated with the following publications: (PMID: 32454213, 35177115, 26417536, 27312461, 24739246, 25447930, 26410222, 27577505, 22842232, 31061839, 31942761) |
Labcorp Genetics |
RCV002514853 | SCV003443339 | pathogenic | Dystonia 12 | 2024-11-11 | criteria provided, single submitter | clinical testing | This sequence change replaces asparagine, which is neutral and polar, with serine, which is neutral and polar, at codon 773 of the ATP1A3 protein (p.Asn773Ser). This variant is not present in population databases (gnomAD no frequency). This missense change has been observed in individual(s) with ATP1A3-related conditions (PMID: 22842232, 32454213). In at least one individual the variant was observed to be de novo. ClinVar contains an entry for this variant (Variation ID: 161138). 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 ATP1A3 protein function with a positive predictive value of 95%. For these reasons, this variant has been classified as Pathogenic. |