Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Clin |
RCV003509643 | SCV004363685 | pathogenic | Hereditary factor IX deficiency disease | 2024-02-09 | reviewed by expert panel | curation | The NM_000133.4(F9):c.720G>A variant predicts a nonsense change, Trp240Ter, in exon 6 of the F9 gene; NMD is predicted, meeting PVS1 criteria. The variant is absent from gnomAD v2.1.1 and v3, meeting PM2_Supporting criteria. At least 8 severe hemophilia B patients meeting the F9 phenotype criteria have been reported in the literature (PMIDs: 23093250, 18479429, 8091381, 15086324), meeting PS4_Very strong. Inhibitors to factor replacement therapies have been reported (EAHAD database). In summary, the variant meets criteria to be classified as pathogenic. ACMG/AMP criteria applied, as specified by the Coagulation Factor Deficiency VCEP for F9: PVS1, PS4_Very strong, PM2_Supporting. |
ARUP Laboratories, |
RCV001000165 | SCV001156653 | pathogenic | not specified | 2019-02-13 | criteria provided, single submitter | clinical testing | The F9 c.720G>A; p.Trp240Ter variant, also known as 20562G>A; Trp194Stop, is published in the medical literature in individuals with severe hemophilia B (Belvini 2005, Jenkins 2008). The variant is absent from general population databases (1000 Genomes Project, Exome Variant Server, and Genome Aggregation Database), indicating it is not a common polymorphism. This variant induces an early termination codon and is predicted to result in a truncated protein or mRNA subject to nonsense-mediated decay. Considering available information, this variant is classified as pathogenic. References: Belvini D et al. Molecular genotyping of the Italian cohort of patients with hemophilia B. Haematologica. 2005 May;90(5):635-42. Jenkins PV et al. Mutation analysis of haemophilia B in the Irish population: increased prevalence caused by founder effect. Haemophilia. 2008 Jul;14(4):717-22. |