Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Labcorp Genetics |
RCV001851550 | SCV002239477 | pathogenic | not provided | 2023-03-17 | criteria provided, single submitter | clinical testing | This missense change has been observed in individual(s) with acute intermittent porphyria (PMID: 2243128, 9281416). This sequence change replaces arginine, which is basic and polar, with glutamine, which is neutral and polar, at codon 173 of the HMBS protein (p.Arg173Gln). This variant is not present in population databases (gnomAD no frequency). ClinVar contains an entry for this variant (Variation ID: 1447). 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 HMBS protein function. Experimental studies have shown that this missense change affects HMBS function (PMID: 9281416). This variant disrupts the p.Arg173 amino acid residue in HMBS. Other variant(s) that disrupt this residue have been determined to be pathogenic (PMID: 1301948, 7635464, 11399210, 15643298, 23815679). This suggests that this residue is clinically significant, and that variants that disrupt this residue are likely to be disease-causing. For these reasons, this variant has been classified as Pathogenic. |
OMIM | RCV000001512 | SCV000021667 | pathogenic | Acute intermittent porphyria | 1992-01-01 | no assertion criteria provided | literature only | |
OMIM | RCV003764511 | SCV004697360 | pathogenic | Encephalopathy, porphyria-related | 1992-01-01 | no assertion criteria provided | literature only |