Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Rady Children's Institute for Genomic Medicine, |
RCV005250356 | SCV005900615 | likely pathogenic | Neurohypophyseal diabetes insipidus | 2024-09-06 | criteria provided, single submitter | clinical testing | This variant is also referred to as c.1884G>A (p.Gly96Asp) in the literature. The c.287G>A (p.Gly96Asp) variant affects a highly conserved amino acid and is predicted by multiple in silico tools to have a deleterious effect on protein function. This variant has been previously reported in patients with neurohypophyseal diabetes insipidus (PMID: 14673472, 24825090). The c.287G>A (p.Gly96Asp) variant segregated within a family with neurohypophyseal diabetes insipidus (PMID: 19129716). Different amino acid changes at the same residue (p.Gly96Val) have been previously reported in individuals with neurohypophyseal diabetes insipidus (PMID: 8626836, 8706292). Functional studies indicate this variant may lead to reduced vasopressin secretion (PMID: 19129716). The c.287G>A (p.Gly96Asp) variant is absent from the latest version of the gnomAD population database and thus is presumed to be rare. Based on the available evidence, c.287G>A (p.Gly96Asp) is classified as Likely Pathogenic. |
Prevention |
RCV004757063 | SCV005365519 | likely pathogenic | AVP-related disorder | 2024-05-02 | no assertion criteria provided | clinical testing | The AVP c.287G>A variant is predicted to result in the amino acid substitution p.Gly96Asp. This variant is also referred to as g.1884G>A in the literature. It has been reported to segregate with disease in families with neurohypophyseal diabetes insipidus (Christensen et al. 2004. PubMed ID: 14673472; Hedrich et al. 2009. PubMed ID: 19129716; Brugnara et al. 2014. PubMed ID: 24825090). In vitro experimental studies suggest this variant impacts protein function (Hedrich et al. 2009. PubMed ID: 19129716). Another nucleotide substitution affecting this amino acid (p.Gly96Val) has also been reported in individuals with neurohypophyseal diabetes insipidus (Rauch et al. 1996. PubMed ID: 8706292). This variant has not been reported in a large population database, indicating this variant is rare. This variant is interpreted as likely pathogenic. |