舌に発生したCD4－CD8－ダブルネガティブT細胞性メトトレキサート関連リンパ増殖性疾患の1例

• Published in: 日本口腔外科学会雑誌 Vol. 68; no. 8; pp. 346 - 353
• Main Authors: 眞野, 亮介, 吉野, 綾, 中村, 真輔, 石田, 晋太郎, 橋口, 志保, 近藤, 誠二
• Format: Journal Article
• Language: Japanese
• Published: 公益社団法人 日本口腔外科学会 20.08.2022
• Subjects: T細胞性リンパ増殖性疾患; メトトレキサート関連性リンパ増殖性疾患; 関節リウマチ
• ISSN: 0021-5163; 2186-1579
• DOI: 10.5794/jjoms.68.346

Recently, methotrexate-associated lymphoproliferative disorders (MTX-LPDs) have received attention as a disease concept in the oral and maxillofacial region. Most cases show histopathological Epstein-Barr virus (EBV)-positive B-cell lymphoma, with spontaneous resolution observed following the cessation of MTX. We herein report a case of T-cell MTX-LPD of the tongue that was negative for EBV and showed a CD4− CD8− double negative T-cell histology. A 70-year-old woman was referred to our hospital with the chief complaint of an intractable ulcer of the tongue which had existed for approximately one month. Examination revealed a 20×10 mm ulcer on the right inferior surface of the tongue. She had a medical history of rheumatoid arthritis treated with MTX. We diagnosed her with EBV-negative, CD4 − CD8 − double negative T-cell MTX-LPD, based on the findings of a blood biochemical specimen and tissue biopsy. The tongue lesion showed spontaneous resolution after MTX withdrawal. The course was uneventful, and there was no evidence of recurrence of the lesion at one year after the withdrawal of MTX.