TY - JOUR
T1 - Comparative study on epstein-barr virus-positive mucocutaneous ulcer and methotrexate-associated lymphoproliferative disorders developed in the oral mucosa
T2 - A case series of 10 patients and literature review
AU - Obata, Kyoichi
AU - Okui, Tatsuo
AU - Ono, Sawako
AU - Umemori, Koki
AU - Ryumon, Shoji
AU - Ono, Kisho
AU - Yao, Mayumi
AU - Yoshioka, Norie
AU - Ibaragi, Soichiro
AU - Sasaki, Akira
N1 - Funding Information:
Funding: This work was partly supported by a Grant-in-Aid for Early-Career Scientists (JP20K18668 to Obata K.) from the Japan Society for the promotion of Sciences, Japan.
Publisher Copyright:
© 2021 by the authors. Licensee MDPI, Basel, Switzerland.
PY - 2021/8
Y1 - 2021/8
N2 - 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.
AB - 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.
KW - Epstein-Barr virus
KW - Lymphoproliferative disorders
KW - Methotrexate
KW - Mucocutaneous ulcer
KW - Rheumatoid arthritis
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U2 - 10.3390/diagnostics11081375
DO - 10.3390/diagnostics11081375
M3 - Article
AN - SCOPUS:85112591072
SN - 2075-4418
VL - 11
JO - Diagnostics
JF - Diagnostics
IS - 8
M1 - 1375
ER -