Total submissions: 1
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Gene |
RCV000200712 | SCV000251995 | pathogenic | not provided | 2012-05-29 | criteria provided, single submitter | clinical testing | p.Leu396His (CTT>CAT): c.1187 T>A in exon 12 of the OPA1 gene (NM_015560.2) The L396H missense change was identified in the OPA1 gene. It has not been published as a mutation, nor has it been reported as a benign polymorphism to our knowledge. However, other missense mutations at the same position (L396R, L396P) have been reported previously in association with hereditary optic atrophy (Thiselton et al., 2002; Ferre et al., 2009). Furthermore, Leucine 396 is a highly conserved residue in the OPA1 protein that is close to a GTP-binding motif in the dynamin-related domain (Thiselton et al., 2002). Therefore, we interpret L396H to be a disease-associated mutation. The variant is found in OAPEO-MITOP panel(s). |