Total submissions: 1
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Clin |
RCV001580214 | SCV001809847 | uncertain significance | Glanzmann thrombasthenia | 2024-06-06 | reviewed by expert panel | curation | The ITGB3 missense variant NM_000212.2:c.1261G>A replaces the valine residue with a methionine residue (p.Val421Met) and is absent from control population databases. This variant has been observed in heterozygosity in an individual with a phenotype specific for Glanzmann's thrombasthenia (GT) (GT-09, PMID: 16463284); history of bleeding and impaired aggregation to at least two agonists, but normal or only mildly reduced agglutination with ristocetin (PP4_Moderate). It was observed in heterozygosity in GT-09 (PMID: 16463284) in combination with ITGB3 variant c.428T>G (p.Leu143Trp), classified pathogenic by the PD-EP, confirmation of phasing was not reported (PM3_supporting). The highest population minor allele frequency in gnomAD v4.1.0 is 0.00002666 (2/75032 alleles) in the African/African American population, which is lower than the ClinGen PD VCEP threshold (<0.0001; PM2_Supporting). In silico tools predict the variant is damaging to protein function (REVEL score of 0.738; PP3). In summary, this variant is of uncertain significance and lacks sufficient evidence to be classified as pathogenic or benign for GT. GT-specific criteria applied: PP4_moderate, PP3, PM2_supporting, PM3_supporting. |