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Volume 11
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10.3390/diagnostics11081375
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Case Report

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

by
Kyoichi Obata
1,*,
Tatsuo Okui
2,*,
Sawako Ono
3,
Koki Umemori
1,
Shoji Ryumon
1,
Kisho Ono
1,
Mayumi Yao
4,
Norie Yoshioka
1,
Soichiro Ibaragi
1 and
Akira Sasaki
1
1
Department of Oral and Maxillofacial Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, 2-5-1, Shikata, Okayama-City 700-8558, Japan
2
Department of Oral and Maxillofacial Surgery, Shimane University Faculty of Medicine, 89-1, Enya, Izumo-City 693-8501, Japan
3
Department of Pathology, Kagawa Prefectural Central Hospital, 1-2-1, Asahi, Takamatsu-City 760-8557, Japan
4
Department of Dentistry and Dental Surgery, Tsuyama Chuo Hospital, 1756, Kawasaki, Tsuyama-City 708-0841, Japan
*
Authors to whom correspondence should be addressed.
Diagnostics 2021, 11(8), 1375; https://doi.org/10.3390/diagnostics11081375
Submission received: 12 July 2021 / Revised: 27 July 2021 / Accepted: 28 July 2021 / Published: 30 July 2021
(This article belongs to the Special Issue Oral Diseases: Anatomy and Clinical Diagnosis)
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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.
Keywords: methotrexate; lymphoproliferative disorders; Epstein-Barr virus; mucocutaneous ulcer; rheumatoid arthritis methotrexate; lymphoproliferative disorders; Epstein-Barr virus; mucocutaneous ulcer; rheumatoid arthritis

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MDPI and ACS Style

Obata, K.; Okui, T.; Ono, S.; Umemori, K.; Ryumon, S.; Ono, K.; Yao, M.; Yoshioka, N.; Ibaragi, S.; Sasaki, A. 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. Diagnostics 2021, 11, 1375. https://doi.org/10.3390/diagnostics11081375

AMA Style

Obata K, Okui T, Ono S, Umemori K, Ryumon S, Ono K, Yao M, Yoshioka N, Ibaragi S, Sasaki A. 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. Diagnostics. 2021; 11(8):1375. https://doi.org/10.3390/diagnostics11081375

Chicago/Turabian Style

Obata, Kyoichi, Tatsuo Okui, Sawako Ono, Koki Umemori, Shoji Ryumon, Kisho Ono, Mayumi Yao, Norie Yoshioka, Soichiro Ibaragi, and Akira Sasaki. 2021. "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" Diagnostics 11, no. 8: 1375. https://doi.org/10.3390/diagnostics11081375

APA Style

Obata, K., Okui, T., Ono, S., Umemori, K., Ryumon, S., Ono, K., Yao, M., Yoshioka, N., Ibaragi, S., & Sasaki, A. (2021). 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. Diagnostics, 11(8), 1375. https://doi.org/10.3390/diagnostics11081375

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