Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Gene |
RCV000182651 | SCV000235029 | pathogenic | not provided | 2012-12-14 | criteria provided, single submitter | clinical testing | p.Ser3938Arg (AGC>AGA): c.11814 C>A in exon 88 of the RYR2 gene (NM_001035.2). The Ser3938Arg mutation in the RYR2 gene has been reported in two unrelated patients with CPVT and it was absent from more than 400 reference alleles (Tester D et al., 2006; Medeiros-Domingo A et al., 2009). Additionally, NHLBI ESP Exome Variant Server reports Ser3938Arg was not observed in approximately 6,000 samples from individuals of European and African American backgrounds, indicating it is not a common benign variant in these populations. Ser3938Arg is located in the channel region, a mutation hot spot, in the RYR2 gene (Medeiros-Domingo A et al., 2009). In summary, Ser3938Arg in the RYR2 gene is interpreted as a disease-causing mutation. The variant is found in CPVT panel(s). |
Labcorp Genetics |
RCV002517785 | SCV000760687 | pathogenic | Catecholaminergic polymorphic ventricular tachycardia 1 | 2024-11-02 | criteria provided, single submitter | clinical testing | This sequence change replaces serine, which is neutral and polar, with arginine, which is basic and polar, at codon 3938 of the RYR2 protein (p.Ser3938Arg). This variant is not present in population databases (gnomAD no frequency). This missense change has been observed in individual(s) with catecholaminergic polymorphic ventricular tachycardia (CPVT) (PMID: 16818210, 30403697; internal data). In at least one individual the variant was observed to be de novo. ClinVar contains an entry for this variant (Variation ID: 201188). 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 RYR2 protein function with a positive predictive value of 95%. For these reasons, this variant has been classified as Pathogenic. |