Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Labcorp Genetics |
RCV001377845 | SCV001575282 | likely pathogenic | not provided | 2020-06-23 | criteria provided, single submitter | clinical testing | This variant has been observed in individual(s) with clinical features of Alport syndrome (Invitae). In summary, the currently available evidence indicates that the variant is pathogenic, but additional data are needed to prove that conclusively. Therefore, this variant has been classified as Likely Pathogenic. Glycine residues within the Gly-Xaa-Yaa repeats of the triple helix domain are required for the structure and stability of fibrillar collagens (PMID: 7695699, 8218237, 19344236). In COL4A5, missense variants at these glycine residues are significantly enriched in individuals with disease (PMID: 23720012, 27627812) compared to the general population (ExAC) This variant is not present in population databases (ExAC no frequency). This sequence change replaces glycine with glutamic acid at codon 1036 of the COL4A5 protein (p.Gly1036Glu). The glycine residue is highly conserved and there is a moderate physicochemical difference between glycine and glutamic acid. |
Gene |
RCV001377845 | SCV001818480 | pathogenic | not provided | 2024-07-15 | criteria provided, single submitter | clinical testing | Affects a glycine residue in a Gly-X-Y motif in the triple helical region of the COL4A5 gene, where the majority of pathogenic missense variants occur, and is predicted to disrupt normal protein folding and function (HGMD; PMID: 10752524); Not observed at significant frequency in large population cohorts (gnomAD); Reported in the heterozygous state in association with renal disease in published literature (PMID: 31328266); In silico analysis supports that this missense variant has a deleterious effect on protein structure/function; This variant is associated with the following publications: (PMID: 31328266) |
Fulgent Genetics, |
RCV002499779 | SCV002782320 | pathogenic | X-linked Alport syndrome | 2021-09-06 | criteria provided, single submitter | clinical testing |