Chidamide-maintained long-term clinical remission with persistent T-Cell clone in refractory T-LGL leukemia-associated pure red cell aplasia: A six-year follow-up
{"title":"Chidamide-maintained long-term clinical remission with persistent T-Cell clone in refractory T-LGL leukemia-associated pure red cell aplasia: A six-year follow-up","authors":"Huan Tao, Nenggang Jiang, Yongqian Jia","doi":"10.1016/j.lrr.2026.100605","DOIUrl":null,"url":null,"abstract":"<div><div>Secondary pure red cell aplasia (PRCA) is one of the most common cytopenic complications in T-cell large granular lymphocyte (T-LGL) leukemia and requires therapeutic intervention. Refractory T-LGL-associated PRCA lacks a unified salvage regimen, and effective targeted agents remain scarce. In recent years, epigenetic modifications have been implicated in the pathogenesis of T-LGL leukemia, yet relevant clinical evidence is limited. We report a case of refractory T-LGL leukemia complicated by PRCA that was resistant to cyclosporine (CsA). Then the patient achieved rapid disease control with chidamide plus thalidomide in the early phase, and has maintained long-term remission for six years on chidamide monotherapy after thalidomide discontinuation. Notably, clonal T-cell receptor (TCR) variable β-chain (Vβ) 3 persisted on flow cytometry throughout the treatment course. As an epigenetic modulator, chidamide represents a more convenient and promising therapeutic strategy for managing T-LGL leukemia, prompting us to present this case.</div></div>","PeriodicalId":38435,"journal":{"name":"Leukemia Research Reports","volume":"26 ","pages":"Article 100605"},"PeriodicalIF":0.8000,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Leukemia Research Reports","FirstCategoryId":"1085","ListUrlMain":"https://www.sciencedirect.com/science/article/pii/S2213048926000452","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2026/9/8 0:00:00","PubModel":"Epub","JCR":"Q4","JCRName":"HEMATOLOGY","Score":null,"Total":0}
引用次数: 0
Abstract
Secondary pure red cell aplasia (PRCA) is one of the most common cytopenic complications in T-cell large granular lymphocyte (T-LGL) leukemia and requires therapeutic intervention. Refractory T-LGL-associated PRCA lacks a unified salvage regimen, and effective targeted agents remain scarce. In recent years, epigenetic modifications have been implicated in the pathogenesis of T-LGL leukemia, yet relevant clinical evidence is limited. We report a case of refractory T-LGL leukemia complicated by PRCA that was resistant to cyclosporine (CsA). Then the patient achieved rapid disease control with chidamide plus thalidomide in the early phase, and has maintained long-term remission for six years on chidamide monotherapy after thalidomide discontinuation. Notably, clonal T-cell receptor (TCR) variable β-chain (Vβ) 3 persisted on flow cytometry throughout the treatment course. As an epigenetic modulator, chidamide represents a more convenient and promising therapeutic strategy for managing T-LGL leukemia, prompting us to present this case.