Raife Thomas, Friedman Kenneth D, Fenwick Brad
Department of Pathology, University of Iowa, Iowa City, Iowa 52242, USA.
Thromb Haemost. 2004 Aug;92(2):387-93. doi: 10.1160/TH03-12-0759.
Microvascular thrombosis is a major cause of organ damage in Shiga toxin-mediated hemolytic uremic syndrome (Stx-HUS). In vitro and clinical studies implicate thrombin-mediated mechanisms in the pathogenesis of Stx microvascular thrombosis. In a greyhound model, administration of 0.03 microg/kg to 0.05 microg/kg Stx1 or Stx2 causes severe bloody diarrhea and HUS with microvascular thrombosis requiring humane euthanasia within 65 hours. Using a greyhound model of Stx-HUS we analyzed early hemostatic changes, and tested the hypothesis that thrombin blockade with lepirudin would prevent lethal Stx effects. Two Stx1-exposed greyhounds were analyzed for hemostatic changes prior to onset of clinical manifestations. Serial hemostasis studies after Stx1 challenge revealed trends of increased aPTT, fibrinogen levels, and prothrombin fragment 1+2, and appearance of abnormally large von Willebrand factor multimers. Three greyhounds were anticoagulated with lepirudin to maintain activated partial thromboplastin times (aPTT) >2.5-fold normal, followed by administration of Stx2 and observation of clinical responses. Among the 3 lepirudin-treated, Stx2-challenged greyhounds, one developed severe illness requiring euthanasia. Remarkably, 2 of the 3 greyhounds developed only hypersalivation and restlessness that resolved (P <.03 compared to 14 historical controls). These two greyhounds were clinically, hematologically and biochemically normal 74 hours after Stx administration, well beyond the time of euthanasia of any previous greyhound. This study suggests that greyhounds exposed to Stx develop procoagulant changes similar to humans, and that thrombin may be a critical factor in the pathogenesis and treatment of Stx-HUS.
微血管血栓形成是志贺毒素介导的溶血尿毒综合征(Stx-HUS)中器官损伤的主要原因。体外和临床研究表明凝血酶介导的机制参与了Stx微血管血栓形成的发病过程。在灵缇犬模型中,给予0.03μg/kg至0.05μg/kg的Stx1或Stx2会导致严重的血性腹泻和伴有微血管血栓形成的HUS,需要在65小时内实施安乐死。我们使用灵缇犬Stx-HUS模型分析早期止血变化,并检验用比伐卢定阻断凝血酶可预防致命性Stx效应的假说。对两只暴露于Stx1的灵缇犬在临床表现出现之前分析其止血变化。Stx1攻击后的系列止血研究显示活化部分凝血活酶时间(aPTT)、纤维蛋白原水平和凝血酶原片段1+2增加的趋势,以及出现异常大的血管性血友病因子多聚体。三只灵缇犬用比伐卢定进行抗凝以维持活化部分凝血活酶时间(aPTT)>正常的2.5倍,随后给予Stx2并观察临床反应。在3只接受比伐卢定治疗、Stx2攻击的灵缇犬中,1只病情严重需要安乐死。值得注意的是,3只灵缇犬中有2只仅出现流涎过多和烦躁不安,随后症状缓解(与14只历史对照相比,P<.03)。这两只灵缇犬在给予Stx后74小时临床、血液学和生化指标均正常,远远超过之前任何灵缇犬实施安乐死的时间。这项研究表明,暴露于Stx的灵缇犬会出现与人类相似的促凝变化,并且凝血酶可能是Stx-HUS发病机制和治疗中的关键因素。