Ishida Mitsuaki, Hodohara Keiko, Yoshii Miyuki, Okuno Hiroko, Horinouchi Akiko, Nakanishi Ryota, Harada Ayumi, Iwai Muneo, Yoshida Keiko, Kagotani Akiko, Yoshida Takashi, Okabe Hidetoshi
Department of Clinical Laboratory Medicine, Shiga University of Medical Science Shiga, Japan ; Division of Diagnostic Pathology, Shiga University of Medical Science Shiga, Japan.
Int J Clin Exp Pathol. 2013 Sep 15;6(10):2237-41. eCollection 2013.
It is well recognized that patients with immunodeficiency have a high risk of development of lymphoproliferative disorders (LPDs), and Epstein-Barr virus (EBV) is associated with the occurrence of LPDs. Methotrexate (MTX) is one of the common cause of iatrogenic-associated LPD, and approximately 40-50% of MTX-related LPD cases occur in extranodal sites. However, the occurrence of MTX-related LPD in the gingiva is extremely rare. Herein, we report the fourth documented case of MTX-related EBV-associated LPD occurring in the gingiva of a patient with rheumatoid arthritis (RA). A 76-year-old Japanese female with a 10-year history of RA, who was treated with MTX and infliximab, presented with a tumorous lesion in the gingiva. Biopsy of the gingiva tumor revealed diffuse proliferation of large-sized lymphoid cells with cleaved nuclei containing conspicuous nucleoli. These lymphoid cells were CD20- and EBER-positive. Therefore, a diagnosis of MTX-related EBV-associated LPD showing features of diffuse large B-cell lymphoma (DLBCL) that occurred in the gingiva was made. Although the occurrence of LPD in the oral region, as seen in the present case, is rare, the prevalence of this disorder may be on the rise due to the increased number of patients undergoing immunosuppression therapy. Moreover, immunosenescence can also be a cause of EBV-associated LPD. Therefore, recognition of the occurrence of this disorder in the oral cavity and consideration of the clinical history can facilitate the correct diagnosis.
众所周知，免疫缺陷患者发生淋巴增殖性疾病（LPD）的风险很高，而爱泼斯坦-巴尔病毒（EBV）与LPD的发生有关。甲氨蝶呤（MTX）是医源性相关LPD的常见原因之一，约40%-50%的MTX相关LPD病例发生在结外部位。然而，MTX相关LPD在牙龈中发生极为罕见。在此，我们报告第四例有文献记载的MTX相关EBV相关LPD发生在一名类风湿关节炎（RA）患者牙龈中的病例。一名76岁的日本女性，有10年RA病史，接受MTX和英夫利昔单抗治疗，牙龈出现肿瘤性病变。牙龈肿瘤活检显示核有裂沟、含明显核仁的大淋巴细胞弥漫性增生。这些淋巴细胞CD20和EBER阳性。因此，诊断为MTX相关EBV相关LPD，表现为发生在牙龈的弥漫性大B细胞淋巴瘤（DLBCL）特征。尽管如本病例所见，LPD在口腔区域的发生很罕见，但由于接受免疫抑制治疗的患者数量增加，这种疾病的患病率可能正在上升。此外，免疫衰老也可能是EBV相关LPD的一个原因。因此，认识到这种疾病在口腔中的发生并考虑临床病史有助于正确诊断。
