Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
ARUP Laboratories, |
RCV001001440 | SCV001158678 | likely pathogenic | not specified | 2019-03-18 | criteria provided, single submitter | clinical testing | The F9 c.190T>C; p.Cys64Arg variant (rs137852224), traditionally known as Cys16Arg, is published in the medical literature and gene-specific databases in individuals with hemophilia B (see link to F9 database and references therein). 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. The amino acid at this position is highly conserved and computational algorithms (PolyPhen-2, SIFT) predict this variant is deleterious. Considering available information, this variant is classified as likely pathogenic. References: Link to F9 database: http://www.factorix.org |
Women's Health and Genetics/Laboratory Corporation of America, |
RCV004587010 | SCV005076761 | likely pathogenic | Hereditary factor IX deficiency disease | 2024-04-10 | criteria provided, single submitter | clinical testing | Variant summary: F9 c.190T>C (p.Cys64Arg) results in a non-conservative amino acid change located in the Gamma-carboxyglutamic acid-rich (GLA) domain (IPR000294) of the encoded protein sequence. Five of five in-silico tools predict a damaging effect of the variant on protein function. The variant was absent in 183087 control chromosomes (gnomAD). c.190T>C has been reported in the literature in individuals affected with Factor IX Deficiency (Hemophilia B) (examples: Ludwig_1992, Manno_2003, Hu_2017). These data indicate that the variant is likely to be associated with disease. At least one publication reported this variant reduced clotting activity (Chavali_2009). The following publications have been ascertained in the context of this evaluation (PMID: 19699296, 29037559, 1346077, 12515715). ClinVar contains an entry for this variant (Variation ID: 811523). Based on the evidence outlined above, the variant was classified as likely pathogenic. |