Epstein-Barr virus-infected subcutaneous panniculitis-like T-cell lymphoma associated with methotrexate treatment

Epstein-Barr virus-infected subcutaneous panniculitis-like T-cell lymphoma associated with methotrexate treatment
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DOI:
10.1007/s12185-010-0642-5
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发表时间:
2010-09-01
影响因子:
2.1
通讯作者:
Daibata, Masanori
Daibata, Masanori
中科院分区:
医学4区
文献类型:
--
作者:
Nemoto, Yuiko;Taniguchi, Ayuko;Daibata, Masanori

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甲氨蝶呤相关淋巴增生性疾病(MTX-LPD)是接受MTX治疗的患者发生的淋巴样增生或淋巴瘤。我们报告一例EB病毒(EBV)阳性的皮下脂膜炎样T细胞淋巴瘤(SPTCL)在一个60岁的日本男子与MTX类风湿关节炎(RA)治疗。SPTCL是一种罕见的细胞毒性T细胞淋巴瘤,其特征是累及皮下脂肪,类似脂膜炎。病人,谁一直在MTX治疗类风湿关节炎,表现出高热和腰痛。体格检查显示躯干(包括腋窝和腹股沟区)有多个皮下结节。腹股沟结节活检显示皮下脂肪组织中大量CD 8(+)T细胞淋巴瘤细胞浸润。根据世界卫生组织分类标准诊断为SPTCL。EB病毒编码的小RNA原位杂交结果显示,淋巴瘤细胞中含有EB病毒基因组。这些细胞对EBV潜伏膜蛋白1呈阳性,但对EBNA 2不呈阳性。MTX停药后,结节自行消退。研究报告,大多数MTX-LPD是B细胞型淋巴瘤和霍奇金淋巴瘤。据我们所知,在接受MTX治疗的患者中尚未报告EBV阳性SPTCL。我们的病例强调了MTX相关SPTCL的临床和病毒学特征的重要性。
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.