Young Guy, Lenting Peter J, Croteau Stacy E, Nolan Beatrice, Srivastava Alok
Hemostasis and Thrombosis Center, Cancer and Blood Diseases Institute, Children's Hospital Los Angeles, University of Southern California Keck School of Medicine, Los Angeles, California, USA.
Laboratory for Hemostasis, Inflammation and Thrombosis, Unité Mixed de Recherche, Institut National de la Santé et de la Recherche Médicale, Université Paris-Saclay, Le Kremlin-Bicêtre.
Res Pract Thromb Haemost. 2023 May 16;7(4):100179. doi: 10.1016/j.rpth.2023.100179. eCollection 2023 May.
Thrombin is a key enzyme in the maintenance of normal hemostatic function and is the central product of an interconnected set of simultaneously occurring cellular and proteolytic events. Antithrombin (AT) is a natural anticoagulant that downregulates different components of the clotting process, particularly thrombin generation. In good health, well-regulated hemostasis is the result of a balance between procoagulant and anticoagulant elements. Cumulative understanding of the regulation of thrombin generation and its central role in hemostasis and bleeding disorders has led to the clinical development of therapeutic strategies that aim to rebalance hemostasis in individuals with hemophilia and other coagulation factor deficiencies to improve bleeding phenotype. The aim of this review is to discuss the rationale for AT lowering in individuals with hemophilia, with a focus on fitusiran, its mechanism of action, and its potential as a prophylactic therapy for individuals with hemophilia A or B, with or without inhibitors. Fitusiran is an investigational small, interfering RNA therapeutic that targets and lowers AT. It is currently in phase III clinical trials and results have shown its potential to increase thrombin generation, leading to enhanced hemostasis and improved quality of life while reducing the overall treatment burden.
凝血酶是维持正常止血功能的关键酶,是一系列同时发生的细胞和蛋白水解事件相互关联的核心产物。抗凝血酶(AT)是一种天然抗凝剂,可下调凝血过程的不同成分,尤其是凝血酶的生成。在健康状态下,良好调节的止血是促凝和抗凝因素之间平衡的结果。对凝血酶生成调节及其在止血和出血性疾病中的核心作用的累积认识,已促成旨在使血友病和其他凝血因子缺乏症患者的止血重新平衡以改善出血表型的治疗策略的临床发展。本综述的目的是讨论血友病患者降低AT的基本原理,重点是fitusiran、其作用机制以及作为血友病A或B患者(无论有无抑制剂)预防性治疗的潜力。Fitusiran是一种研究性的小分子干扰RNA疗法,可靶向并降低AT。它目前正处于III期临床试验阶段,结果显示其有增加凝血酶生成的潜力,从而增强止血效果、改善生活质量,同时减轻总体治疗负担。