Total submissions: 1
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Laboratory for Molecular Medicine, |
RCV000826123 | SCV000967632 | likely pathogenic | Rare genetic deafness | 2018-02-05 | criteria provided, single submitter | clinical testing | The p.Asn1665fs variant in OTOGL has not been previously reported in individuals with hearing loss and was absent from large population studies. This variant is predicted to cause a frameshift, which alters the protein?s amino acid sequence beginning at position 1665 and leads to a premature termination codon 13 amino acids downstream. This alteration is then predicted to lead to a truncated or ab sent protein. Loss of function of the OTOGL gene is an established disease mecha nism in autosomal recessive hearing loss. In summary, although additional studie s are required to fully establish its clinical significance, this variant is lik ely pathogenic for autosomal recessive hearing loss based on its predicted impac t on the protein. ACMG/AMP Criteria applied: PVS1, PM2. |