Total submissions: 4
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Gene |
RCV000338902 | SCV000329848 | pathogenic | not provided | 2016-09-30 | criteria provided, single submitter | clinical testing | The G274S variant has also previously been reported in association with SCAD deficiency (Pedersen et al., 2008). Functional analysis of G274S found that it is associated with reduced tetramer formation, increased aggregation tendency, and increased chaperone retention compared to wildtype similar to other pathogenic variants in the ACADS gene (Pedersen et al., 2008). Therefore, we interpret G274S to be a pathogenic variant. |
Counsyl | RCV000675088 | SCV000800601 | uncertain significance | Deficiency of butyryl-CoA dehydrogenase | 2017-09-26 | criteria provided, single submitter | clinical testing | |
Labcorp Genetics |
RCV000675088 | SCV000826273 | uncertain significance | Deficiency of butyryl-CoA dehydrogenase | 2022-07-21 | criteria provided, single submitter | clinical testing | This sequence change replaces glycine, which is neutral and non-polar, with serine, which is neutral and polar, at codon 274 of the ACADS protein (p.Gly274Ser). This variant is present in population databases (rs746368198, gnomAD 0.005%). This missense change has been observed in individual(s) with short-chain acyl-CoA dehydrogenase (SCAD) deficiency (PMID: 18523805). ClinVar contains an entry for this variant (Variation ID: 280061). 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 expected to disrupt ACADS protein function. Experimental studies have shown that this missense change affects ACADS function (PMID: 18523805). Algorithms developed to predict the effect of sequence changes on RNA splicing suggest that this variant may disrupt the consensus splice site. 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. |
Genome- |
RCV000675088 | SCV002033252 | likely pathogenic | Deficiency of butyryl-CoA dehydrogenase | 2021-11-07 | criteria provided, single submitter | clinical testing |