Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Clin |
RCV004692603 | SCV005196413 | uncertain significance | Hereditary thrombocytopenia and hematologic cancer predisposition syndrome | 2024-08-01 | reviewed by expert panel | curation | NM_001754.5(RUNX1):c.792G>C (p.Gln264His) is a missense variant which is absent from gnomAD v2 and v3 (PM2_Supporting) and has only been reported in a patient with oligopolyposis (PMID: 31942411). The computational predictor REVEL gives a score of 0.392, which is below the threshold of 0.50, and the splice site predictor SpliceAI indicated that the variant has no impact on splicing, evidence that does not predict a damaging effect on RUNX1 function (BP4). In summary, this variant meets the criteria to be classified as a VUS 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: PM2_Supporting and BP4. |
Labcorp Genetics |
RCV001344575 | SCV001538635 | uncertain significance | Hereditary thrombocytopenia and hematological cancer predisposition syndrome associated with RUNX1 | 2022-05-26 | criteria provided, single submitter | clinical testing | This variant is not present in population databases (gnomAD no frequency). In summary, the available evidence is currently insufficient to determine the role of this variant in disease. Therefore, it has been classified as a Variant of Uncertain Significance. Algorithms developed to predict the effect of missense changes on protein structure and function (SIFT, PolyPhen-2, Align-GVGD) all suggest that this variant is likely to be tolerated. ClinVar contains an entry for this variant (Variation ID: 1040855). This variant has not been reported in the literature in individuals affected with RUNX1-related conditions. This sequence change replaces glutamine, which is neutral and polar, with histidine, which is basic and polar, at codon 264 of the RUNX1 protein (p.Gln264His). |