Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Clin |
RCV000824707 | SCV000965659 | likely pathogenic | Hereditary thrombocytopenia and hematological cancer predisposition syndrome associated with RUNX1 | 2019-07-26 | reviewed by expert panel | curation | The NM_001754.4:c.314A>C (p.His105Pro) variant affects one of the residues (AA 105-204) within the RHD (PM1_Supporting). This variant is completely absent from all population databases with at least 20x coverage for RUNX1 (PM2). This missense variant has a REVEL score >0.75 (0.953) (PP3). This variant is a missense change at the same residue (p.His105Pro) where a different missense change has been previously established as a likely pathogenic variant (NM_001754.4:c.315C>A (p.His105Glu)) based on MM-VCEP rules for RUNX1 and RNA data or agreement in splicing predictors (SSF and MES) show no splicing effects (PM5_Supporting). This variant has been reported in one proband meeting at least one of the RUNX1-phenotypic criteria (PS4_ Supporting; SCV000807773.1). 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: PM2, PP3, PM1_supporting, PM5_supporting, PS4_supporting. |
Prevention |
RCV003389725 | SCV000807773 | uncertain significance | RUNX1-related disorder | 2024-01-04 | no assertion criteria provided | clinical testing | The RUNX1 c.314A>C variant is predicted to result in the amino acid substitution p.His105Pro. To our knowledge, this variant has not been reported in RUNX1-related patients in the literature or in a large population database, indicating this variant is rare. This variant is interpreted as likely pathogenic by the ClinGen Myeloid Malignancy Variant Curation Expert Panel (https://erepo.clinicalgenome.org/evrepo/ui/interpretation/42a3f999-4991-4180-8cb2-0e8975c5e5bb). Although we suspect that this variant may be pathogenic, at this time, the clinical significance of this variant is uncertain due to the absence of conclusive functional and genetic evidence. |