Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Clin |
RCV003883161 | SCV004697853 | uncertain significance | Monogenic diabetes | 2023-12-25 | reviewed by expert panel | curation | The c.593T>G variant in the hepatic nuclear factor 4-alpha gene, HNF4A, causes an amino acid change of leucine to arginine at codon 198 (p.(Leu198Arg)) of NM_175914.5. This variant is located within the ligand-binding domain (codons 180-220 and 300-350) of HNF4A, which is defined as critical for the protein’s function by the ClinGen MDEP (PM1_Supporting). This variant is predicted to be deleterious by computational evidence, with a REVEL score of 0.98, which is greater than the MDEP VCEP threshold of 0.70 (PP3). This variant is absent from gnomAD v2.1.1 (PM2_Supporting). In summary, c.593T>G meets the criteria to be classified as a variant of uncertain significance for monogenic diabetes. ACMG/AMP criteria applied, as specified by the ClinGen MDEP (specification version 2.0.0, approved 10/22/2023): PP3, PM1_Supporting, PM2_Supporting. |
Athena Diagnostics | RCV000711962 | SCV000842373 | uncertain significance | not provided | 2018-02-28 | criteria provided, single submitter | clinical testing | |
Clinical Genomics, |
RCV003148852 | SCV003804759 | likely risk allele | Maturity onset diabetes mellitus in young | criteria provided, single submitter | research | Potent mutations in HNF4A are associated with poor insulin secretion in response to hyperglycemia. Associated with MODY1. Patients initially respond well to sulfonylureas but eventually become insulin dependent. However, more evidence is required to ascertain the role of this particular variant rs1568735272 in MODY, yet. |