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鞘内注射利妥昔单抗治疗儿童CD20阳性中枢神经系统局限性淋巴增殖性疾病:两例报告及文献综述

Intrathecal rituximab in pediatric CD20-positive central nervous system limited lymphoproliferative disease: report of two cases and literature review.

作者信息

Gao Huixia, Zhang Ningning, Zhou Chunju, Jin Ling, Yang Jing, Huang Shuang, Zhang Meng, Li Nan, Wang Tianyou, Duan Yanlong

机构信息

Medical Oncology Department, Pediatric Oncology Center, Beijing Children's Hospital, Capital Medical University, National Center for Children's Health, Beijing, China.

National Key Clinical Discipline of Pediatric Hematology, Key Laboratory of Major Diseases in Children, Ministry of Education, National Key Discipline of Pediatrics (Capital Medical University), Beijing, China.

出版信息

Ann Hematol. 2025 May 5. doi: 10.1007/s00277-025-06386-y.

DOI:10.1007/s00277-025-06386-y
PMID:40320486
Abstract

Isolated central nervous system (CNS) involvement due to posttransplantation proliferative disorder (PTLD) is even rarer, with only a few cases reported in the literature. CNS involvement in patients with mature B-cell non-Hodgkin's (NHL) and PTLD confers a significantly worse prognosis as compared to patients without CNS lymphoma disease. Treatment of CNS lymphoma (CNSL) is challenging due to resistance to conventional cytotoxic and intrathecal chemotherapy. Here, we report the successful use of intrathecal rituximab in two pediatric cases of CD20 + isolated CNSL that had failed to respond to standard chemotherapy, intravenous rituximab and Epstein-Barr virus (EBV)-specific cellular therapy. However, after repeated intrathecal administration of rituximab, both patients' clinical symptoms were alleviated, which has created opportunities for further treatments. We emphasise that intrathecal rituximab may be a safe and promising strategy for the treatment of pediatric patients with CD20 + isolated CNSL. The literature on this topic was also reviewed.

摘要

移植后增殖性疾病(PTLD)导致的孤立性中枢神经系统(CNS)受累更为罕见,文献中仅报道了少数病例。与无中枢神经系统淋巴瘤疾病的患者相比,成熟B细胞非霍奇金淋巴瘤(NHL)和PTLD患者的中枢神经系统受累预后明显更差。由于对传统细胞毒性和鞘内化疗耐药,中枢神经系统淋巴瘤(CNSL)的治疗具有挑战性。在此,我们报告了鞘内注射利妥昔单抗成功治疗两例CD20 + 孤立性CNSL儿科病例,这两例患者对标准化疗、静脉注射利妥昔单抗和爱泼斯坦-巴尔病毒(EBV)特异性细胞治疗均无反应。然而,在反复鞘内注射利妥昔单抗后,两名患者的临床症状均得到缓解,这为进一步治疗创造了机会。我们强调,鞘内注射利妥昔单抗可能是治疗CD20 + 孤立性CNSL儿科患者的一种安全且有前景的策略。本文还对该主题的文献进行了综述。

相似文献

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Intrathecal rituximab in pediatric CD20-positive central nervous system limited lymphoproliferative disease: report of two cases and literature review.鞘内注射利妥昔单抗治疗儿童CD20阳性中枢神经系统局限性淋巴增殖性疾病:两例报告及文献综述
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本文引用的文献

1
Intraventricular Rituximab in Pediatric CD20-positive Refractory Primary Central Nervous System Lymphoma.小儿CD20阳性难治性原发性中枢神经系统淋巴瘤的脑室内利妥昔单抗治疗
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Diagnosis and management of primary central nervous system lymphoma.原发性中枢神经系统淋巴瘤的诊断与管理
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Primary CNS Lymphoma.原发性中枢神经系统淋巴瘤
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Intrathecal rituximab for EBV-associated post-transplant lymphoproliferative disorder with central nervous system involvement unresponsive to intravenous rituximab-based treatments: a prospective study.鞘内注射利妥昔单抗治疗对基于静脉注射利妥昔单抗的治疗无反应的伴有中枢神经系统受累的EB病毒相关移植后淋巴细胞增生性疾病:一项前瞻性研究。
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Intrathecal therapy with rituximab in central nervous system involvement of post-transplant lymphoproliferative disorder.鞘内注射利妥昔单抗治疗移植后淋巴组织增生性疾病中枢神经系统累及。
Leuk Lymphoma. 2013 Mar;54(3):503-6. doi: 10.3109/10428194.2012.718342. Epub 2012 Aug 31.
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Conservative management of post-transplant central nervous system lymphoma.移植后中枢神经系统淋巴瘤的保守治疗。
Int Urol Nephrol. 2013 Aug;45(4):1219-22. doi: 10.1007/s11255-012-0146-8. Epub 2012 Apr 3.
9
Post transplant lymphoproliferative disorders: risk, classification, and therapeutic recommendations.移植后淋巴组织增生性疾病:风险、分类和治疗建议。
Curr Treat Options Oncol. 2012 Mar;13(1):122-36. doi: 10.1007/s11864-011-0177-x.
10
Sustained response to intrathecal rituximab in EBV associated Post-transplant lymphoproliferative disease confined to the central nervous system following haematopoietic stem cell transplant.造血干细胞移植后局限于中枢神经系统的 EBV 相关移植后淋巴组织增生性疾病患者鞘内注射利妥昔单抗后获得持续缓解。
Pediatr Blood Cancer. 2012 Mar;58(3):459-61. doi: 10.1002/pbc.23134. Epub 2011 May 16.