Total submissions: 4
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Labcorp Genetics |
RCV002512937 | SCV003439573 | likely pathogenic | not provided | 2023-10-22 | criteria provided, single submitter | clinical testing | This sequence change replaces proline, which is neutral and non-polar, with leucine, which is neutral and non-polar, at codon 254 of the KCNJ11 protein (p.Pro254Leu). This variant is present in population databases (rs104894237, gnomAD 0.003%). This missense change has been observed in individuals with autosomal recessive hyperinsulinism (PMID: 15579781, 27188453). ClinVar contains an entry for this variant (Variation ID: 8675). 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 KCNJ11 protein function. Experimental studies have shown that this missense change affects KCNJ11 function (PMID: 15579781). 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. |
Baylor Genetics | RCV003466840 | SCV004198051 | likely pathogenic | Type 2 diabetes mellitus | 2023-09-23 | criteria provided, single submitter | clinical testing | |
OMIM | RCV000009210 | SCV000029428 | pathogenic | Hyperinsulinemic hypoglycemia, familial, 2 | 2004-12-01 | no assertion criteria provided | literature only | |
Clinical Genomics, |
RCV002226640 | SCV002505433 | uncertain significance | Maturity onset diabetes mellitus in young | flagged submission | research | Mutations in KCNJ11 gene can cause decreased production and secretion of insulin. This can lead to MODY which may be responsive to oral sulfonylureas. It is also associated with Neonatal Diabetes. However, no sufficient evidence is found to ascertain the role of rs104894237 variant in MODY yet. |