Department of Hematology and Central Hematology Laboratory, Inselspital, Bern University Hospital, Freiburgstrasse, Bern, Switzerland.
Department of Clinical Research, University of Bern, Bern, Switzerland.
Nat Rev Dis Primers. 2017 Apr 6;3:17020. doi: 10.1038/nrdp.2017.20.
Thrombotic thrombocytopenic purpura (TTP; also known as Moschcowitz disease) is characterized by the concomitant occurrence of often severe thrombocytopenia, microangiopathic haemolytic anaemia and a variable degree of ischaemic organ damage, particularly affecting the brain, heart and kidneys. Acute TTP was almost universally fatal until the introduction of plasma therapy, which improved survival from <10% to 80-90%. However, patients who survive an acute episode are at high risk of relapse and of long-term morbidity. A timely diagnosis is vital but challenging, as TTP shares symptoms and clinical presentation with numerous conditions, including, for example, haemolytic uraemic syndrome and other thrombotic microangiopathies. The underlying pathophysiology is a severe deficiency of the activity of a disintegrin and metalloproteinase with thrombospondin motifs 13 (ADAMTS13), the protease that cleaves von Willebrand factor (vWF) multimeric strings. Ultra-large vWF strings remain uncleaved after endothelial cell secretion and anchorage, bind to platelets and form microthrombi, leading to the clinical manifestations of TTP. Congenital TTP (Upshaw-Schulman syndrome) is the result of homozygous or compound heterozygous mutations in ADAMTS13, whereas acquired TTP is an autoimmune disorder caused by circulating anti-ADAMTS13 autoantibodies, which inhibit the enzyme or increase its clearance. Consequently, immunosuppressive drugs, such as corticosteroids and often rituximab, supplement plasma exchange therapy in patients with acquired TTP.
血栓性血小板减少性紫癜(TTP;也称为 Moschcowitz 病)的特征是同时发生严重的血小板减少症、微血管病性溶血性贫血和不同程度的缺血性器官损伤,特别是影响大脑、心脏和肾脏。急性 TTP 在血浆治疗引入之前几乎普遍致命,该治疗将生存率从<10%提高到 80-90%。然而,急性发作后存活的患者有很高的复发和长期发病风险。及时诊断至关重要,但具有挑战性,因为 TTP 与许多疾病共享症状和临床表现,例如溶血尿毒症综合征和其他血栓性微血管病。潜在的病理生理学是一种严重缺乏具有血小板反应蛋白 13 结构域的金属蛋白酶 13(ADAMTS13)的活性,该蛋白酶可切割血管性血友病因子(vWF)多聚体链。内皮细胞分泌和锚定后,超大 vWF 链仍未被切割,与血小板结合形成微血栓,导致 TTP 的临床表现。先天性 TTP(Upshaw-Schulman 综合征)是由于 ADAMTS13 的纯合子或复合杂合突变所致,而获得性 TTP 是由循环抗 ADAMTS13 自身抗体引起的自身免疫性疾病,该自身抗体抑制酶或增加其清除率。因此,免疫抑制药物,如皮质类固醇和通常的利妥昔单抗,在获得性 TTP 患者中补充血浆置换治疗。