A case of CD4－ CD8－ double negative T-cell methotrexate-associated lymphoproliferative disorder of the tongue

Abstract

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.