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Case Report
Massive hemorrhage and osteonecrosis of the jaw in the patient with methotrexate-associated lymphoproliferative disorder: Report of a case and review of literature
1 Department of Dentistry and Oral Surgery, Kawasaki Municipal Kawasaki Hospital, Kawasaki, Japan
2 Department of Dentistry and Oral Surgery, School of Medicine, Keio University, Tokyo, Japan
3 Department of Dentistry and Oral Surgery, Kawasaki Municipal Ida Hospital, Kawasaki, Japan
Address correspondence to:
Takazumi Yasui
DDS, PhD, Department of Dentistry and Oral Surgery, Kawasaki Municipal Kawasaki Hospital, 12-1 Shinkawadori, Kawasaki-ku, Kawasaki, Kanagawa 210-0013,
Japan
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Article ID: 101202Z01MK2021
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Kimura M, Yasui T, Nagamine H, Yajima S, Kodaka R, Onizawa K. Massive hemorrhage and osteonecrosis of the jaw in the patient with methotrexate-associated lymphoproliferative disorder: Report of a case and review of literature. Int J Case Rep Images 2021;12:101202Z01MK2021.ABSTRACT
Introduction: Methotrexate-associated lymphopro-liferative disorder (MTX-LPD) is one of the adverse effects of methotrexate (MTX). We report a case of MTX-LPD that caused osteonecrosis and massive bleeding without regression following MTX withdrawal.
Case Report: A 61-year-old woman was referred to our hospital because of delayed socket healing after dental extraction. She had been diagnosed with rheumatoid arthritis (RA) and received MTX treatment for six years. At the first visit, a 15 mm × 15 mm bone exposure and swelling of the surrounding gingiva in the right upper molar region were observed. We performed an incisional biopsy of the necrotic bone and surrounding mucosal tissue. The histopathological diagnosis was diffuse large B-cell lymphoma (DLBCL). Therefore, we referred the patient to the Department of Hematology in our hospital. She was diagnosed with MTX-LPD based on the history of MTX therapy for RA, and MTX was discontinued immediately. However, the lesion progressed and caused bleeding that required blood transfusion and hemostasis by microfibrous collagen and tie-over even after the withdrawal of MTX. Subsequently, rituximab, cyclophosphamide, vincristine, and prednisolone (R-COP) chemotherapy was initiated. After 3 courses of chemotherapy, sequestrectomy was performed under local anesthesia. She achieved complete remission after 8 courses of chemotherapy, and there was no recurrence of necrotic bone exposure or gingival swelling.
Conclusion: Approximately 80% of MTX-LPD cases occurring in the oral region show regression after MTX discontinuation, but we experienced a case in which bleeding occurred with progression of the lesion after discontinuation. Careful follow-up is required during the MTX discontinuation period.
Keywords: Diffuse large B-cell lymphoma, Massive hemorrhage, Methotrexate-associated lymphoproliferative disorder, Osteonecrosis of the jaw
SUPPORTING INFORMATION
Author Contributions
Moemi Kimura - Conception of the work, Design of the work, Acquisition of data, Analysis of data, Drafting the work, Revising the work critically for important intellectual content, Final approval of the version to be published, Agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.
Takazumi Yasui - Conception of the work, Design of the work, Acquisition of data, Analysis of data, Drafting the work, Revising the work critically for important intellectual content, Final approval of the version to be published, Agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.
Hiroki Nagamine - Conception of the work, Design of the work, Acquisition of data, Analysis of data, Drafting the work, Revising the work critically for important intellectual content, Final approval of the version to be published, Agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.
Shosuke Yajima - Conception of the work, Design of the work, Acquisition of data, Analysis of data, Drafting the work, Revising the work critically for important intellectual content, Final approval of the version to be published, Agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.
Rie Kodaka - Conception of the work, Design of the work, Acquisition of data, Analysis of data, Drafting the work, Revising the work critically for important intellectual content, Final approval of the version to be published, Agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.
Katsuhiro Onizawa - Conception of the work, Design of the work, Acquisition of data, Analysis of data, Drafting the work, Revising the work critically for important intellectual content, Final approval of the version to be published, Agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.
Guarantor of SubmissionThe corresponding author is the guarantor of submission.
Source of SupportNone
Consent StatementWritten informed consent was obtained from the patient for publication of this article.
Data AvailabilityAll relevant data are within the paper and its Supporting Information files.
Conflict of InterestAuthors declare no conflict of interest.
Copyright© 2021 Moemi Kimura et al. This article is distributed under the terms of Creative Commons Attribution License which permits unrestricted use, distribution and reproduction in any medium provided the original author(s) and original publisher are properly credited. Please see the copyright policy on the journal website for more information.
