Primary Adrenal Lymphoma Possibly Associated With Epstein-Barr Virus Reactivation Due to Immunosuppression Under Methotrexate Therapy.
Primary Adrenal Lymphoma Possibly Associated With Epstein-Barr Virus Reactivation Due to Immunosuppression Under Methotrexate Therapy.
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DOI：
10.1097/md.0000000000001270
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发表时间：
2015-08
期刊：
影响因子：
1.6
通讯作者：
Matsuda M
中科院分区：
文献类型：
--
作者：
Ohkura Y;Shindoh J;Haruta S;Kaji D;Ota Y;Fujii T;Hashimoto M;Watanabe G;Matsuda M
原发性肾上腺淋巴组织增生性疾病（LPD）是一种极为罕见的疾病，广泛认为与类风湿性关节炎（RA）患者使用甲氨蝶呤（MTX）有关。一位70岁的老人在一次体检中偶然发现肝脏背侧有一个肿瘤。他有接受MTX治疗的RA病史。腹部超音波检查显示在肝脏背侧有一个低回声肿块（直径30毫米），位于下腔静脉附近。 术前鉴别诊断包括肝内胆管细胞癌、肾上腺肿瘤和肝脏恶性淋巴瘤，但没有明确的诊断。在剖腹探查术中，肿瘤似乎来自右肾上腺，并紧紧地粘附在肝脏的第7段。因此，进行了右肾上腺切除术，部分切除了肝脏第7段。病理结果显示弥漫性炎性细胞浸润，伴小的非典型淋巴样细胞，EB病毒（EBV）免疫组化阳性。最终诊断为原发性肾上腺医源性EBV阳性LPD，分类为“其他医源性免疫缺陷相关LPD：霍奇金样病变”。本文阐述了甲氨蝶呤相关性LPD（MTX-LPD）在治疗前自发愈合的可能性，以及怀疑MTX-LPD和对坏死组织进行免疫染色的重要性。据我们所知，这是第一例MTX相关的EBV阳性LPD，霍奇金样病变，单侧肾上腺RA患者。
Primary adrenal lymphoproliferative disorder (LPD) is an extremely rare disease that is widely known to be associated with methotrexate (MTX) use in patients with rheumatoid arthritis (RA). A 70-year-old man was incidentally found to have a tumor at the dorsal part of the liver in a medical check-up. He had a history of RA treated with MTX. Abdominal ultrasonography demonstrated a low echoic mass (30 mm in diameter) at the dorsal part of the liver, located close to the inferior vena cava. Preoperative differential diagnoses included intrahepatic cholangiocarcinoma, adrenal tumor, and hepatic malignant lymphoma, but no definitive diagnosis was reached. On exploratory laparotomy, the tumor seemed to be derived from the right adrenal gland and adhered tightly to segment 7 of the liver. Therefore, right adrenectomy with partial resection of segment 7 of the liver was performed. Pathological findings revealed diffuse inflammatory cell infiltration with a population of small atypical lymphoid cells, with positive immunohistochemical evidence for Epstein–Barr virus (EBV). Final diagnosis was primary adrenal iatrogenic EBV-positive LPD, classified as “other iatrogenic immunodeficiency-associated LPDs: Hodgkin-like lesions.” In this report, we described the possibility of the spontaneous healing of MTX-associated LPD (MTX-LPD) before treatment and the importance of doubting MTX-LPD and doing immunostaining to necrotic tissue. To our knowledge, this is the first reported case of MTX-related EBV-positive LPD, Hodgkin-like lesion, of the unilateral adrenal gland in patient with RA.