Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Clin |
RCV004577531 | SCV005061684 | likely pathogenic | Glanzmann thrombasthenia | 2024-05-02 | reviewed by expert panel | curation | The frameshift variant NM_000419.5(ITGA2B):c.1973del (p.Ala658GlufsTer?) has been reported in one individual with a disorder of platelet function, (TGP0048 in PMID:31064749). Communication with the authors confirmed a diagnosis of Glanzmann thrombasthenia. The patient had impaired platelet aggregation and prolonged bleeding after dental extraction. This individual, TGP0048, is compound heterozygous for this frameshift variant and a variant of uncertain significance (NM_000419.5(ITGA2B):c.188+5G>T). The c.1973del frameshift variant causes a premature stop codon in exon 20/30, which is predicted to undergo nonsense mediated decay in a disease that is loss of function (PVS1). The highest population minor allele frequency in gnomAD v4.0.0 is 0.00006335 (1/15786 alleles) in the African/African American population, which is lower than the ClinGen PD VCEP threshold (<0.0001; PM2_Supporting). In summary, this variant meets the criteria to be classified as Likely Pathogenic for autosomal recessive Glanzmann Thrombasthenia based on the ACMG/AMP criteria applied, as specified by the ClinGen PD VCEP: PVS1 and PM2_supporting (VCEP specifications version 2.1.0). |
NIHR Bioresource Rare Diseases, |
RCV000851728 | SCV000899581 | likely pathogenic | Abnormal platelet aggregation | 2019-02-01 | criteria provided, single submitter | research |