Frontiers in Cardiovascular Medicine · 2022 · 13 citations · 30 references
MedicineGeneticsHematologyGenetic EpidemiologyHuman GeneticsMedical GeneticsCoagulation AbnormalitiesCoagulation DisordersCoagulation Factor DeficienciesGenetic DisordersThrombosisThrombotic DisordersCase-control Study DesignCofactor Protein S DeficiencyCohort Study InvestigationDisease-causing VariantsGenetic ThrombophiliaInherited Coagulation Factor DeficienciesProtein Deficiency DisordersThrombotic Risk Assessment
Plasma levels of the anticoagulant cofactor protein S and PROS1 mutation are reported to impart increased risk of thromboembolism in European and south east Asian populations, but the relationship is not yet documented in Han Chinese in population-based study. Therefore, we undertook a case-control study of this relationship among patients with venous thromboembolism, and probed the genetic factors contributing to low protein S deficiency. Among the 603 consecutively recruited venous thromboembolism patients, 51 (8.5%) proved to be deficient in free protein S antigen (lower than 38.6 U/dl), among whom 30 cases were identified to have a causative mutation by direct sequencing. In contrast, six cases (1.0%) of the 584 healthy controls had low free antigen levels, among whom direct sequencing confirmed disease-causing gene mutations in four controls (0.7%). After adjusting for age and gender, the odds ratio of developing venous thromboembolism in individuals with protein S deficiency based on free protein S tests was 8.1 (95% CI = 3.6–19.9, P < 0.001). Gene sequencing yielded 24 different heterozygous mutations in the 34 participants, of which 13 were newly described. 17 (50%) of the 34 mutations in our study cohort occurred in exons 12 and 13, indicating the LGR2 domain to be a hotspot mutation region for the protein. These findings are conducive to the clinical application of protein S assays for the molecular diagnosis of thrombophilia.
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Familial protein S deficiency is associated with recurrent thrombosis.
P C Comp, Randal R. Nixon, M. Robert Cooper et al. · Journal of Clinical Investigation · 1984 · 603 citations · Full text
Medicine, Biochemistry, Hematology +17
Inherited Thrombophilia: Part 1
David A. Lane, Pier Mannuccio Mannucci, Kenneth A. Bauer et al. · Thrombosis and Haemostasis · 1996 · 594 citations
Medicine, Genetics, Hematology +17
Plasma protein S deficiency in familial thrombotic disease
HP Schwarz, M Fischer, P Hopmeier et al. · Blood · 1984 · 521 citations
Medicine, Genetics, Hematology +14
Ted Koster, Frits R. Rosendaal, E Briët et al. · Blood · 1995 · 371 citations · Full text
Anne C. Dykes, Isobel D. Walker, Alex D. McMahon et al. · British Journal of Haematology · 2001 · 239 citations · Full text
Medicine, Endocrinology, Gynecology +11