Total submissions: 4
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Clin |
RCV004702402 | SCV001244324 | likely pathogenic | Hereditary thrombocytopenia and hematologic cancer predisposition syndrome | 2024-08-28 | reviewed by expert panel | curation | NM_001754.4:c.593A>T (p.Asp198Val) is a missense variant which affects a hotspot residue within the RHD (PM1). Two probands meeting RUNX1-phenotypic criteria were reported to have this variant (PMID: 27479822; Internal laboratory data) (PS4_moderate). This variant has a REVEL score of 0.959 > 0.75 (PP3). This variant is completely absent from all population databases with at least 20x coverage for RUNX1 (PM2_Supporting). Additionally, this variant is a missense change at the same residue (p.D198) where a different missense change has been previously established as a likely pathogenic variant (ClinVar ID 561251, 2665098) based on MM-VCEP rules for RUNX1 (PM5_Supporting). In summary, this variant meets criteria to be classified as likely pathogenic. ACMG/AMP criteria applied, as specified by the ClinGen Myeloid Malignancy Variant Curation Expert Panel for RUNX1: PM1, PS4_moderate, PP3, PM2_supporting, PM5_supporting. |
NIHR Bioresource Rare Diseases, |
RCV000852167 | SCV000899823 | likely pathogenic | Thrombocytopenia | 2019-02-01 | criteria provided, single submitter | research | |
Birmingham Platelet Group; University of Birmingham | RCV001270576 | SCV001450875 | likely pathogenic | Abnormal bleeding; Thrombocytopenia | 2020-05-01 | no assertion criteria provided | research | |
Prevention |
RCV004723162 | SCV005336585 | pathogenic | RUNX1-related disorder | 2024-07-02 | no assertion criteria provided | clinical testing | The RUNX1 c.593A>T variant is predicted to result in the amino acid substitution p.Asp198Val. This variant has been reported in several individuals with inherited thrombocytopenia/platelet disorder (reported as p.Asp171Val in Johnson. 2016. PubMed ID: 27479822; Table 2, Almazni et al. 2020. PubMed ID: 32935436; Table S3, Downes et al. 2019. PubMed ID: 31064749; Brown et al. 2020. PubMed ID: 32208489). This variant has not been reported in a large population database, indicating this variant is rare. This variant is interpreted as pathogenic. |