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遗传性异常纤维蛋白原血症:与五种不同纤维蛋白原结构缺陷相关的临床表型。

Inherited dysfibrinogenemia: clinical phenotypes associated with five different fibrinogen structure defects.

作者信息

Miesbach Wolfgang, Scharrer Inge, Henschen Agnes, Neerman-Arbez Marguerite, Spitzer Silvia, Galanakis Dennis

机构信息

Medical Clinic III, Institute of Transfusion Medicine, Goethe University, Frankfurt, Germany.

出版信息

Blood Coagul Fibrinolysis. 2010 Jan;21(1):35-40. doi: 10.1097/MBC.0b013e328331e6db.

Abstract

Hereditary dysfibrinogenemia is a rare clotting disorder, which results from mutations in at least one of the three fibrinogen genes. We examined the frequency of hemostatic clinical and laboratory anomalies at presentation of 37 probands from 12 unrelated families with five different defects (Aalpha R16C, gamma A357T, gamma318-319 del, gamma M310T, and Aalpha R16S), among. The median age was 51 years (11-86 years). Among 62% who were women three (13%) had experienced one or more spontaneous abortion. More than half of the probands had experienced one or more undue bleeding episode, easy bruising being by far the most common. In 19% of probands (9/37, all above age of 50 years), had experienced at least one episode of arterial or venous thrombosis. Among these, were two (7%) with deep venous thrombosis, seven with arterial thrombosis, and five (14%) had experienced both. We propose that the higher frequency of prolonged PT than aPTT, in ours and in other reported studies, reflects the polymerization delay, which in aPTT is attenuated owing to contact activation prior to calcium addition.

摘要

遗传性异常纤维蛋白原血症是一种罕见的凝血障碍,由三种纤维蛋白原基因中至少一种发生突变引起。我们研究了来自12个无亲缘关系的家庭的37名先证者的止血临床和实验室异常情况,这些家庭存在五种不同缺陷(α链R16C、γ链A357T、γ链318 - 319缺失、γ链M310T和α链R16S)。中位年龄为51岁(11 - 86岁)。在62%为女性的先证者中,有三人(13%)经历过一次或多次自然流产。超过一半的先证者经历过一次或多次异常出血事件,其中瘀斑最为常见。19%的先证者(9/37,均超过50岁)经历过至少一次动脉或静脉血栓形成事件。其中,有两人(7%)发生过深静脉血栓形成,七人发生过动脉血栓形成,五人(14%)两者都经历过。我们认为,在我们的研究以及其他已报道的研究中,凝血酶原时间(PT)延长比活化部分凝血活酶时间(aPTT)延长更为常见,这反映了聚合延迟,而在aPTT中,由于在添加钙之前的接触激活,这种延迟有所减轻。

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