Total submissions: 5
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Women's Health and Genetics/Laboratory Corporation of America, |
RCV001194005 | SCV001363224 | likely pathogenic | Maturity onset diabetes mellitus in young | 2019-11-06 | criteria provided, single submitter | clinical testing | Variant summary: HNF1A c.52G>T (p.Glu18X) results in a premature termination codon, predicted to cause a truncation of the encoded protein or absence of the protein due to nonsense mediated decay, which are commonly known mechanisms for disease. Truncations downstream of this position have been classified as pathogenic by our laboratory (c.511C>T, p.Arg171X; c.526C>T, p.Gln176X). The variant was absent in 248304 control chromosomes (gnomAD). To our knowledge, no occurrence of c.52G>T in individuals affected with Maturity Onset Diabetes of the Young 3 and no experimental evidence demonstrating its impact on protein function have been reported. No clinical diagnostic laboratories have submitted clinical-significance assessments for this variant to ClinVar after 2014. Based on the evidence outlined above, the variant was classified as likely pathogenic. |
Geisinger Clinic, |
RCV002285453 | SCV002562138 | pathogenic | Maturity-onset diabetes of the young type 3 | 2022-08-02 | criteria provided, single submitter | research | PVS1, PM2 |
Clinical Genomics, |
RCV001194005 | SCV002601659 | pathogenic | 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 rs1876084046 with MODY3. | |
Ambry Genetics | RCV001194005 | SCV002643089 | pathogenic | Maturity onset diabetes mellitus in young | 2016-03-16 | criteria provided, single submitter | clinical testing | The p.E18* pathogenic mutation (also known as c.52G>T), located in coding exon 1 of the HNF1A gene, results from a G to T substitution at nucleotide position 52. This changes the amino acid from a glutamic acid to a stop codon within coding exon 1. Since premature stop codons are typically deleterious in nature, this alteration is interpreted as a disease-causing mutation (ACMG Recommendations for Standards for Interpretation and Reporting of Sequence Variations. Revision 2007. Genet Med. 2008;10:294). |
Gene |
RCV003324818 | SCV004030816 | likely pathogenic | not provided | 2023-02-22 | criteria provided, single submitter | clinical testing | Nonsense variant predicted to result in protein truncation or nonsense mediated decay in a gene for which loss of function is a known mechanism of disease; Not observed at significant frequency in large population cohorts (gnomAD); Has not been previously published as pathogenic or benign to our knowledge; This variant is associated with the following publications: (PMID: 36257325, 35673428, 35328643, 32395877, 31517624) |