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硫唑嘌呤治疗后并发骨髓再生障碍的肝脾γδT 细胞淋巴瘤一例:治疗过程及文献复习

英文原题:A Case of Hepatosplenic Gamma Delta T Cell Lymphoma With Concomitant Bone Marrow Aplasia Following Azathioprine Therapy, Treatment Course and Review of the Literature.

PubMed 2025/09/01(内容时间) Cancer Rep (Hoboken) Q3 · IF 2.5(JCR 2025)

研究概要

据我们所知,这是首例报道的HSTCL与再生障碍性贫血同时发生且既往有硫唑嘌呤暴露的病例。其潜在病理生理机制可能是多因素的,涉及免疫介导的抑制和药物诱导的毒性。临床医生对于接受硫唑嘌呤治疗期间出现不明原因全血细胞减少的患者,应高度警惕淋巴瘤的可能。

研究思路结论见上方概要

肝脾T细胞淋巴瘤(HSTCL)是一种罕见且侵袭性强的外周T细胞淋巴瘤亚型,预后较差,主要影响年轻成年男性,其中许多人具有免疫抑制或自身免疫性疾病的背景。病例:我们报告一例27岁男性,既往接受过硫唑嘌呤治疗,出现全血细胞减少。骨髓活检显示严重再生障碍,伴有γ-δ T淋巴细胞(Tγδ)部分浸润。通过肝活检确立了HSTCL的明确诊断。患者接受了异环磷酰胺、卡铂、依托泊苷(ICE方案)的诱导化疗,但仍持续全血细胞减少,遂进行异基因干细胞移植。治疗后六十个月,他仍处于完全缓解状态。

展开英文摘要原文

BACKGROUND: Hepatosplenic T-cell lymphoma (HSTCL) is a rare and aggressive subtype of peripheral T-cell lymphoma with a poor prognosis, primarily affecting young adult males, many with a background of immunosuppression or autoimmune disease. CASE: We present the case of a 27-year-old male previously treated with azathioprine who developed pancytopenia. Bone marrow biopsy revealed severe aplasia with partial infiltration by gamma-delta T-lymphocytes (Tγδ). A definitive diagnosis of HSTCL was established through liver biopsy. The patient received induction chemotherapy with the Ifosfamide, Carboplatin, Etoposide (ICE regimen) but remained pancytopenic, prompting allogeneic stem cell transplantation. Sixty months post-treatment, he remains in complete remission. CONCLUSION: To our knowledge, this is the first reported case of HSTCL presenting concurrently with aplastic anemia in the context of prior azathioprine exposure. The underlying pathophysiology is likely multifactorial, involving both immune-mediated suppression and drug-induced toxicity. Clinicians should maintain a high index of suspicion for lymphoma in patients developing unexplained pancytopenia while on azathioprine therapy.

论文信息

作者
Younis R、Hauspurg H、Voss A
单位
Department of Oncology and Haematology, Klinikum Oldenburg and Faculty of Medicine, University of Oldenburg, Oldenburg, Germany.Germany
文献类型
病例报告 · 综述
期刊
Cancer reports (Hoboken, N.J.)2025 Sep
原文标识
PubMed 40907496 · DOI 10.1002/cnr2.70334