Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Athena Diagnostics | RCV002472922 | SCV002771009 | pathogenic | not provided | 2022-04-06 | criteria provided, single submitter | clinical testing | This variant has not been reported in large, multi-ethnic general populations (http://gnomad.broadinstitute.org). This variant segregates with Charcot-Marie-Tooth disease in at least one family. Assessment of experimental evidence suggests this variant results in abnormal protein function. Knock-in mice displayed a phenotype similar to Charcot-Marie-Tooth disease (PMID: 25206829, 28780615). Additionally, motor neurons expressing this variant exhibited neurite degeneration (PMID: 20538880). Further studies also showed that this variant leads to decreased binding to BAG3 (PMID: 20858900), but increased binding to HSPB1 and the formation of intracellular aggregates (PMID: 15122253, 20157854). |
OMIM | RCV000002737 | SCV000022895 | pathogenic | Charcot-Marie-Tooth disease axonal type 2L | 2005-02-01 | no assertion criteria provided | literature only | |
Inherited Neuropathy Consortium | RCV000192250 | SCV000929353 | uncertain significance | Charcot-Marie-Tooth disease | no assertion criteria provided | literature only |