Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Gene |
RCV000519479 | SCV000617776 | pathogenic | not provided | 2017-07-14 | criteria provided, single submitter | clinical testing | The R310Q pathogenic variant in the GDAP1 gene has been previously reported with a pathogenic variant on the opposite allele (in trans) as a in two siblings with with autosomal recessive CMT (Azzedine et al., 2003). Functional studies demonstrate that R310Q induces peroxisomal fission and allows fusion of the mitochondrial matrix (Huber et al., 2013). The R310Q variant is not observed in large population cohorts (Lek et al., 2016; 1000 Genomes Consortium et al., 2015; Exome Variant Server). The R310Q variant is a semi-conservative amino acid substitution, which may impact secondary protein structure as these residues differ in some properties. This substitution occurs at a position that is conserved across species. Therefore, R282C is interpreted to be a pathogenic variant. |
Inherited Neuropathy Consortium | RCV000789782 | SCV000929166 | uncertain significance | Charcot-Marie-Tooth disease | no assertion criteria provided | literature only |