Department of Pediatrics, Children's Hospital Los Angeles, Los Angeles, California.
Department of Pediatrics, University of Southern California, Los Angeles, California.
Cancer. 2020 Feb 1;126(3):593-601. doi: 10.1002/cncr.32552. Epub 2019 Oct 29.
Optimal chemotherapy for treating mixed-phenotype acute leukemia (MPAL) and the role of hematopoietic stem cell transplantation (HSCT) remain uncertain. Major limitations in interpreting available data are MPAL's rarity and the use of definitions other than the currently widely accepted criteria: the World Health Organization 2016 (WHO2016) classification.
To assess the relative efficacy of chemotherapy types for treating pediatric MPAL, the Children's Oncology Group (COG) Acute Leukemia of Ambiguous Lineage Task Force assembled a retrospective cohort of centrally reviewed WHO2016 MPAL cases selected from banking studies for acute lymphoblastic leukemia (ALL) and acute myeloid leukemia (AML). Patients were not treated in COG trials; treatment and outcome data were captured separately. The findings were then integrated with the available, mixed literature to develop a prospective trial in pediatric MPAL.
The central review confirmed that 54 of 70 cases fulfilled WHO2016 criteria for MPAL. ALL induction regimens achieved remission in 72% of the cases (28 of 39), whereas AML regimens achieved remission in 69% (9 of 13). The 5-year event-free survival (EFS) and overall survival (OS) rates for the entire cohort were 72% ± 8% and 77% ± 7%, respectively. EFS and OS were 75% ± 13% and 84% ± 11%, respectively, for those receiving ALL chemotherapy alone without HSCT (n = 21).
The results of the COG MPAL cohort and a literature review suggest that ALL chemotherapy without HSCT may be the preferred initial therapy. A prospective trial within the COG is proposed to investigate this approach; AML chemotherapy and/or HSCT will be reserved for those with treatment failure as assessed by minimal residual disease. Embedded biology studies will provide further insight into MPAL genomics.
治疗混合表型急性白血病(MPAL)的最佳化疗方案以及造血干细胞移植(HSCT)的作用仍不确定。解释现有数据的主要限制是 MPAL 的罕见性,以及使用不同于目前广泛接受的标准的定义:2016 年世界卫生组织(WHO2016)分类。
为了评估治疗儿科 MPAL 的化疗类型的相对疗效,儿童肿瘤学组(COG)急性白血病不确定谱系工作组组建了一个回顾性队列,对从急性淋巴细胞白血病(ALL)和急性髓细胞白血病(AML)的银行研究中选择的经过中央审查的符合 WHO2016 标准的 MPAL 病例进行了回顾性分析。患者未接受 COG 试验治疗;分别采集治疗和结局数据。然后将这些发现与现有混合文献相结合,为儿科 MPAL 制定了一项前瞻性试验。
中央审查证实,70 例中的 54 例符合 WHO2016 标准的 MPAL。ALL 诱导方案使 72%的病例(39 例中的 28 例)缓解,而 AML 方案使 69%的病例(13 例中的 9 例)缓解。整个队列的 5 年无事件生存(EFS)和总生存(OS)率分别为 72%±8%和 77%±7%。单独接受 ALL 化疗且未接受 HSCT 的患者(n=21)的 EFS 和 OS 分别为 75%±13%和 84%±11%。
COG MPAL 队列的结果和文献回顾表明,不进行 HSCT 的 ALL 化疗可能是首选的初始治疗方法。拟在 COG 中开展一项前瞻性试验,以对此方法进行研究;AML 化疗和/或 HSCT 将保留给那些经微小残留病评估治疗失败的患者。嵌入式生物学研究将为 MPAL 基因组学提供进一步的见解。