Total submissions: 1
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Clingen Thrombosis Variant Curation Expert Panel, |
RCV004577659 | SCV005061621 | uncertain significance | Hereditary antithrombin deficiency | 2024-05-09 | reviewed by expert panel | curation | The NM_000488.4:c.391C>G variant in SERPINC1 is a missense variant predicted to cause substitution of Leucine by Valine at amino acid 131 (p.Leu131Val). This variant is also known as antithrombin Southport (Legacy nomenclature: Leu99Val) in the literature. One proband from PMID: 7734360 with AT deficiency (type II-HBS) meets criteria for PP4. At least one patient with this variant displayed AT deficiency (type II- HBS) which is highly specific for SERPINC1 (PP4, PMID: 7734360). Another missense variant, c.391C>T (p.Leu131Phe), in the same codon has been classified as pathogenic for antithrombin deficiency by the ClinGen Thrombosis VCEP (PM5). The computational predictor REVEL gives a score of 0.764, which is above the threshold of 0.6, evidence that correlates with impact to SERPINC1 gene function (PP3). This variant is absent from gnomAD v2.1.1-v4. In summary, this variant meets the criteria to be classified as a variant of uncertain significance for antithrombin deficiency based on the ACMG/AMP criteria applied, as specified by the ClinGen Thrombosis VCEP. (Specifications version 1.0.0; date of approval: 7/17/2023) |