Total submissions: 1
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Molecular Genetics, |
RCV002225221 | SCV002503810 | uncertain significance | Congenital muscular hypertrophy-cerebral syndrome | 2023-03-30 | criteria provided, single submitter | clinical testing | This sequence change is predicted to replace glutamine with arginine at codon 958 of the SMC1A protein (p.(Gln958Arg)). The glutamine residue is invariant across species (100 vertebrates, UCSC), and is a critical residue in an ATM kinase substrate motif adjacent to an established phosphoserine in the N terminal domain (PM1; PMID: 11877377, 16273072). There is a small physicochemical difference between glutamine and arginine. The variant is absent in a large population cohort (PM2; gnomAD v2.1, v3.0), and has not been reported in the relevant medical literature or databases. Multiple lines of computational evidence predict a benign effect for the missense substitution (5/6 algorithms), but the gene is highly missense-constrained. Based on the classification scheme RMH ACMG Guidelines v1.2.1, this variant is classified as a VARIANT OF UNCERTAIN SIGNIFICANCE. Following criteria are met: PM1, PM2. |