To analyze the causative gene and the molecular pathogenesis in a pedigree with compound hereditary coagulation factor V deficiency. Routine blood coagulation indexes and factor V antigen (FV:Ag) were detected by the one-stage clotting method and ELISA. Function of the mutant protein was evaluated by the method Calibrated Automated Thrombogram (CAT). The factor V gene was amplified by PCR with direct sequencing. The possible impact of the mutations were analyzed by bioinformatics tools. The proband's factor V activity and FV:Ag were reduced to 3 and 6%. Gene sequencing revealed compound heterozygous mutations c.911G>A (Gly276Glu) in exon 6 and c.5343C>G (Ser1781Arg) in exon 16. The thrombin generation test showed that the mutant protein markedly decreased thrombin. Bioinformatics indicated that mutations were deleterious. The compound heterozygous mutations Gly276Glu and Ser1781Arg were responsible for the decrease of factor V activity and FV:Ag, of which Ser1781Arg was first reported in the world.
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Shiraz Univ Med Sci, Hematol Res Ctr, Hematol & Thrombosis Unit, Shiraz, IranShiraz Univ Med Sci, Hematol Res Ctr, Hematol & Thrombosis Unit, Shiraz, Iran
Afrasiabi, Abdolreza
Sarikhani, Sanaz
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Shiraz Univ Med Sci, Hematol Res Ctr, Hematol & Thrombosis Unit, Shiraz, IranShiraz Univ Med Sci, Hematol Res Ctr, Hematol & Thrombosis Unit, Shiraz, Iran
机构:
Sun Yat Sen Univ, ZhongShan Sch Med, Dept Med Genet, 74,Zhongshan Rd 2, Guangzhou 510080, Guangdong, Peoples R ChinaSun Yat Sen Univ, ZhongShan Sch Med, Dept Med Genet, 74,Zhongshan Rd 2, Guangzhou 510080, Guangdong, Peoples R China
Sun, Liwei
Yan, Qijiang
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Guangzhou Kingmed Diagnost Technol Co LTD, Guangzhou, Guangdong, Peoples R ChinaSun Yat Sen Univ, ZhongShan Sch Med, Dept Med Genet, 74,Zhongshan Rd 2, Guangzhou 510080, Guangdong, Peoples R China
Yan, Qijiang
Wang, Yonghua
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Sun Yat Sen Univ, ZhongShan Sch Med, Dept Med Genet, 74,Zhongshan Rd 2, Guangzhou 510080, Guangdong, Peoples R ChinaSun Yat Sen Univ, ZhongShan Sch Med, Dept Med Genet, 74,Zhongshan Rd 2, Guangzhou 510080, Guangdong, Peoples R China
Wang, Yonghua
Luo, Hualei
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Sun Yat Sen Univ, ZhongShan Sch Med, Dept Med Genet, 74,Zhongshan Rd 2, Guangzhou 510080, Guangdong, Peoples R ChinaSun Yat Sen Univ, ZhongShan Sch Med, Dept Med Genet, 74,Zhongshan Rd 2, Guangzhou 510080, Guangdong, Peoples R China
Luo, Hualei
Du, Peng
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Sun Yat Sen Univ, ZhongShan Sch Med, Dept Med Genet, 74,Zhongshan Rd 2, Guangzhou 510080, Guangdong, Peoples R ChinaSun Yat Sen Univ, ZhongShan Sch Med, Dept Med Genet, 74,Zhongshan Rd 2, Guangzhou 510080, Guangdong, Peoples R China
Du, Peng
Hassan, Reem
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Sun Yat Sen Univ, ZhongShan Sch Med, Dept Med Genet, 74,Zhongshan Rd 2, Guangzhou 510080, Guangdong, Peoples R ChinaSun Yat Sen Univ, ZhongShan Sch Med, Dept Med Genet, 74,Zhongshan Rd 2, Guangzhou 510080, Guangdong, Peoples R China
Hassan, Reem
Liu, Li
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Guangzhou Women & Childrens Med Ctr, Guangzhou, Guangdong, Peoples R ChinaSun Yat Sen Univ, ZhongShan Sch Med, Dept Med Genet, 74,Zhongshan Rd 2, Guangzhou 510080, Guangdong, Peoples R China
Liu, Li
Jiang, Weiying
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Sun Yat Sen Univ, ZhongShan Sch Med, Dept Med Genet, 74,Zhongshan Rd 2, Guangzhou 510080, Guangdong, Peoples R ChinaSun Yat Sen Univ, ZhongShan Sch Med, Dept Med Genet, 74,Zhongshan Rd 2, Guangzhou 510080, Guangdong, Peoples R China
机构:
Ege Univ, Med Fac, Izmir, TurkeySt Thomas Hosp, Viapath Analyt LLP, Mol Haemostasis & Thrombosis, London, England
Kavakli, Kaan
Liesner, Ri
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Great Ormond St Hosp Sick Children, Haemophilia Comprehens Care Ctr, London, EnglandSt Thomas Hosp, Viapath Analyt LLP, Mol Haemostasis & Thrombosis, London, England
Liesner, Ri
Payne, Jeanette
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Dept Paediat Haematol, Sheffield, S Yorkshire, EnglandSt Thomas Hosp, Viapath Analyt LLP, Mol Haemostasis & Thrombosis, London, England
Payne, Jeanette
Norton, Miranda
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Bio Prod Lab Ltd, Elstree, EnglandSt Thomas Hosp, Viapath Analyt LLP, Mol Haemostasis & Thrombosis, London, England
Norton, Miranda
Austin, Steven
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St Georges Univ Hosp NHS Fdn Trust, St Georges Haemophilia Ctr, London, EnglandSt Thomas Hosp, Viapath Analyt LLP, Mol Haemostasis & Thrombosis, London, England