Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Gene |
RCV001588282 | SCV001826714 | pathogenic | not provided | 2022-08-01 | criteria provided, single submitter | clinical testing | Affects a glycine residue in a Gly-X-Y motif in the triple helical region of the COL4A4 gene, where the majority of pathogenic missense variants occur, and is predicted to disrupt normal protein folding and function (HGMD; Jais et al., 2000); Not observed at a significant frequency in large population cohorts (gnomAD); 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: 29204651, 24077912, 10752524, 34400539) |
Fulgent Genetics, |
RCV002495959 | SCV002802061 | pathogenic | Autosomal recessive Alport syndrome; Benign familial hematuria | 2022-03-23 | criteria provided, single submitter | clinical testing | |
Labcorp Genetics |
RCV001588282 | SCV003453968 | uncertain significance | not provided | 2022-10-13 | criteria provided, single submitter | clinical testing | This sequence change replaces glycine, which is neutral and non-polar, with alanine, which is neutral and non-polar, at codon 382 of the COL4A4 protein (p.Gly382Ala). This variant is present in population databases (rs751952236, gnomAD 0.0009%). This missense change has been observed in individual(s) with clinical features of Alport syndrome (PMID: 29204651; Invitae). ClinVar contains an entry for this variant (Variation ID: 1219193). Advanced modeling of protein sequence and biophysical properties (such as structural, functional, and spatial information, amino acid conservation, physicochemical variation, residue mobility, and thermodynamic stability) performed at Invitae indicates that this missense variant is expected to disrupt COL4A4 protein function. This variant disrupts the p.Gly382 amino acid residue in COL4A4. Other variant(s) that disrupt this residue have been observed in individuals with COL4A4-related conditions (Invitae), which suggests that this may be a clinically significant amino acid residue. In summary, the available evidence is currently insufficient to determine the role of this variant in disease. Therefore, it has been classified as a Variant of Uncertain Significance. |