Total submissions: 6
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Gene |
RCV000235881 | SCV000294195 | uncertain significance | not provided | 2019-11-12 | criteria provided, single submitter | clinical testing | Not observed at a significant frequency in large population cohorts (Lek et al., 2016); In silico analysis, which includes protein predictors and evolutionary conservation, supports a deleterious effect; This variant is associated with the following publications: (PMID: 28493438, 31791873) |
Labcorp Genetics |
RCV000687989 | SCV000815584 | pathogenic | Charcot-Marie-Tooth disease type 2 | 2023-09-12 | criteria provided, single submitter | clinical testing | For these reasons, this variant has been classified as Pathogenic. Experimental studies have shown that this missense change affects AARS function (PMID: 28493438). Advanced modeling of protein sequence and biophysical properties (such as structural, functional, and spatial information, amino acid conservation, physicochemical variation, residue mobility, and thermodynamic stability) performed at Invitae indicates that this missense variant is not expected to disrupt AARS protein function. ClinVar contains an entry for this variant (Variation ID: 246598). This missense change has been observed in individual(s) with autosomal recessive developmental and epileptic encephalopathy (PMID: 28493438, 31791873, 34446925). In at least one individual the data is consistent with being in trans (on the opposite chromosome) from a pathogenic variant. It has also been observed to segregate with disease in related individuals. This variant has been reported in individual(s) with autosomal dominant Charcot-Marie-Tooth disease (Invitae); however, the role of the variant in this condition is currently unclear. This variant is present in population databases (rs369774476, gnomAD 0.006%). This sequence change replaces glycine, which is neutral and non-polar, with aspartic acid, which is acidic and polar, at codon 913 of the AARS protein (p.Gly913Asp). |
Institute of Human Genetics Munich, |
RCV000995470 | SCV001149655 | pathogenic | Developmental and epileptic encephalopathy, 29 | 2018-07-30 | criteria provided, single submitter | clinical testing | |
Institute of Human Genetics, |
RCV000995470 | SCV001439950 | uncertain significance | Developmental and epileptic encephalopathy, 29 | 2019-01-01 | criteria provided, single submitter | clinical testing | This variant was identified as compound heterozygous. |
Institute of Medical Genetics and Applied Genomics, |
RCV000235881 | SCV001446715 | pathogenic | not provided | 2020-10-23 | criteria provided, single submitter | clinical testing | |
OMIM | RCV000995470 | SCV002051752 | pathogenic | Developmental and epileptic encephalopathy, 29 | 2022-03-02 | no assertion criteria provided | literature only |