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[Rare VPS33B gene mutation combined with GP1BA mutation causes severe decrease in plasma VWF levels: a case report and literature review].

Authors :
Ma SQ
Bai X
Cao LJ
Ma ZN
Ding ZX
Yu ZJ
Jiang M
Source :
Zhonghua xue ye xue za zhi = Zhonghua xueyexue zazhi [Zhonghua Xue Ye Xue Za Zhi] 2024 Jun 14; Vol. 45 (6), pp. 602-605.
Publication Year :
2024

Abstract

A 28-year-old woman was found to have coagulation factor Ⅷ activity (FⅧ∶C) <1% and von Willebrand factor antigen (VWF∶Ag) <1% during routine prenatal examinations. No pathogenic variation was found in the exon region of the VWF gene using next-generation sequencing. The clinical presentation of this patient does not match the clinical characteristics of type Ⅲ hemophilia [von Willebrand disease (VWD) ]; therefore, third-generation sequencing technology was used to perform whole-genome sequencing on the patient and her family members. Multiple members of the patient's paternal family carried a heterozygous variant of VPS33B, c.869G>C. The family members carrying this variant all had varying degrees of reduced VWF levels (39% -56% ). Moreover, the proband was detected with the heterozygous variant c.1474dupA in GP1BA. The ACMG and Clinvar databases determined that this variation was associated with platelet-type pseudo VWD. The decrease in VWF levels caused by heterozygous variations in VPS33B in families is the first international report, and no previous studies have reported cases of severe decrease in plasma VWF levels caused by double heterozygous variations in VPS33B and GP1BA.

Details

Language :
Chinese
ISSN :
0253-2727
Volume :
45
Issue :
6
Database :
MEDLINE
Journal :
Zhonghua xue ye xue za zhi = Zhonghua xueyexue zazhi
Publication Type :
Academic Journal
Accession number :
39134495
Full Text :
https://doi.org/10.3760/cma.j.cn121090-20231216-00317