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从基因到疾病;因ADAMTS13基因突变导致的先天性血栓性血小板减少性紫癜

[From gene to disease; congenital thrombotic thrombocytopenic purpura due to mutations in the ADAMTS13 gene].

作者信息

Schiphorst R H, van de Kar N C, van den Heuvel L P

机构信息

Universitair Medisch Centrum St Radboud, afd. Kindernefrologie, Geert Grooteplein Zuid 20, 6525 GA Nijmegen.

出版信息

Ned Tijdschr Geneeskd. 2003 Dec 6;147(49):2422-4.

Abstract

Thrombotic thrombocytopenic purpura (TTP) is characterised by haemolytic anaemia with fragmented erythrocytes and thrombocytopenia, accompanied by other symptoms such as renal dysfunction, neurological signs and fever. Proteolysis of the Von Willebrand blood-clotting factor (VWF) by a VWF cleaving protease or ADAMTS13 is decreased in patients with TTP, leading to ultra-large Von Willebrand multimers in the circulation. A lack of ADAMTS13 activity can be caused by autoimmune inhibitors or may be due to a constitutional deficiency of this protein. Recently, the ADAMTS13 gene that encodes for the ADAMTS13 protein was found. It was mapped to chromosome 9q34 and consists of 29 exons. Several mutations have been identified in the ADAMTS13 gene in patients with the congenital form of TTP. Symptomatic episodes in congenital TTP can be prevented by prophylactic plasma infusions every 2-3 weeks, hereby preventing further organ damage due to thrombotic microangiopathy.

摘要

血栓性血小板减少性紫癜(TTP)的特征是伴有红细胞碎片的溶血性贫血和血小板减少,并伴有其他症状,如肾功能不全、神经体征和发热。TTP患者中,血管性血友病凝血因子(VWF)被VWF裂解蛋白酶或ADAMTS13进行的蛋白水解作用减弱,导致循环中出现超大的血管性血友病多聚体。ADAMTS13活性缺乏可能由自身免疫抑制剂引起,也可能是由于该蛋白的先天性缺陷。最近,发现了编码ADAMTS13蛋白的ADAMTS13基因。它被定位到9号染色体长臂3区4带,由29个外显子组成。在先天性TTP患者的ADAMTS13基因中已鉴定出几种突变。先天性TTP的症状性发作可通过每2-3周预防性输注血浆来预防,从而防止血栓性微血管病导致进一步的器官损伤。

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