Department of Microbiology and Infection, Kochi Medical School, Kochi University, Kochi, Japan.
Int J Hematol. 2010 Sep;92(2):364-8. doi: 10.1007/s12185-010-0642-5. Epub 2010 Jul 28.
Methotrexate-associated lymphoproliferative disorder (MTX-LPD) is a lymphoid proliferation or lymphoma in patients treated with MTX. We report a case of Epstein-Barr virus (EBV)-positive subcutaneous panniculitis-like T-cell lymphoma (SPTCL) in a 60-year-old Japanese man treated with MTX for rheumatoid arthritis (RA). SPTCL is a rare cytotoxic T-cell lymphoma characterized by involvement of subcutaneous fat mimicking panniculitis. The patient, who had been on MTX therapy for RA, manifested high fever and lumbago. Physical examination showed multiple subcutaneous nodules on the trunk including axillary and inguinal regions. Biopsy of the inguinal nodule showed profuse infiltration of CD8(+) T-cell lymphoma cells in the subcutaneous adipose tissues. A diagnosis of SPTCL was made according to the diagnostic criteria of World Health Organization classification. EBV-encoded small RNA in situ hybridization revealed that the lymphoma cells contained EBV genome. The cells were positive for EBV latent membrane protein 1, but not for EBNA2. After discontinuation of MTX, the nodules regressed spontaneously. Studies have reported that most MTX-LPDs are B-cell type lymphomas and Hodgkin lymphoma. To the best of our knowledge, EBV-positive SPTCL has not been reported in patients receiving MTX. Our case emphasizes the importance of clinical and virological characterization of MTX-associated SPTCL.
甲氨蝶呤相关性淋巴组织增生性疾病(MTX-LPD)是接受甲氨蝶呤(MTX)治疗的患者发生的淋巴增生或淋巴瘤。我们报告了一例接受 MTX 治疗类风湿关节炎(RA)的 60 岁日本男性发生的 EBV 阳性皮下脂膜炎样 T 细胞淋巴瘤(SPTCL)。SPTCL 是一种罕见的细胞毒性 T 细胞淋巴瘤,其特征是皮下脂肪受累类似于脂膜炎。该患者因 RA 接受 MTX 治疗,表现为高热和腰痛。体格检查显示躯干多处皮下结节,包括腋窝和腹股沟区域。腹股沟结节活检显示皮下脂肪组织中大量浸润 CD8(+) T 细胞淋巴瘤细胞。根据世界卫生组织分类的诊断标准诊断为 SPTCL。EBV 编码的小 RNA 原位杂交显示淋巴瘤细胞含有 EBV 基因组。细胞阳性表达 EBV 潜伏膜蛋白 1,但不表达 EBNA2。停用 MTX 后,结节自发消退。研究报道大多数 MTX-LPD 为 B 细胞型淋巴瘤和霍奇金淋巴瘤。据我们所知,接受 MTX 治疗的患者中尚未报道 EBV 阳性 SPTCL。我们的病例强调了对 MTX 相关性 SPTCL 进行临床和病毒学特征分析的重要性。