Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Clin |
RCV002222006 | SCV002499494 | uncertain significance | Monogenic diabetes | 2022-04-07 | reviewed by expert panel | curation | The computational splicing predictor SpliceAI gives a score of 0.56 for donor loss, predicting that the c.326+4A>G variant in the HNF1 homeobox A gene, HNF1A (NM_000545.8), variant disrupts the donor site of intron 1 of HNF1A (PP3). This variant was also identified in an individual with a clinical history highly specific for HNF1A-MODY (MODY probability calculator result >50%, negative genetic testing for HNF4A, and negative antibodies) (PP4_Moderate; PMID: 28862987). This variant is absent from gnomAD v2.1.1 (PM2_Supporting). This variant was identified in three unrelated individuals with non-autoimmune and non-absolute/near-absolute insulin-deficient diabetes; however, PS4_Moderate cannot be applied because this number is below the ClinGen MDEP threshold (PMID: 28862987, internal lab contributors). In summary, c.326+4A>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 1.1, approved 9/30/2021): PP3, PP4_Moderate, PM2_Supporting. |
Women's Health and Genetics/Laboratory Corporation of America, |
RCV000030500 | SCV000053171 | uncertain | Maturity-onset diabetes of the young type 3 | 2011-08-18 | criteria provided, single submitter | curation | Converted during submission to Uncertain significance. |
Clinical Genomics, |
RCV002326708 | SCV002601687 | 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 rs193922595 with MODY3. |