Hematology-Oncology Section, Department of Internal Medicine, University of Oklahoma Health Sciences Center, Oklahoma City, OK 73104, USA.
Jimmy Everest Section of Pediatric Hematology-Oncology, Department of Pediatrics, University of Oklahoma Health Sciences Center, Oklahoma City, OK 73104, USA.
Genes (Basel). 2023 Oct 18;14(10):1956. doi: 10.3390/genes14101956.
Hereditary thrombotic thrombocytopenic purpura (hTTP), also known as Upshaw-Schulman syndrome, is a rare genetic disorder caused by mutations in the ADAMTS13 gene that leads to decreased or absent production of the plasma von Willebrand factor (VWF)-cleaving metalloprotease ADAMTS13. The result is circulating ultra-large multimers of VWF that can cause microthrombi, intravascular occlusion and organ damage, especially at times of turbulent circulation. Patients with hTTP may have many overt or clinically silent manifestations, and a high index of suspicion is required for diagnosis. For the treatment of hTTP, the goal is simply replacement of ADAMTS13. The primary treatment is prophylaxis with plasma infusions or plasma-derived factor VIII products, providing sufficient ADAMTS13 to prevent acute episodes. When acute episodes occur, prophylaxis is intensified. Recombinant ADAMTS13, which is near to approval, will immediately be the most effective and also the most convenient treatment. In this review, we discuss the possible clinical manifestations of this rare disease and the relevant differential diagnoses in different age groups. An extensive discussion on prophylaxis and treatment strategies is also presented. Unique real patient cases have been added to highlight critical aspects of hTTP manifestations, diagnosis and treatment.
遗传性血栓性血小板减少性紫癜(hTTP),又称 Ushaws-Schulman 综合征,是一种罕见的遗传性疾病,由 ADAMTS13 基因突变引起,导致血浆血管性血友病因子(VWF)裂解金属蛋白酶 ADAMTS13 的产生减少或缺失。其结果是循环中超大的 VWF 多聚体,可导致微血栓、血管内闭塞和器官损伤,尤其是在血流湍急时。hTTP 患者可能有许多显性或临床无症状的表现,因此需要高度怀疑才能诊断。hTTP 的治疗目的很简单,就是替代 ADAMTS13。主要治疗方法是预防性输注血浆或血浆源性 VIII 因子制品,提供足够的 ADAMTS13 以预防急性发作。当急性发作时,会加强预防措施。即将批准的重组 ADAMTS13 将立即成为最有效、最方便的治疗方法。在这篇综述中,我们讨论了这种罕见疾病在不同年龄组中的可能临床表现及相关鉴别诊断。还对预防和治疗策略进行了广泛的讨论。添加了真实患者的病例,以突出 hTTP 表现、诊断和治疗的关键方面。