目的 分析儿童伯基特淋巴瘤(Burkitt's lymphoma,BL)的临床特点、化疗疗效,以及利妥昔单抗治疗对BL患儿预后的影响。 方法 回顾性收集62例BL患儿的临床资料,对BL患儿的临床特点、疗效及预后相关因素进行分析,采用Cox回归分析BL患儿预后不良的相关因素。根据是否应用利妥昔单抗治疗将晚期(Ⅲ/Ⅳ期)BL患儿分为化疗联合利妥昔单抗组和单纯化疗组,比较两组预后情况。 结果 62例患儿发病时中位年龄5(范围1~14)岁,男58例(94%),女4例(6%)。原发部位为腹腔者41例(66%),头颈部者16例(26%)。Ⅰ、Ⅱ、Ⅲ、Ⅳ期患儿分别为1例(2%)、8例(13%)、33例(53%)、20例(32%)。中位随访时间29个月,进展/复发患儿15例(24%),3年总生存率、无事件生存率分别为82.8%±5.2%、77.3%±5.8%。Ⅲ/Ⅳ期患儿中,化疗联合利妥昔单抗组(n=16)与单纯化疗组(n=30)3年总生存率分别为93.3%±6.4%、65.6%±9.9%,差异有统计学意义(P=0.042);3年无事件生存率分别为86.2%±9.1%、61.8%±10.1%,差异无统计学意义(P>0.05)。Cox回归分析结果显示:中枢神经系统侵犯、乳酸脱氢酶水平>1 000 U/L、早期未完全缓解为BL患儿预后不良的相关因素(P<0.05)。 结论 化疗联合利妥昔单抗治疗能改善Ⅲ、Ⅳ期BL患儿预后;中枢神经系统侵犯、乳酸脱氢酶水平升高、早期未完全缓解可能提示BL患儿预后不良。 引用格式:中国当代儿科杂志,2022,24(5):561-565
Abstract
Objective To study the clinical features and chemotherapy response of Burkitt's lymphoma (BL) in children and the influence of rituximab on the prognosis of children with BL. Methods A retrospective analysis was performed for the medical data of 62 children with BL, including clinical features, therapeutic efficacy, and prognostic factors. The Cox regression model was used to identify the factors associated with poor prognosis in children with BL. According to whether rituximab was used, the children with advanced (stage III/IV) BL were divided into two groups: chemotherapy plus rituximab and chemotherapy alone. The prognosis was compared between the two groups. Results For these 62 children, the median age of onset was 5 years (range 1-14 years), and there were 58 boys (94%) and 4 girls (6%). The primary site was abdominal cavity in 41 children (66%), and head and neck in 16 children (26%). There were 1 child with stage I BL (2%), 8 with stage II BL (13%), 33 with stage III BL (53%), and 20 with stage IV BL (32%). The median follow-up time was 29 months, with progression/recurrence observed in 15 children (24%), and the 3-year overall survival (OS) rate and event-free survival (EFS) rate were 82.8%±5.2% and 77.3%±5.8%, respectively. For the children with stage III/IV BL, there was a significant difference in the 3-year the OS rate between the chemotherapy plus rituximab group (16 children) and the chemotherapy alone group (30 children) (93.3%±6.4% vs 65.6%±9.9%, P=0.042), while there was no significant difference in the 3-year EFS rate between the two groups (86.2%±9.1% vs 61.8%±10.1%, P>0.05). The Cox regression analysis showed that central nervous system involvement, lactate dehydrogenase >1 000 U/L, and early incomplete remission were the factors associated with poor prognosis (P<0.05). Conclusions Chemotherapy combined with rituximab can improve the prognosis of children with stage III/IV BL. Central nervous system involvement, elevated lactate dehydrogenase level, and early incomplete remission may indicate a poor prognosis in children with BL. Citation:Chinese Journal of Contemporary Pediatrics, 2022, 24(5): 561-565
关键词
伯基特淋巴瘤 /
利妥昔单抗 /
预后 /
儿童
Key words
Burkitt's lymphoma /
Rituximab /
Prognosis /
Child
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参考文献
1 Kaatsch P. Epidemiology of childhood cancer[J]. Cancer Treat Rev, 2010, 36(4): 277-285. PMID: 20231056. DOI: 10.1016/j.ctrv.2010.02.003.
2 中国抗癌协会淋巴瘤专业委员会, 中国医师协会肿瘤医师分会, 中国医疗保健国际交流促进会肿瘤内科分会. 中国淋巴瘤治疗指南(2021年版)[J]. 中华肿瘤杂志, 2021, 43(7): 707-735. PMID: 34289565. DOI: 10.3760/cma.j.cn112152-20210516-00382.
3 Zhen Z, Zhu J, Wang J, et al. Rituximab is highly effective in children and adolescents with Burkitt lymphoma in risk groups R2 to R4[J]. PediatrHematol Oncol, 2020, 37(6): 489-499. PMID: 32364412. DOI: 10.1080/08880018.2020.1759741.
4 张梦, 金玲, 杨菁, 等. 儿童伯基特淋巴瘤186例临床特征及疗效分析[J]. 中华儿科杂志, 2018, 56(8): 605-610. PMID: 30078243. DOI: 10.3760/cma.j.issn.0578-1310.2018.08.010.
5 中华医学会儿科学分会血液学组, 中国抗癌协会儿科专业委员会, 《中华儿科杂志》编辑委员会. 儿童非霍奇金淋巴瘤诊疗建议[J]. 中华儿科杂志, 2011, 49(3): 186-192. PMID: 21575366. DOI: 10.3760/cma.j.issn.0578-1310.2011.03.006.
6 Oliveira MCL, Sampaio KC, Brito AC, et al. 30 years of experience with non-Hodgkin lymphoma in children and adolescents: a retrospective cohort study[J]. Rev Assoc Med Bras, 2020, 66(1): 25-30. PMID: 32130377. DOI: 10.1590/1806-9282.66.1.25.
7 Cairo MS, Sposto R, Gerrard M, et al. Advanced stage, increased lactate dehydrogenase, and primary site, but not adolescent age (≥ 15 years), are associated with an increased risk of treatment failure in children and adolescents with mature B-cell non-Hodgkin's lymphoma: results of the FAB LMB 96 study[J]. J Clin Oncol, 2012, 30(4): 387-393. PMID: 22215753. PMCID: PMC3269965. DOI: 10.1200/JCO.2010.33.3369.
8 Maschan A, Myakova N, Aleinikova O, et al. Rituximab and reduced-intensity chemotherapy in children and adolescents with mature B-cell lymphoma: interim results for 231 patients enrolled in the second Russian-Belorussian multicentre study B-NHL-2010M[J]. Br J Haematol, 2019, 186(3): 477-483. PMID: 31069789. DOI: 10.1111/bjh.15944.
9 李永新, 尹青松, 艾昊, 等. 利妥昔单抗联合改良NHL-BFM-90方案对儿童及青少年伯基特淋巴瘤的远期疗效[J]. 中华医学杂志, 2019, 99(8): 605-610. PMID: 30818930. DOI: 10.3760/cma.j.issn.0376-2491.2019.08.008.
10 Evens AM, Danilov A, Jagadeesh D, et al. Burkitt lymphoma in the modern era: real-world outcomes and prognostication across 30 US cancer centers[J]. Blood, 2021, 137(3): 374-386. PMID: 32663292. PMCID: PMC8765121. DOI: 10.1182/blood.2020006926.
11 李艳, 弓晓媛, 赵邢力, 等. 利妥昔单抗联合短周期、高强度方案治疗Burkitt白血病疗效及安全性分析[J]. 中华血液学杂志, 2020, 41(6): 502-505. PMID: 32654465. PMCID: PMC7378285. DOI: 10.3760/cma.j.issn.0253-2727.2020.06.012.
12 Goldman S, Smith L, Anderson JR, et al. Rituximab and FAB/LMB 96 chemotherapy in children with Stage III/IV B-cell non-Hodgkin lymphoma: a Children's Oncology Group report[J]. Leukemia, 2013, 27(5): 1174-1177. PMID: 22940833. PMCID: PMC4539148. DOI: 10.1038/leu.2012.255.
13 Minard-Colin V, Aupérin A, Pillon M, et al. Rituximab for high-risk, mature B-cell non-Hodgkin's lymphoma in children[J]. N Engl J Med, 2020, 382(23): 2207-2219. PMID: 32492302. PMCID: PMC7720281. DOI: 10.1056/NEJMoa1915315.
14 张梦, 金玲, 杨菁, 等. 改良LMB 89 C组方案治疗儿童高危伯基特淋巴瘤172例临床分析[J]. 中华血液学杂志, 2019, 40(8): 633-638. PMID: 31495128. PMCID: PMC7342876. DOI: 10.3760/cma.j.issn.0253-2727.2019.08.002.
15 Moleti ML, Testi AM, Foà R. Treatment of relapsed/refractory paediatric aggressive B-cell non-Hodgkin lymphoma[J]. Br J Haematol, 2020, 189(5): 826-843. PMID: 32141616. DOI: 10.1111/bjh.16461.
16 黄爽, 金玲, 杨菁, 等. 复发儿童B细胞淋巴瘤19例临床病理特征及治疗预后分析[J]. 中华儿科杂志, 2017, 55(10): 748-753. PMID: 29050112. DOI: 10.3760/cma.j.issn.0578-1310.2017.10.007.
17 Cairo M, Auperin A, Perkins SL, et al. Overall survival of children and adolescents with mature B cell non-Hodgkin lymphoma who had refractory or relapsed disease during or after treatment with FAB/LMB 96: a report from the FAB/LMB 96 study group[J]. Br J Haematol, 2018, 182(6): 859-869. PMID: 29984828. PMCID: PMC6128751. DOI: 10.1111/bjh.15491.