Division of Hematology and Bone Marrow Transplantation, Fondazione IRCCS Istituto Nazionale dei Tumori, Milan, Italy.
Department of Hematology, Sir Charles Gairdner Hospital, Perth, Australia.
Blood. 2022 Oct 27;140(17):1907-1916. doi: 10.1182/blood.2022015560.
Central nervous system (CNS) relapse of mantle cell lymphoma (MCL) is a rare phenomenon with dismal prognosis, where no standard therapy exists. Since the covalent Bruton tyrosine kinase (BTK) inhibitor ibrutinib is effective in relapsed/refractory MCL and penetrates the blood-brain barrier (BBB), on behalf of Fondazione Italiana Linfomi and European Mantle Cell Lymphoma Network we performed a multicenter retrospective international study to investigate the outcomes of patients treated with ibrutinib or chemoimmunotherapy. In this observational study, we recruited patients with MCL with CNS involvement at relapse who received CNS-directed therapy between 2000 and 2019. The primary objective was to compare the overall survival (OS) of patients treated with ibrutinib or BBB crossing chemotherapy. A propensity score based on a multivariable binary regression model was applied to balance treatment cohorts. Eighty-eight patients were included. The median age at study entry was 65 years (range, 39-87), 76% were males, and the median time from lymphoma diagnosis to CNS relapse was 16 months (range, 1-122). Patients were treated with ibrutinib (n = 29, ibrutinib cohort), BBB crossing chemotherapy (ie, high-dose methotrexate ± cytarabine; n = 29, BBB cohort), or miscellaneous treatments (n = 30, other therapy cohort). Both median OS (16.8 vs 4.4 months; P = .007) and median progression-free survival (PFS) (13.1 vs 3.0 months; P = .009) were superior in the ibrutinib cohort compared with the BBB cohort. Multivariable Cox regression model revealed that ibrutinib therapeutic choice was the strongest independent favorable predictive factor for both OS (hazard ratio [HR], 6.8; 95% confidence interval [CI], 2.2-21.3; P < .001) and PFS (HR, 4.6; 95% CI, 1.7-12.5; P = .002), followed by CNS progression of disease (POD) >24 months from first MCL diagnosis (HR for death, 2.4; 95% CI, 1.1-5.3; P = .026; HR for death or progression, 2.3; 95% CI, 1.1-4.6; P = .023). The addition of intrathecal (IT) chemotherapy to systemic CNS-directed therapy was not associated with superior OS (P = .502) as the morphological variant (classical vs others, P = .118). Ibrutinib was associated with superior survival compared with BBB-penetrating chemotherapy in patients with CNS relapse of MCL and should be considered as a therapeutic option.
中枢神经系统(CNS)套细胞淋巴瘤(MCL)复发是一种罕见的现象,预后不良,目前尚无标准治疗方法。由于共价 Bruton 酪氨酸激酶(BTK)抑制剂伊布替尼对复发/难治性 MCL 有效,并且可以穿透血脑屏障(BBB),代表意大利淋巴瘤基金会和欧洲套细胞淋巴瘤网络,我们进行了一项多中心回顾性国际研究,以调查接受伊布替尼或化疗免疫治疗的患者的结局。在这项观察性研究中,我们招募了在复发时伴有 CNS 受累的 MCL 患者,这些患者在 2000 年至 2019 年间接受了针对 CNS 的治疗。主要目的是比较接受伊布替尼或 BBB 穿透化疗治疗的患者的总生存(OS)。应用基于多变量二项回归模型的倾向评分来平衡治疗队列。共纳入 88 例患者。研究入组时的中位年龄为 65 岁(范围,39-87 岁),76%为男性,从淋巴瘤诊断到 CNS 复发的中位时间为 16 个月(范围,1-122 个月)。患者接受伊布替尼(n=29,伊布替尼组)、BBB 穿透化疗(即高剂量甲氨蝶呤+阿糖胞苷;n=29,BBB 组)或其他治疗(n=30,其他治疗组)。与 BBB 组相比,伊布替尼组的中位 OS(16.8 个月比 4.4 个月;P=0.007)和中位无进展生存(PFS)(13.1 个月比 3.0 个月;P=0.009)均更优。多变量 Cox 回归模型显示,伊布替尼治疗选择是 OS(风险比[HR],6.8;95%置信区间[CI],2.2-21.3;P<0.001)和 PFS(HR,4.6;95%CI,1.7-12.5;P=0.002)的最强独立有利预测因素,其次是 CNS 疾病进展(POD)距首次 MCL 诊断时间>24 个月(死亡 HR,2.4;95%CI,1.1-5.3;P=0.026;死亡或进展 HR,2.3;95%CI,1.1-4.6;P=0.023)。与系统性 CNS 定向治疗联合鞘内(IT)化疗并不能提高 OS(P=0.502),形态学变异(经典型与其他型,P=0.118)也不能提高 OS。与穿透 BBB 的化疗相比,伊布替尼在 CNS 复发的 MCL 患者中与生存获益相关,应被视为一种治疗选择。