Guy's and St Thomas' Hospital Foundation Trust, Westminster Bridge Rd, London, SE1 7EH, UK.
Radboud University Medical Centre, Nijmegen, Netherlands.
Ann Hematol. 2020 Jun;99(6):1177-1191. doi: 10.1007/s00277-020-04002-9. Epub 2020 Mar 20.
Myelofibrosis is a BCR-ABL1-negative myeloproliferative neoplasm characterized by anemia, progressive splenomegaly, extramedullary hematopoiesis, bone marrow fibrosis, constitutional symptoms, leukemic progression, and shortened survival. Constitutive activation of the Janus kinase/signal transducers and activators of transcription (JAK-STAT) pathway, and other cellular pathways downstream, leads to myeloproliferation, proinflammatory cytokine expression, and bone marrow remodeling. Transplant is the only curative option for myelofibrosis, but high rates of morbidity and mortality limit eligibility. Several prognostic models have been developed to facilitate treatment decisions. Until the recent approval of fedratinib, a JAK2 inhibitor, ruxolitinib was the only available JAK inhibitor for treatment of intermediate- or high-risk myelofibrosis. Ruxolitinib reduces splenomegaly to some degree in almost all treated patients; however, many patients cannot tolerate ruxolitinib due to dose-dependent drug-related cytopenias, and even patients with a good initial response often develop resistance to ruxolitinib after 2-3 years of therapy. Currently, there is no consensus definition of ruxolitinib failure. Until fedratinib approval, strategies to overcome ruxolitinib resistance or intolerance were mainly different approaches to continued ruxolitinib therapy, including dosing modifications and ruxolitinib rechallenge. Fedratinib and two other JAK2 inhibitors in later stages of clinical development, pacritinib and momelotinib, have been shown to induce clinical responses and improve symptoms in patients previously treated with ruxolitinib. Fedratinib induces robust spleen responses, and pacritinib and momelotinib may have preferential activity in patients with severe cytopenias. Reviewed here are strategies to ameliorate ruxolitinib resistance or intolerance, and outcomes of clinical trials in patients with myelofibrosis receiving second-line JAK inhibitors after ruxolitinib treatment.
骨髓纤维化是一种 BCR-ABL1 阴性骨髓增殖性肿瘤,其特征为贫血、进行性脾肿大、髓外造血、骨髓纤维化、全身症状、白血病进展和生存期缩短。Janus 激酶/信号转导和转录激活因子(JAK-STAT)通路的组成性激活和下游其他细胞通路导致骨髓增殖、促炎细胞因子表达和骨髓重塑。移植是骨髓纤维化的唯一治愈方法,但发病率和死亡率高限制了其适用范围。已经开发了几种预后模型来促进治疗决策。直到最近 fedratinib(一种 JAK2 抑制剂)获得批准,ruxolitinib 是唯一可用于治疗中高危骨髓纤维化的 JAK 抑制剂。ruxolitinib 在几乎所有接受治疗的患者中都在一定程度上减少了脾肿大;然而,由于剂量依赖性药物相关血细胞减少,许多患者无法耐受 ruxolitinib,即使是对初始反应良好的患者,在接受 2-3 年治疗后也经常对 ruxolitinib 产生耐药性。目前,对于 ruxolitinib 失败还没有共识定义。在 fedratinib 获得批准之前,克服 ruxolitinib 耐药或不耐受的策略主要是不同的 ruxolitinib 继续治疗方法,包括剂量调整和 ruxolitinib 再挑战。fedratinib 和另外两种处于临床开发后期的 JAK2 抑制剂 pacritinib 和 momelotinib 已被证明可诱导先前接受 ruxolitinib 治疗的患者产生临床反应并改善症状。Fedratinib 可诱导强烈的脾脏反应,而 pacritinib 和 momelotinib 可能对严重血细胞减少的患者具有优先活性。本文回顾了在 ruxolitinib 治疗后接受二线 JAK 抑制剂治疗的骨髓纤维化患者中,改善 ruxolitinib 耐药或不耐受的策略以及临床试验结果。