Department of Pediatric Hemato-Immunology, Hôpital Robert Debré and Paris-Diderot University, Paris, France.
Clinica Pediatrica, Università degli Studi di Milano-Bicocca, Ospedale San Gerardo, Monza, Italy.
Biol Blood Marrow Transplant. 2018 Sep;24(9):1848-1855. doi: 10.1016/j.bbmt.2018.05.009. Epub 2018 May 14.
Allogeneic hematopoietic stem cell transplantation (HSCT) is beneficial for pediatric patients with relapsed or (very) high-risk acute lymphoblastic leukemia (ALL) in remission. A total of 1115 consecutive patients were included in the ALL SCT 2003 BFM study and the ALL SCT 2007 I-BFM study and were stratified according to relapse risk (standard versus high versus very high risk of relapse) and donor type (matched sibling versus matched donor versus mismatched donor). A total of 148 patients (60% boys; median age, 8.7 years; B cell precursor ALL, 75%) were transplanted from mismatched donors, which was defined as either less than 9/10 HLA-compatible donors or less than 5/6 unrelated cord blood after myeloablative conditioning regimen (total body irradiation based, 67%) for high relapse risk (HRR; n = 42) or very HRR (VHRR) disease (n = 106). The stem cell source was either bone marrow (n = 31), unmanipulated peripheral stem cells (n = 28), T cell ex vivo depleted peripheral stem cells (n = 59), or cord blood (n = 25). The median follow-up was 5.1 years. The 4-year rates of overall survival (OS) and event-free survival were 56% ± 4% and 52% ± 4%, respectively, for the entire cohort. Patients transplanted from mismatched donors for HRR disease obtained remarkable 4-year OS and event-free survival values of 82% ± 6% and 80% ± 6%, respectively, whereas VHRR patients obtained values of 45% ± 5% and 42% ± 5% (P < .001), respectively. The cumulative incidence of relapse was 29% ± 4% and that of nonrelapse mortality 19% ± 3%. The cumulative incidence of limited and extensive chronic graft-versus-host disease was 13% ± 3% and 15% ± 4%, respectively, among the 120 patients living beyond day 100. Multivariate analysis showed that OS was lower for transplanted VHRR patients (P = .002; hazard ratio [HR], 3.62; 95% confidence interval [CI], 1.60 to 8.20) and for patients beyond second complete remission (CR2) versus first complete remission (P < .001; HR, 3.68; 95% CI, 1.79 to 7.56); relapse occurred more frequently in patients with VHRR disease (P = .026; HR, 3.30; 95% CI, 1.16 to 9.60) and for those beyond CR2 (P = .005; HR, 4.16; 95% CI, 1.52 to 10.59). Nonrelapse mortality was not significantly higher for cytomegalovirus-positive recipients receiving cytomegalovirus-negative grafts (P = .12; HR, 1.96; 95% CI, .84 to 4.58). HSCT with a mismatched donor is feasible in pediatric ALL patients but leads to inferior results compared with HSCT with better matched donors, at least for patients transplanted for VHRR disease. The results are strongly affected by disease status. The main cause of treatment failure is still relapse, highlighting the urgent need for interventional strategies after HSCT for patients with residual leukemia before and/or after transplantation.
异基因造血干细胞移植(HSCT)对缓解后复发或(非常)高危急性淋巴细胞白血病(ALL)的儿科患者有益。共有 1115 例连续患者被纳入 ALL SCT 2003 BFM 研究和 ALL SCT 2007 I-BFM 研究,并根据复发风险(标准、高、非常高复发风险)和供体类型(匹配的同胞、匹配的供体、不匹配的供体)进行分层。共有 148 例患者(60%为男孩;中位年龄 8.7 岁;B 细胞前体 ALL,75%)从不匹配的供体进行移植,这定义为在高复发风险(HRR;n=42)或非常 HRR(VHRR)疾病(n=106)的情况下,接受不超过 9/10 HLA 相容供体或不超过骨髓移植后 6 个无关脐带血的供体(基于全身照射的 67%)。干细胞来源为骨髓(n=31)、未经处理的外周血干细胞(n=28)、体外 T 细胞耗尽的外周血干细胞(n=59)或脐带血(n=25)。中位随访时间为 5.1 年。整个队列的总生存(OS)和无事件生存(EFS)率分别为 56%±4%和 52%±4%。对于接受 HRR 疾病的患者,从不匹配供体进行 HSCT 获得了显著的 4 年 OS 和 EFS 值,分别为 82%±6%和 80%±6%,而 VHRR 患者的相应值为 45%±5%和 42%±5%(P<.001)。复发累积发生率为 29%±4%,非复发相关死亡率为 19%±3%。在 120 例存活超过 100 天的患者中,局限性和广泛性慢性移植物抗宿主病的累积发生率分别为 13%±3%和 15%±4%。多变量分析显示,VHRR 患者的 OS 较低(P=.002;风险比[HR],3.62;95%置信区间[CI],1.60 至 8.20),且第二次完全缓解(CR2)后患者的 OS 低于首次完全缓解(P<.001;HR,3.68;95% CI,1.79 至 7.56);VHRR 疾病患者的复发频率更高(P=.026;HR,3.30;95% CI,1.16 至 9.60),且 CR2 后患者的复发频率更高(P=.005;HR,4.16;95% CI,1.52 至 10.59)。对于接受 CMV 阴性移植物的 CMV 阳性受者,非复发相关死亡率没有显著增加(P=.12;HR,1.96;95% CI,0.84 至 4.58)。HSCT 采用不匹配供体在儿科 ALL 患者中是可行的,但与更好匹配的供体进行 HSCT 相比,结果较差,至少对于接受 VHRR 疾病 HSCT 的患者而言。结果受到疾病状态的强烈影响。治疗失败的主要原因仍然是复发,这突出表明需要在移植前后对残留白血病患者进行移植后的干预策略。