Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Clin |
RCV003448386 | SCV004176237 | pathogenic | Hereditary thrombocytopenia and hematologic cancer predisposition syndrome | 2023-12-09 | reviewed by expert panel | curation | The NM_001754.5(RUNX1):c.270dup (p.Arg91AlafsTer47) variant in RUNX1 is a frameshift variant predicted to cause a premature stop codon in biologically relevant exon 5/9 leading to nonsense mediated decay in a gene in which loss-of-function is an established disease mechanism (PVS1). This variant is absent from gnomAD v2, v3, and v4 with at least 20x coverage (PM2_Supporting). The variant has been reported in a 78yo female with AML, but variant origin is unclear (PMID: 37680325); it has also been reported in 1/3219 patients referred for clinical diagnostic testing who underwent WGS, but phenotype and germline origin are unspecified (PMID: 33726816) (PS4_Supporting is not met). However, other pathogenic/likely pathogenic frameshift alterations in exons 4 or 5 have been reported (PMID: 35764482) (PM5_Supporting). In summary, this variant meets the criteria to be classified as pathogenic for autosomal dominant hereditary thrombocytopenia and hematologic cancer predisposition syndrome based on the ACMG/AMP criteria applied, as specified by the ClinGen Myeloid Malignancy VCEP: PVS1, PM2_Supporting, and PM5_Supporting. (Version 2; date of approval) |
Clinical Genetics and Genomics, |
RCV001269762 | SCV001450005 | pathogenic | not provided | 2018-01-25 | criteria provided, single submitter | clinical testing |