Division of Hematology-Oncology, Department of Medicine.
Division of Pediatric Hematology-Oncology, Department of Pediatrics, and.
Blood Adv. 2020 Dec 22;4(24):6240-6249. doi: 10.1182/bloodadvances.2020002977.
Acquired hemophilia A (AHA) is a rare bleeding disorder in which acquired autoantibodies to endogenous factor VIII (FVIII) decrease FVIII activity and lead to a bleeding phenotype. A substantial majority of individuals who develop AHA present with severe bleeding. Effective treatment requires both immunosuppressive therapy and prompt hemostatic treatment. Bleeding is commonly treated with bypassing agents (BPAs) such as recombinant activated FVII (rFVIIa) or activated prothrombin complex concentrates Disadvantages to BPAs include the inability to monitor response with standard laboratory assays, inconsistent hemostatic efficacy, and thrombosis. Recombinant porcine FVIII (rpFVIII: Obizur, Baxter, Deerfield, IL) was approved by the US Food and Drug Administration (FDA) for bleed treatment in AHA in 2014, and has the advantage of laboratory monitoring of FVIII activity levels and known hemostatic efficacy in the presence of anti-human FVIII inhibitors and after failure of BPAs. Using an algorithm-based approach, rpFVIII has been used to successfully treat 18 patients with AHA at our center with substantially lower doses than the current FDA-recommended dosing. Additionally, data from our cohort show that the preexposure anti-porcine Bethesda titer does not reliably predict the clinical response to rpFVIII treatment and is not correlated with the anti-human Bethesda titer. We also present data showing lower total rpFVIII use for initial bleed resolution when rpVIII is used upfront, as compared with use as rescue therapy. We validated our dosing algorithm, which uses much lower than FDA-recommended doses with 14 more patients than in our previously reported patient series.
获得性血友病 A(AHA)是一种罕见的出血性疾病,其中获得性自身抗体针对内源性因子 VIII(FVIII)降低 FVIII 活性并导致出血表型。绝大多数发生 AHA 的个体都有严重的出血。有效的治疗需要免疫抑制治疗和及时的止血治疗。出血通常用旁路制剂(BPAs)治疗,如重组活化 FVII(rFVIIa)或活化的凝血酶原复合物浓缩物。BPAs 的缺点包括无法用标准实验室检测来监测反应、止血效果不一致和血栓形成。重组猪 FVIII(rpFVIII:Obizur,Baxter,Deerfield,IL)于 2014 年获得美国食品和药物管理局(FDA)批准用于 AHA 的出血治疗,其优点是可以监测 FVIII 活性水平,并在存在抗人 FVIII 抑制剂和 BPAs 治疗失败后具有已知的止血效果。我们中心采用基于算法的方法,使用 rpFVIII 成功治疗了 18 名 AHA 患者,剂量明显低于目前 FDA 推荐的剂量。此外,我们队列的数据表明,暴露前抗猪贝塞斯达滴度不能可靠地预测 rpFVIII 治疗的临床反应,也与抗人贝塞斯达滴度无关。我们还提供了数据,显示当 rpVIII 作为一线治疗时,初始出血缓解时的总 rpFVIII 使用量较低,与作为挽救治疗时相比。我们验证了我们的剂量算法,该算法使用的剂量远低于 FDA 推荐的剂量,比我们之前报告的患者系列多 14 例。