Post-transplant lymphoproliferative disorders after allogeneic hematopoietic stem cell transplantation: a case report, meta-analysis, and systematic review.

IF 2.4 3区 医学 Q2 PATHOLOGY Diagnostic Pathology Pub Date : 2024-09-07 DOI:10.1186/s13000-024-01544-8
You-Yuan Su, Ya-Fei Yu, Zhen-Yu Yan, Ya-Jing Zhao, Jian-Wei Lou, Feng Xue, Miao Xu, Qi Feng, Xue-Bin Ji, Xiao-Yuan Dong, Wen Wang, Chuan-Fang Liu, Jun Peng, Xin-Guang Liu
{"title":"Post-transplant lymphoproliferative disorders after allogeneic hematopoietic stem cell transplantation: a case report, meta-analysis, and systematic review.","authors":"You-Yuan Su, Ya-Fei Yu, Zhen-Yu Yan, Ya-Jing Zhao, Jian-Wei Lou, Feng Xue, Miao Xu, Qi Feng, Xue-Bin Ji, Xiao-Yuan Dong, Wen Wang, Chuan-Fang Liu, Jun Peng, Xin-Guang Liu","doi":"10.1186/s13000-024-01544-8","DOIUrl":null,"url":null,"abstract":"<p><strong>Background: </strong>Post-transplant lymphoproliferative disorders (PTLD) are rare but severe complications that occur after solid organ or allogeneic hematopoietic stem cell transplantations (allo-HSCT), with rapid progression and high mortality. Primary central nervous system (CNS)-PTLD are rarely recognized histo-pathologically. In addition, the diagnostic value of the Epstein-Barr virus (EBV) DNA copies in CNS-PTLD remains poorly understood.</p><p><strong>Objectives: </strong>We herein report a case of monomorphic EBV-associated CNS-PTLD (diffuse large B-cell lymphoma, DLBCL) after allo-HSCT and perform a meta-analysis to assess the efficacy of PTLD treatment strategies in recent years.</p><p><strong>Methods: </strong>We present the case report covering clinical manifestations, diagnosis, treatment, and outcomes of a patient with primary CNS-PTLD. Additionally, we include a systematic review and meta-analysis of the clinical characteristics of 431 patients with PTLD after allo-HSCT. We evaluate the main treatment options and outcomes of PTLD management, including rituximab, chemotherapies, and autologous or human leukocyte antigen (HLA)-matched EBV-specific cytotoxic T lymphocyte infusion (EBV-CTLs)/donor lymphocyte infusion (DLI).</p><p><strong>Results: </strong>The meta-analysis revealed an overall response rate of 69.0% for rituximab alone (95% CI: 0.47-0.84), 45.0% for rituximab plus chemotherapies (95% CI: 0.15-0.80), and 91.0% for rituximab plus EBV-CTLs/DLI (95% CI: 0.83-0.96). The complete response (CR) rate after treatments for PTLD was 67.0% (95% CI: 0.56-0.77). Moreover, the 6-month and 1-year overall survival (OS) rate was 64.0% (95% CI: 0.31-0.87) and 49.0% (95% CI: 0.31-0.68), respectively.</p><p><strong>Conclusions: </strong>This case highlighted the urgent need for effective, low-toxic treatment regimens for CNS-PTLD. Our meta-analysis suggested that rituximab combined with EBV-CTLs/DLI could be a favorable strategy for the management of PTLD after allo-HSCT.</p>","PeriodicalId":11237,"journal":{"name":"Diagnostic Pathology","volume":null,"pages":null},"PeriodicalIF":2.4000,"publicationDate":"2024-09-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11380407/pdf/","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Diagnostic Pathology","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1186/s13000-024-01544-8","RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q2","JCRName":"PATHOLOGY","Score":null,"Total":0}
引用次数: 0

Abstract

Background: Post-transplant lymphoproliferative disorders (PTLD) are rare but severe complications that occur after solid organ or allogeneic hematopoietic stem cell transplantations (allo-HSCT), with rapid progression and high mortality. Primary central nervous system (CNS)-PTLD are rarely recognized histo-pathologically. In addition, the diagnostic value of the Epstein-Barr virus (EBV) DNA copies in CNS-PTLD remains poorly understood.

Objectives: We herein report a case of monomorphic EBV-associated CNS-PTLD (diffuse large B-cell lymphoma, DLBCL) after allo-HSCT and perform a meta-analysis to assess the efficacy of PTLD treatment strategies in recent years.

Methods: We present the case report covering clinical manifestations, diagnosis, treatment, and outcomes of a patient with primary CNS-PTLD. Additionally, we include a systematic review and meta-analysis of the clinical characteristics of 431 patients with PTLD after allo-HSCT. We evaluate the main treatment options and outcomes of PTLD management, including rituximab, chemotherapies, and autologous or human leukocyte antigen (HLA)-matched EBV-specific cytotoxic T lymphocyte infusion (EBV-CTLs)/donor lymphocyte infusion (DLI).

Results: The meta-analysis revealed an overall response rate of 69.0% for rituximab alone (95% CI: 0.47-0.84), 45.0% for rituximab plus chemotherapies (95% CI: 0.15-0.80), and 91.0% for rituximab plus EBV-CTLs/DLI (95% CI: 0.83-0.96). The complete response (CR) rate after treatments for PTLD was 67.0% (95% CI: 0.56-0.77). Moreover, the 6-month and 1-year overall survival (OS) rate was 64.0% (95% CI: 0.31-0.87) and 49.0% (95% CI: 0.31-0.68), respectively.

Conclusions: This case highlighted the urgent need for effective, low-toxic treatment regimens for CNS-PTLD. Our meta-analysis suggested that rituximab combined with EBV-CTLs/DLI could be a favorable strategy for the management of PTLD after allo-HSCT.

查看原文
分享 分享
微信好友 朋友圈 QQ好友 复制链接
本刊更多论文
同种异体造血干细胞移植后淋巴增生性疾病:病例报告、荟萃分析和系统综述。
背景:移植后淋巴增生性疾病(PTLD)是实体器官移植或异体造血干细胞移植(allo-HSCT)后出现的罕见但严重的并发症,病情发展迅速,死亡率高。原发性中枢神经系统(CNS)-PTLD 在组织病理学上很少被发现。此外,EBV DNA拷贝在CNS-PTLD中的诊断价值仍鲜为人知:目的:我们在此报告一例异基因造血干细胞移植后的单形性EBV相关中枢神经系统-PTLD(弥漫大B细胞淋巴瘤,DLBCL)病例,并进行荟萃分析以评估近年来PTLD治疗策略的疗效:我们提供了一份病例报告,内容包括一名原发性 CNS-PTLD 患者的临床表现、诊断、治疗和预后。此外,我们还对 431 例异体 HSCT 后 PTLD 患者的临床特征进行了系统回顾和荟萃分析。我们评估了PTLD治疗的主要治疗方案和结果,包括利妥昔单抗、化疗、自体或人类白细胞抗原(HLA)匹配的EBV特异性细胞毒性T淋巴细胞输注(EBV-CTLs)/供体淋巴细胞输注(DLI):荟萃分析显示,单用利妥昔单抗的总反应率为69.0%(95% CI:0.47-0.84),利妥昔单抗加化疗的总反应率为45.0%(95% CI:0.15-0.80),利妥昔单抗加EBV-CTLs/DLI的总反应率为91.0%(95% CI:0.83-0.96)。PTLD治疗后的完全应答(CR)率为67.0%(95% CI:0.56-0.77)。此外,6个月和1年总生存率(OS)分别为64.0%(95% CI:0.31-0.87)和49.0%(95% CI:0.31-0.68):该病例突出表明,中枢神经系统-PTLD急需有效、低毒的治疗方案。我们的荟萃分析表明,利妥昔单抗联合EBV-CTLs/DLI可能是治疗allo-HSCT后PTLD的有利策略。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
求助全文
约1分钟内获得全文 去求助
来源期刊
Diagnostic Pathology
Diagnostic Pathology 医学-病理学
CiteScore
4.60
自引率
0.00%
发文量
93
审稿时长
1 months
期刊介绍: Diagnostic Pathology is an open access, peer-reviewed, online journal that considers research in surgical and clinical pathology, immunology, and biology, with a special focus on cutting-edge approaches in diagnostic pathology and tissue-based therapy. The journal covers all aspects of surgical pathology, including classic diagnostic pathology, prognosis-related diagnosis (tumor stages, prognosis markers, such as MIB-percentage, hormone receptors, etc.), and therapy-related findings. The journal also focuses on the technological aspects of pathology, including molecular biology techniques, morphometry aspects (stereology, DNA analysis, syntactic structure analysis), communication aspects (telecommunication, virtual microscopy, virtual pathology institutions, etc.), and electronic education and quality assurance (for example interactive publication, on-line references with automated updating, etc.).
期刊最新文献
Oral mucosal changes caused by nicotine pouches: case series Pseudoinvasion and squamous metaplasia/morules in colorectal adenomatous polyp: a case report and literature review Model for detecting metastatic deposits in lymph nodes of colorectal carcinoma on digital/ non-WSI images Clinicopathological characteristics and genetic features of young and senior Ewing sarcoma patients Navigating the diagnostic gray zone: a challenging case of pancreatic high-grade neuroendocrine neoplasm
×
引用
GB/T 7714-2015
复制
MLA
复制
APA
复制
导出至
BibTeX EndNote RefMan NoteFirst NoteExpress
×
×
提示
您的信息不完整,为了账户安全,请先补充。
现在去补充
×
提示
您因"违规操作"
具体请查看互助需知
我知道了
×
提示
现在去查看 取消
×
提示
确定
0
微信
客服QQ
Book学术公众号 扫码关注我们
反馈
×
意见反馈
请填写您的意见或建议
请填写您的手机或邮箱
已复制链接
已复制链接
快去分享给好友吧!
我知道了
×
扫码分享
扫码分享
Book学术官方微信
Book学术文献互助
Book学术文献互助群
群 号:481959085
Book学术
文献互助 智能选刊 最新文献 互助须知 联系我们:info@booksci.cn
Book学术提供免费学术资源搜索服务,方便国内外学者检索中英文文献。致力于提供最便捷和优质的服务体验。
Copyright © 2023 Book学术 All rights reserved.
ghs 京公网安备 11010802042870号 京ICP备2023020795号-1