Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Laboratory for Molecular Medicine, |
RCV000616141 | SCV000713099 | likely pathogenic | Primary dilated cardiomyopathy; Arrhythmogenic right ventricular cardiomyopathy | 2017-06-13 | criteria provided, single submitter | clinical testing | The p.Gln76X variant in DSP has not been previously reported in individuals with cardiomyopathy or in large population studies. This nonsense variant leads to a premature termination codon at position 76, which is predicted to lead to a tru ncated or absent protein. Heterozygous loss of function of the DSP gene is an es tablished disease mechanism in individuals with arrhythmogenic right ventricular cardiomyopathy, and has recently been associated with dilated cardiomyopathy. I n summary, although additional studies are required to fully establish its clini cal significance, the p.Gln76X variant is likely pathogenic. |
Gene |
RCV004721473 | SCV005328131 | likely pathogenic | not provided | 2023-07-14 | criteria provided, single submitter | clinical testing | Nonsense variant predicted to result in protein truncation or nonsense mediated decay in a gene for which loss of function is a known mechanism of disease; Not observed at significant frequency in large population cohorts (gnomAD); Has not been previously published in association with DSP-related disorders to our knowledge; This variant is associated with the following publications: (PMID: 31638835, 31447099, 33684294) |