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采用噻替哌和白消安预处理方案的自体干细胞移植成功治疗原发性睾丸淋巴瘤孤立性中枢神经系统复发。

Successful treatment of isolated central nervous system recurrence of primary testicular lymphoma by autologous stem cell transplantation using a conditioning regimen of thiotepa and busulfan.

机构信息

Department of Hematology, Shinko Hospital, Kobe, Japan.

Department of Pathology, Shinko Hospital, Kobe, Japan.

出版信息

J Clin Exp Hematop. 2024 Mar 28;64(1):32-36. doi: 10.3960/jslrt.23039. Epub 2024 Feb 28.

DOI:10.3960/jslrt.23039
PMID:38417873
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11079983/
Abstract

Primary testicular lymphoma (PTL) frequently relapses in the central nervous system (CNS) despite prophylactic intrathecal chemotherapy, and the outcome for CNS recurrence of PTL is very poor. We report a case of isolated CNS recurrence of bilateral PTL. Our patient achieved complete response (CR) after rituximab-combination chemotherapy for PTL. Approximately five years later, isolated CNS recurrence of PTL occurred. Our patient achieved CR again after high-dose methotrexate therapy and autologous stem cell transplantation (ASCT) with a conditioning regimen of thiotepa and busulfan as a consolidation therapy. The secondary failure of platelet recovery, probably caused by busulfan, occurred after the platelet engraftment. Our patient has remained in CR for over three years. The treatment strategy for CNS recurrence of PTL is mainly whole-brain radiotherapy or high-dose methotrexate-based chemotherapy; however, CNS recurrence of PTL may occur again even after achieving CR. ASCT with a conditioning regimen of thiotepa and busulfan is the optimal consolidation therapy for secondary CNS lymphoma. To the best of our knowledge, this is the second reported case of a patient with isolated CNS recurrence of PTL successfully treated by ASCT with a conditioning regimen of thiotepa and busulfan as a consolidation therapy.

摘要

原发性睾丸淋巴瘤(PTL)尽管进行了预防性鞘内化疗,但仍经常在中枢神经系统(CNS)复发,PTL 中枢神经系统复发的预后非常差。我们报告了一例双侧 PTL 孤立性中枢神经系统复发的病例。我们的患者在接受利妥昔单抗联合化疗治疗 PTL 后达到完全缓解(CR)。大约五年后,PTL 出现孤立性中枢神经系统复发。我们的患者在高剂量甲氨蝶呤治疗和以噻替哌和白消安为巩固治疗的自体干细胞移植(ASCT)后再次达到 CR。血小板植入后,可能由于白消安导致血小板恢复的二次失败。我们的患者已持续 CR 超过三年。PTL 中枢神经系统复发的治疗策略主要是全脑放疗或大剂量甲氨蝶呤为基础的化疗;然而,即使达到 CR,PTL 中枢神经系统复发仍可能再次发生。以噻替哌和白消安为巩固治疗的 ASCT 是治疗继发性中枢神经系统淋巴瘤的最佳巩固治疗。据我们所知,这是第二例成功接受以噻替哌和白消安为巩固治疗的 ASCT 治疗的孤立性 PTL 中枢神经系统复发的患者报道。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d303/11079983/a6738a826196/jslrt-64-32-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d303/11079983/c97edee7e8c1/jslrt-64-32-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d303/11079983/4e961b7f3669/jslrt-64-32-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d303/11079983/a6738a826196/jslrt-64-32-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d303/11079983/c97edee7e8c1/jslrt-64-32-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d303/11079983/4e961b7f3669/jslrt-64-32-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d303/11079983/a6738a826196/jslrt-64-32-g003.jpg

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本文引用的文献

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Real-world experiences of CNS-directed chemotherapy followed by autologous stem cell transplantation for secondary CNS involvement in relapsed or refractory diffuse large B-cell lymphoma.中枢神经系统定向化疗后自体干细胞移植治疗复发或难治性弥漫性大B细胞淋巴瘤继发中枢神经系统受累的真实世界经验。
Front Oncol. 2023 Jan 19;12:1071281. doi: 10.3389/fonc.2022.1071281. eCollection 2022.
2
Secondary failure of platelet recovery in patients treated with high-dose thiotepa and busulfan followed by autologous stem cell transplantation.接受大剂量噻替派和白消安治疗后行自体干细胞移植的患者血小板恢复的继发性失败
Int J Hematol. 2020 Nov;112(5):609-613. doi: 10.1007/s12185-020-03007-4. Epub 2020 Sep 26.
3
Durable Survival Outcomes in Primary and Secondary Central Nervous System Lymphoma After High-dose Chemotherapy and Autologous Stem Cell Transplantation Using a Thiotepa, Busulfan, and Cyclophosphamide Conditioning Regimen.
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Clin Lymphoma Myeloma Leuk. 2020 Jul;20(7):468-479. doi: 10.1016/j.clml.2020.02.009. Epub 2020 Feb 20.
4
Autologous Stem Cell Transplantation in Central Nervous System Lymphoma: A Multicenter Retrospective Series and a Review of the Literature.中枢神经系统淋巴瘤自体干细胞移植:一项多中心回顾性系列研究及文献复习。
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Leuk Lymphoma. 2015 Feb;56(2):361-7. doi: 10.3109/10428194.2014.916800. Epub 2014 Jun 27.
9
Central nervous system recurrence of systemic lymphoma in the era of stem cell transplantation--an International Primary Central Nervous System Lymphoma Study Group project.**标题**:干细胞移植时代全身性淋巴瘤中枢神经系统复发——国际原发性中枢神经系统淋巴瘤研究组项目 **摘要**:背景:在接受自体造血干细胞移植(ASCT)巩固治疗的全身性淋巴瘤患者中,中枢神经系统(CNS)复发的发生率很低。我们对在 ASCT 时代,接受过强化疗和 ASCT 的原发性中枢神经系统淋巴瘤(PCNSL)患者的 CNS 复发率和结果进行了评估。 **方法**:我们对国际原发性中枢神经系统淋巴瘤研究组 38 个成员中心的前瞻性数据库进行了回顾性分析,纳入了 2001 年至 2015 年接受强化疗和 ASCT 的 PCNSL 患者。 **结果**:在 201 例患者中,22 例(11%)在 ASCT 后出现了 CNS 复发。中位随访时间为 5.5 年(范围为 0.6 至 14.5 年),5 年和 10 年时无 CNS 事件的累积发生率分别为 79%和 76%。在多变量分析中,诊断时的国际预后指数评分(IPI)(P=0.0004)、强化疗后的 MRI 强化(P=0.003)和 ASCT 后的脑脊液(CSF)寡克隆带(P=0.003)是 CNS 复发的独立预测因素。与未发生 CNS 复发的患者相比,CNS 复发患者的总生存期(OS)和无进展生存期(PFS)显著缩短(P<0.001)。 **结论**:尽管强化疗和 ASCT 可降低 PCNSL 患者的 CNS 复发率,但仍有 10%以上的患者会出现 CNS 复发。CNS 复发与 OS 和 PFS 显著缩短相关。
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