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

IF 0.8 Q4 HEMATOLOGY
Leukemia Research Reports Pub Date : 2026-01-01 Epub Date: 2026-09-08 DOI:10.1016/j.lrr.2026.100605
Huan Tao, Nenggang Jiang, Yongqian Jia
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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.
难治性T-LGL白血病相关纯红细胞发育不全患者持续t细胞克隆的奇达胺维持长期临床缓解:6年随访
继发性纯红细胞发育不全(PRCA)是t细胞大颗粒淋巴细胞(T-LGL)白血病中最常见的细胞减少性并发症之一,需要治疗干预。难治性t - lgl相关的PRCA缺乏统一的挽救方案,有效的靶向药物仍然稀缺。近年来,表观遗传修饰与T-LGL白血病的发病机制有关,但相关临床证据有限。我们报告一例难治性T-LGL白血病合并PRCA耐环孢素(CsA)。然后患者在早期使用奇达胺联合沙利度胺实现了快速的疾病控制,停药后继续使用奇达胺单药维持了6年的长期缓解。值得注意的是,在整个治疗过程中,流式细胞术显示克隆t细胞受体(TCR)可变β链(Vβ) 3持续存在。作为一种表观遗传调节剂,奇达胺代表了一种更方便和有希望的治疗T-LGL白血病的策略,促使我们提出这个病例。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
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来源期刊
Leukemia Research Reports
Leukemia Research Reports Medicine-Oncology
CiteScore
1.70
自引率
0.00%
发文量
70
审稿时长
23 weeks
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