Hemophilia and Thrombosis Treatment Center, Rady Children's Hospital San Diego, San Diego, CA, USA.
Department of Pediatrics, University of California, San Diego, La Jolla, CA, USA.
BioDrugs. 2023 Sep;37(5):595-606. doi: 10.1007/s40259-023-00615-4. Epub 2023 Jul 25.
Hemophilia is characterized by a deficiency in coagulation factors VIII or IX. The general standard of care for severe hemophilia is frequent intravenous recombinant or plasma-derived factor replacement to prevent bleeding. While this treatment is effective in preventing bleeding, frequent infusions are burdensome for patients. Nonadherence to the therapeutic regimen leaves people with hemophilia at risk for spontaneous and traumatic bleeds into joints as well as life-threatening bleeds such as intracranial hemorrhage. The chronicity of the disorder often leads to the formation of target joints, causing long-term pain and impairing mobility. As a monogenic disorder with well-understood genetics, hemophilia is an ideal disorder for implementing innovations in gene therapies. Indeed, recent approvals of two gene therapy products have the potential to shift the hemophilia treatment paradigm. Valoctocogene roxaparvovec and etranacogene dezaparvovec-drlb are gene therapies for hemophilia A and B, respectively. These therapies, given as a single intravenous infusion, may improve patients' quality of life, decreasing treatment burden and resulting in factor expression that virtually eliminates the need for factor replacement. Since both treatments involve viral vectors targeted to the liver, short- and long-term safety and efficacy monitoring involves monitoring liver enzymes to track liver health. Long-term monitoring of efficacy, durability of gene expression, and safety are ongoing. Gene therapy presents a promising new therapeutic option for patients with hemophilia and warrants continued innovation and investigation.
血友病的特征是凝血因子 VIII 或 IX 缺乏。严重血友病的一般标准治疗方法是频繁静脉注射重组或血浆衍生的因子替代物以预防出血。虽然这种治疗方法在预防出血方面非常有效,但频繁的输注会给患者带来负担。不遵守治疗方案会使血友病患者面临自发性和创伤性关节出血以及危及生命的出血(如颅内出血)的风险。该疾病的慢性通常会导致靶关节的形成,导致长期疼痛和活动能力受损。作为一种具有良好遗传理解的单基因疾病,血友病是实施基因治疗创新的理想疾病。事实上,最近批准的两种基因治疗产品有可能改变血友病的治疗模式。Valoctocogene roxaparvovec 和 etranacogene dezaparvovec-drlb 分别是治疗血友病 A 和 B 的基因疗法。这些疗法作为单次静脉输注给药,可能会改善患者的生活质量,减轻治疗负担,并导致因子表达几乎消除了对因子替代物的需求。由于这两种治疗方法都涉及靶向肝脏的病毒载体,因此短期和长期的安全性和疗效监测都需要监测肝酶以跟踪肝脏健康。正在对疗效、基因表达的持久性和安全性进行长期监测。基因治疗为血友病患者提供了一种有前途的新治疗选择,值得继续创新和研究。