Total submissions: 4
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Clin |
RCV004733216 | SCV005367829 | likely pathogenic | Monogenic diabetes | 2024-09-10 | reviewed by expert panel | curation | The c.343G>T variant in the HNF1 homeobox A gene, HNF1A, causes an amino acid change of valine to leucine at codon 115 (p.(Val115Leu)) of NM_000545.8. This variant is located within a conserved region of the DNA binding domain (codons 107-174 and 201-280) of HNF1A, which is defined as critical for the protein’s function by the ClinGen MDEP (PM1_Supporting). This variant was identified in five unrelated individuals with non-autoimmune and non-absolute/near-absolute insulin-deficient diabetes (PS4_Moderate; ClinVar, internal lab contributors). This variant is predicted to be deleterious by computational evidence, with a REVEL score of 0.902, which is greater than the MDEP VCEP threshold of 0.70 (PP3). This variant was identified in at least two individuals with a clinical history highly specific for HNF1A-MODY (MODY probability calculator result >50% and negative genetic testing for HNF4A) (PP4; internal lab contributors). This variant is absent in gnomAD v2.1.1 (PM2_Supporting). In summary, c.343G>T meets the criteria to be classified as likely pathogenic for monogenic diabetes. ACMG/AMP criteria applied, as specified by the ClinGen MDEP (specification version 2.1.0, approved 8/11/2023): PM1_Supporting, PS4_Moderate, PP3, PP4, PM2_Supporting. |
Broad Center for Mendelian Genomics, |
RCV001248888 | SCV001422568 | uncertain significance | Maturity-onset diabetes of the young type 3 | 2020-01-22 | criteria provided, single submitter | curation | The p.Val115Leu variant in HNF1A has been reported in at least 1 individual with maturity-onset diabetes of the young (PMID: 16917892), and was absent from large population studies. Computational prediction tools and conservation analyses suggest that this variant may impact the protein, though this information is not predictive enough to determine pathogenicity. In summary, while there is some suspicion for a pathogenic role, the clinical significance of this variant is uncertain. ACMG/AMP Criteria applied: PM2, PP3 (Richards 2015). |
Geisinger Clinic, |
RCV001248888 | SCV002562132 | likely pathogenic | Maturity-onset diabetes of the young type 3 | 2022-08-02 | criteria provided, single submitter | research | PM2, PP3, PP4, PS4_Moderate, PM5 |
Clinical Genomics, |
RCV002327605 | SCV002601729 | likely risk allele | Maturity onset diabetes mellitus in young | criteria provided, single submitter | research | Mutations in HNF1A gene can predispose to MODY3. It is associated with both micro and macrovascular complications of diabetes, especially cardiovascular complications. Associated with glucosuria. May respond well to sulfonylureas. However, more evidence is required to confer the association of this particular variant rs1876667856 with MODY3. |