Methotrexate-Related Epstein-Barr Virus (EBV)-Associated Lymphoproliferative Disorder-So-Called ''Hodgkin-Like Lesion''-of the Oral Cavity in a Patient with Rheumatoid Arthritis
Methotrexate-Related Epstein-Barr Virus (EBV)-Associated Lymphoproliferative Disorder-So-Called ''Hodgkin-Like Lesion''-of the Oral Cavity in a Patient with Rheumatoid Arthritis
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DOI：
10.1007/s12105-010-0202-6
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发表时间：
2010-12-01
影响因子：
2.1
通讯作者：
Kusama, Kaoru
中科院分区：
文献类型：
--
作者：
Kikuchi, Kentaro;Miyazaki, Yuji;Kusama, Kaoru
患有自身免疫性疾病(类风湿性关节炎、牛皮癣、皮肌炎)的患者接受甲氨蝶呤(MTX)治疗，有时会发展为淋巴增生性疾病(LPD)。在报告的病例中，大约40%的受累部位位于结外，包括胃肠道、皮肤、肺、肾脏和软组织。然而，MTX相关性LPD(MTX-LPD)在口腔中极为罕见。在此，我们报告一位69岁的日本女性患类风湿性关节炎(RA)，她的左上颌骨出现类似霍奇金氏病的MTX-LPD，即所谓的“霍奇金样病变”。组织病理学观察发现，溃烂口腔黏膜肉芽组织中可见大的非典型淋巴样细胞，包括霍奇金细胞或Reed-Sternberg样细胞。免疫组织化学染色显示CD20、CD30和EB病毒潜伏感染膜蛋白-1(LMP-1)阳性，CD15阴性。用EBV编码的小RNA(EBER)原位杂交(ISH)和聚合酶链式反应(PCR)检测石蜡包埋标本中LMP-1和EBNA-2。据我们所知，这是第一例报告的甲氨蝶呤相关EBV相关的LPD(MTX-EBVLPD)，“霍奇金样病变”，在类风湿关节炎患者的口腔。
Patients affected by autoimmune diseases (rheumatoid arthritis, psoriasis, dermatomyositis) who are treated with methotrexate (MTX) sometimes develop lymphoproliferative disorders (LPDs). In approximately 40% of reported cases, the affected sites have been extranodal, and have included the gastrointestinal tract, skin, lung, kidney, and soft tissues. However, MTX-associated LPD (MTX-LPD) is extremely rare in the oral cavity. Here we report a 69-year-old Japanese woman with rheumatoid arthritis (RA) who developed MTX-LPD resembling Hodgkin's disease-so-called ''Hodgkin-like lesion''-in the left upper jaw. Histopathologically, large atypical lymphoid cells including Hodgkin or Reed-Sternberg-like cells were found to have infiltrated into granulation tissue in the ulcerative oral mucosa. Immunohistochemistry showed that the large atypical cells were positive for CD20, CD30 and Epstein-Barr virus (EBV)-latent infection membrane protein- 1 (LMP-1) and negative for CD15. EBV was detected by in situ hybridization (ISH) with EBV-encoded small RNA (EBER), and polymerase chain reaction (PCR) for LMP-1 and EBNA-2 in material taken from the formalinfixed, paraffin-embedded specimen. To our knowledge, this is the first reported case of MTX-related EBV-associated LPD (MTX-EBVLPD), ''Hodgkin-like lesion'', of the oral cavity in a patient with RA.