Coşkun Çağri, Aksu Tekin, Gülhan Bora, Düzova Ali, Ünal Şule
Departments of Pediatric Hematology.
Pediatric Nephrology, Hacettepe University Faculty of Medicine, Ankara, Turkey.
J Pediatr Hematol Oncol. 2025 Jan 1;47(1):e65-e67. doi: 10.1097/MPH.0000000000002963. Epub 2024 Nov 20.
Congenital thrombotic thrombocytopenic purpura (cTTP), which is associated with mutations in the gene for a disintegrin and metalloproteinase with a thrombospondin type 1 motif member 13 (ADAMTS13), is a chronic and lifelong disease. The clinical course is variable. Regularly using ADAMTS13-containing products such as fresh frozen plasma (FFP) for long-term prophylaxis is the most important treatment to prevent thrombotic microangiopathy (TMA) episodes. Here, we identified novel pathogenic mutations of ADAMTS13 in our patients who experienced severe acute renal failure. Infections can trigger acute hemolytic episodes, and if the initiation of FFP therapy is delayed, this leads to severe organ dysfunction, as in our case. We have shown that regular use of products containing ADAMTS13 can reverse TMA episodes and long-term morbidity and mortality. When severe acute renal failure occurs, daily plasma exchange and N-acetylcysteine (NAC) are useful.
先天性血栓性血小板减少性紫癜(cTTP)与具有血小板反应蛋白-1型基序的去整合素和金属蛋白酶13(ADAMTS13)基因突变相关,是一种慢性终身疾病。临床病程多变。长期规律使用含ADAMTS13的产品,如新鲜冰冻血浆(FFP)进行预防,是预防血栓性微血管病(TMA)发作的最重要治疗方法。在此,我们在经历严重急性肾衰竭的患者中鉴定出ADAMTS13的新致病突变。感染可引发急性溶血发作,如果FFP治疗开始延迟,就会像我们的病例一样导致严重器官功能障碍。我们已经表明,定期使用含ADAMTS13的产品可逆转TMA发作以及长期的发病率和死亡率。当发生严重急性肾衰竭时,每日血浆置换和N-乙酰半胱氨酸(NAC)是有用的。