Fishman Jesse, Kuranz Seth, Yeh Michael M, Brzozowski Kaylen, Chen Herman
Apellis Pharmaceuticals, Inc., Waltham, MA 02451, USA.
TriNetX, LLC, Cambridge, MA 02140, USA.
Hematol Rep. 2023 Apr 21;15(2):266-282. doi: 10.3390/hematolrep15020027.
Paroxysmal nocturnal hemoglobinuria (PNH), a rare acquired hematologic disorder, can be treated with C5 inhibitors (C5i) such as eculizumab or ravulizumab. This retrospective study is the first to describe real-world treatment patterns and changes in hematologic PNH-monitoring laboratory tests among C5i-treated US patients. Data were extracted from TriNetX Dataworks Network and included patients with a PNH diagnosis between 1 January 2010, and 20 August 2021. Patients were stratified into three cohorts based on their C5i usage: eculizumab, ravulizumab (prior eculizumab), and ravulizumab (eculizumab naïve). Hematological markers (hemoglobin [Hb], lactate dehydrogenase [LDH], and absolute reticulocyte count [ARC]) and relevant clinical events (e.g., breakthrough hemolysis [BTH], complement-amplifying conditions [CAC], thrombosis, infection, and all-cause mortality) were captured any time within 12 months post-index treatment. Of the 143 (eculizumab), 43 (ravulizumab, prior eculizumab), and 33 (ravulizumab, eculizumab naïve) patients, mean age across cohorts was 42-51 years, 55-61% were female, 63-73% were White, and 33-40% had aplastic anemia. Among all cohorts 12 months post-C5i treatment, 50-82% remained anemic, 8-32% required ≥1 transfusion, and 13-59% had BTH, of which 33%-54% had CACs. Additionally, thrombosis was seen in 7-15% of patients, infection in 20-25%, and mortality in 1-7%. These findings suggest many C5i-treated patients experience suboptimal disease control.
阵发性睡眠性血红蛋白尿(PNH)是一种罕见的获得性血液系统疾病,可用依库珠单抗或ravulizumab等C5抑制剂(C5i)进行治疗。这项回顾性研究首次描述了接受C5i治疗的美国患者的实际治疗模式以及血液学PNH监测实验室检查的变化。数据取自TriNetX Dataworks Network,纳入了2010年1月1日至2021年8月20日期间诊断为PNH的患者。根据C5i使用情况将患者分为三组:依库珠单抗组、ravulizumab(曾用依库珠单抗)组和ravulizumab(未用依库珠单抗)组。在索引治疗后12个月内的任何时间收集血液学指标(血红蛋白[Hb]、乳酸脱氢酶[LDH]和绝对网织红细胞计数[ARC])以及相关临床事件(如突破性溶血[BTH]、补体放大情况[CAC]、血栓形成、感染和全因死亡率)。在143例(依库珠单抗组)、43例(ravulizumab,曾用依库珠单抗组)和33例(ravulizumab,未用依库珠单抗组)患者中,各组的平均年龄为42 - 51岁,55 - 61%为女性,63 - 73%为白人,33 - 40%患有再生障碍性贫血。在所有队列中,C5i治疗后12个月时,50 - 82%的患者仍贫血,8 - 32%的患者需要≥1次输血,13 - 59%的患者发生BTH,其中33% - 54%的患者有CACs。此外,7 - 15%的患者出现血栓形成,20 - 25%的患者出现感染,1 - 7%的患者死亡。这些发现表明,许多接受C5i治疗的患者疾病控制效果欠佳。