Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Clin |
RCV004567930 | SCV005050173 | uncertain significance | Monogenic diabetes | 2024-05-09 | reviewed by expert panel | curation | The c.640T>A variant in the hepatocyte nuclear factor 4-alpha gene, HNF4A, causes an amino acid change of serine to threonine at codon 214 (p.Ser214Thr)) of NM_175914.5. This variant is located within the ligand-binding domain (codons 180-220) of HNF4A, which is defined as critical for the protein's function by the ClinGen MDEP (PM1_Supporting). It is also predicted to be deleterious by computational evidence, with a REVEL score of 0.966, which is greater than the MDEP VCEP threshold of 0.70 (PP3). This variant is absent in gnomAD v2.1.1 (PM2_Supporting). A number of other missense variants at this codon (c.640T>G, p.Ser214Ala; c.641C>T, p.Ser214Phe; c.641C>A, p.Ser214Tyr) have been classified as VUS by the ClinGen MDEP; therefore, PM5 will not be applied. In summary, c.640T>A 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 10/11/2023): PP3, PM1_Supporting, PM2_Supporting. |
Clinical Genomics, |
RCV003148740 | SCV003804760 | 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 rs1060499693 in MODY, yet. | |
Bioscientia Institut fuer Medizinische Diagnostik Gmb |
RCV000449632 | SCV000537783 | likely pathogenic | Maturity-onset diabetes of the young type 1 | no assertion criteria provided | clinical testing |