Comparative study on epstein-barr virus-positive mucocutaneous ulcer and methotrexate-associated lymphoproliferative disorders developed in the oral mucosa: A case series of 10 patients and literature review

Kyoichi Obata, Tatsuo Okui, Sawako Ono, Koki Umemori, Shoji Ryumon, Kisho Ono, Mayumi Yao, Norie Yoshioka, Soichiro Ibaragi, Akira Sasaki

Research output: Contribution to journalArticlepeer-review

Abstract

Methotrexate-associated lymphoproliferative disorder (MTX-LPD) is an iatrogenic immunodeficiency-associated lymphoproliferative disorder that occurs mainly with MTX use. This disorder has been associated with Epstein-Barr virus (EBV) infection. In 2017, the WHO newly defined the disease concept of EBV-positive mucocutaneous ulcer (EBV-MCU) as a good-prognosis EBV-related disease. Here, we report 10 cases of MTX-LPD or EBV-MCU in the oral mucosa. This retrospective, observational study was conducted with MTX-LPD or EBV-MCU in the oral mucosa patients who visited us during the nine year period from 2012 to 2021. We gathered the basic information, underlying disease, histopathological evaluation, treatment and prognosis for the subjects. All were being treated with MTX for rheumatoid arthritis. EBV infection was positive in all cases by immunohistochemistry. A complete or partial response was obtained in all cases with the withdrawal of MTX. Our results suggests that the most common risk factor for developing EBV-MCU is the use of immunosuppressive drugs. The most common site of onset is the oral mucosa, which may be attributed to the mode of EBV infection and the high incidence of chronic irritation of the oral mucosa. A small number of patients had been diagnosed with MTX-LPD, but we consider that these cases were EBV-MCU based on our study.

Original languageEnglish
Article number1375
JournalDiagnostics
Volume11
Issue number8
DOIs
Publication statusPublished - Aug 2021

Keywords

  • Epstein-Barr virus
  • Lymphoproliferative disorders
  • Methotrexate
  • Mucocutaneous ulcer
  • Rheumatoid arthritis

ASJC Scopus subject areas

  • Clinical Biochemistry

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