Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Labcorp Genetics |
RCV001961352 | SCV002256260 | likely pathogenic | not provided | 2025-01-01 | criteria provided, single submitter | clinical testing | This sequence change replaces aspartic acid, which is acidic and polar, with asparagine, which is neutral and polar, at codon 390 of the PKLR protein (p.Asp390Asn). This variant is not present in population databases (gnomAD no frequency). This missense change has been observed in individual(s) with pyruvate kinase deficiency (PMID: 9160692, 17977029, 26315463, 27871768; internal data). ClinVar contains an entry for this variant (Variation ID: 1469002). Invitae Evidence Modeling of protein sequence and biophysical properties (such as structural, functional, and spatial information, amino acid conservation, physicochemical variation, residue mobility, and thermodynamic stability) indicates that this missense variant is expected to disrupt PKLR protein function with a positive predictive value of 80%. Experimental studies have shown that this missense change affects PKLR function (PMID: 11960989, 26549847). This variant disrupts the p.Asp390 amino acid residue in PKLR. Other variant(s) that disrupt this residue have been observed in individuals with PKLR-related conditions (PMID: 31747117), which suggests that this may be a clinically significant amino acid residue. 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. |
Mayo Clinic Laboratories, |
RCV001961352 | SCV004226307 | pathogenic | not provided | 2022-06-03 | criteria provided, single submitter | clinical testing | PP3, PM1, PM2, PM3, PS3, PS4_moderate |
Revvity Omics, |
RCV001961352 | SCV004238109 | pathogenic | not provided | 2023-05-19 | criteria provided, single submitter | clinical testing |