Funakoshi Yasutomo, Okada Masahiko, Matsumoto Masanori, Kokame Koichi, Moriuchi Hiroyuki
Department of Pediatrics, Nagasaki University Hospital.
Department of Blood Transfusion Medicine, Nara Medical University.
Rinsho Ketsueki. 2017;58(8):933-937. doi: 10.11406/rinketsu.58.933.
Congenital thrombotic thrombocytopenic purpura (TTP) is a rare hereditary deficiency of ADAMTS13 (von Willebrand factor-cleaving protease) characterized by thrombocytopenia and microangiopathic hemolytic anemia. The spectrum of the clinical phenotype is wide, ranging from asymptomatic episodes of thrombocytopenia to life-threatening multiorgan failure. Reportedly, some patients develop isolated thrombocytopenia during childhood. We herein report sibling cases of congenital TTP. An 11-year-old boy with thrombocytopenia accompanied by influenza virus infection was referred to our hospital. He had a history of severe neonatal jaundice. His 15-year-old brother also had recurrent thrombocytopenia with approximately 10 episodes of recurrence since 3 years of age. Their ADAMTS13 activities were low and ADAMTS13 inhibitors were negative, and a gene analysis confirmed the diagnosis of congenital TTP. Notably, congenital TTP should be included in the differential diagnosis, and it is essential to determine the ADAMTS13 activity for pediatric patients with thrombocytopenia of unknown etiology.
先天性血栓性血小板减少性紫癜(TTP)是一种罕见的遗传性ADAMTS13(血管性血友病因子裂解蛋白酶)缺乏症,其特征为血小板减少和微血管病性溶血性贫血。临床表型范围广泛,从无症状的血小板减少发作到危及生命的多器官衰竭。据报道,一些患者在儿童期出现孤立性血小板减少。我们在此报告先天性TTP的同胞病例。一名11岁血小板减少伴流感病毒感染的男孩被转诊至我院。他有严重新生儿黄疸病史。他15岁的哥哥自3岁起也反复出现血小板减少,复发约10次。他们的ADAMTS13活性较低,ADAMTS13抑制剂为阴性,基因分析确诊为先天性TTP。值得注意的是,先天性TTP应纳入鉴别诊断,对于病因不明的血小板减少儿科患者,确定ADAMTS13活性至关重要。