{"title":"HSCT后复发ebv相关T/ nk细胞淋巴增生性疾病dli诱导缓解：1例报告","authors":"Toru Nagata, Hirohito Kubota, Itaru Kato, Kazushi Izawa, Ryo Akazawa, Tomoyasu Jo, Yasuyuki Arai, Shohei Azumi, Tatsuya Kamitori, Satoshi Saida, Katsutsugu Umeda, Ken-Ichi Imadome, Takahiro Yasumi, Hidefumi Hiramatsu, Junko Takita","doi":"10.1007/s12185-025-03965-7","DOIUrl":null,"url":null,"abstract":"<p><strong>Background: </strong>Epstein-Barr virus (EBV)-associated T/natural killer (NK)-cell lymphoproliferative diseases (T/NK-LPDs) are characterized by clonal proliferation of EBV-infected T or NK cells, and the only curative treatment for progressive T/NK-LPDs is allogeneic hematopoietic stem cell transplantation (HSCT). There are limited reports concerning optimal management strategies for disease relapse after HSCT in patients with EBV-associated T/NK-LPDs.</p><p><strong>Case presentation: </strong>A 2-year-old girl was diagnosed with severe mosquito bite allergy, a cutaneous form of EBV-associated T/NK-LPD. Following HSCT from her human leukocyte antigen-matched sister, complete donor-type chimerism was achieved and whole blood EBV-DNA load became undetectable. However, the whole blood EBV-DNA load became elevated again and donor type chimerism decreased. Donor lymphocyte infusion (DLI) was performed to control the disease relapse. A complete virological response and complete donor-type chimerism were attained after four doses of DLI. No adverse effects were observed during the treatment course.</p><p><strong>Conclusions: </strong>This case highlights the potential of DLI as an alternative therapeutic approach to re-transplantation for recurrent EBV-associated T/NK-LPDs.</p>","PeriodicalId":13992,"journal":{"name":"International Journal of Hematology","volume":" ","pages":""},"PeriodicalIF":1.7000,"publicationDate":"2025-03-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"DLI-induced remission in recurrent EBV-associated T/NK-cell lymphoproliferative disease following HSCT: a case report.\",\"authors\":\"Toru Nagata, Hirohito Kubota, Itaru Kato, Kazushi Izawa, Ryo Akazawa, Tomoyasu Jo, Yasuyuki Arai, Shohei Azumi, Tatsuya Kamitori, Satoshi Saida, Katsutsugu Umeda, Ken-Ichi Imadome, Takahiro Yasumi, Hidefumi Hiramatsu, Junko Takita\",\"doi\":\"10.1007/s12185-025-03965-7\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><strong>Background: </strong>Epstein-Barr virus (EBV)-associated T/natural killer (NK)-cell lymphoproliferative diseases (T/NK-LPDs) are characterized by clonal proliferation of EBV-infected T or NK cells, and the only curative treatment for progressive T/NK-LPDs is allogeneic hematopoietic stem cell transplantation (HSCT). There are limited reports concerning optimal management strategies for disease relapse after HSCT in patients with EBV-associated T/NK-LPDs.</p><p><strong>Case presentation: </strong>A 2-year-old girl was diagnosed with severe mosquito bite allergy, a cutaneous form of EBV-associated T/NK-LPD. Following HSCT from her human leukocyte antigen-matched sister, complete donor-type chimerism was achieved and whole blood EBV-DNA load became undetectable. However, the whole blood EBV-DNA load became elevated again and donor type chimerism decreased. Donor lymphocyte infusion (DLI) was performed to control the disease relapse. A complete virological response and complete donor-type chimerism were attained after four doses of DLI. No adverse effects were observed during the treatment course.</p><p><strong>Conclusions: </strong>This case highlights the potential of DLI as an alternative therapeutic approach to re-transplantation for recurrent EBV-associated T/NK-LPDs.</p>\",\"PeriodicalId\":13992,\"journal\":{\"name\":\"International Journal of Hematology\",\"volume\":\" \",\"pages\":\"\"},\"PeriodicalIF\":1.7000,\"publicationDate\":\"2025-03-21\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"International Journal of Hematology\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.1007/s12185-025-03965-7\",\"RegionNum\":4,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q3\",\"JCRName\":\"HEMATOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"International Journal of Hematology","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1007/s12185-025-03965-7","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q3","JCRName":"HEMATOLOGY","Score":null,"Total":0}
引用次数: 0
摘要
背景：eb病毒（EBV）相关的T/NK细胞淋巴增殖性疾病（T/NK- lpd）的特征是EBV感染的T或NK细胞的克隆性增殖，治疗进展性T/NK- lpd的唯一治疗方法是同种异体造血干细胞移植（HSCT）。关于ebv相关T/ nk - lpd患者HSCT后疾病复发的最佳管理策略的报道有限。病例介绍：一名2岁女孩被诊断患有严重的蚊虫叮咬过敏，这是ebv相关T/NK-LPD的一种皮肤形式。从她的人类白细胞抗原匹配的姐妹中进行HSCT后，实现了完全的供体型嵌合，全血EBV-DNA载量无法检测到。然而，全血EBV-DNA载量再次升高，供体型嵌合减少。供体淋巴细胞输注（DLI）控制疾病复发。四剂DLI后获得了完全的病毒学应答和完全的供体型嵌合。治疗过程中未见不良反应。结论：该病例强调了DLI作为复发性ebv相关T/ nk - lpd再移植的替代治疗方法的潜力。
DLI-induced remission in recurrent EBV-associated T/NK-cell lymphoproliferative disease following HSCT: a case report.
Background: Epstein-Barr virus (EBV)-associated T/natural killer (NK)-cell lymphoproliferative diseases (T/NK-LPDs) are characterized by clonal proliferation of EBV-infected T or NK cells, and the only curative treatment for progressive T/NK-LPDs is allogeneic hematopoietic stem cell transplantation (HSCT). There are limited reports concerning optimal management strategies for disease relapse after HSCT in patients with EBV-associated T/NK-LPDs.
Case presentation: A 2-year-old girl was diagnosed with severe mosquito bite allergy, a cutaneous form of EBV-associated T/NK-LPD. Following HSCT from her human leukocyte antigen-matched sister, complete donor-type chimerism was achieved and whole blood EBV-DNA load became undetectable. However, the whole blood EBV-DNA load became elevated again and donor type chimerism decreased. Donor lymphocyte infusion (DLI) was performed to control the disease relapse. A complete virological response and complete donor-type chimerism were attained after four doses of DLI. No adverse effects were observed during the treatment course.
Conclusions: This case highlights the potential of DLI as an alternative therapeutic approach to re-transplantation for recurrent EBV-associated T/NK-LPDs.
期刊介绍：
The International Journal of Hematology, the official journal of the Japanese Society of Hematology, has a long history of publishing leading research in hematology. The journal comprises articles that contribute to progress in research not only in basic hematology but also in clinical hematology, aiming to cover all aspects of this field, namely, erythrocytes, leukocytes and hematopoiesis, hemostasis, thrombosis and vascular biology, hematological malignancies, transplantation, and cell therapy. The expanded [Progress in Hematology] section integrates such relevant fields as the cell biology of stem cells and cancer cells, and clinical research in inflammation, cancer, and thrombosis. Reports on results of clinical trials are also included, thus contributing to the aim of fostering communication among researchers in the growing field of modern hematology. The journal provides the best of up-to-date information on modern hematology, presenting readers with high-impact, original work focusing on pivotal issues.
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