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.
Highlights
Methotrexate-associated lymphoproliferative disorder (MTX-LPD) is one of the adverse effects of methotrexate (MTX)
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
Medication-related osteonecrosis of the jaw (MRONJ), which is associated with bisphosphonates, anti-receptor activator of nuclear factor κB ligand, and antiangiogenic medications, has been widely recognized [3]
Summary
Methotrexate (MTX) is currently the most widely used first-line conventional synthetic disease-modifying. The patient was referred to Department of Dentistry and Oral Surgery, because of delayed socket healing after dental extraction. Her past medical history was significant for RA and myocardial infarction. Oral examination revealed a 15 mm × 15 mm area of exposed bone with necrotic soft tissue in the right posterior maxilla (Figure 1). Histopathological findings of the bone confirmed a sequestrum with empty osteocytic lacunae and bacterial infection, including colonies of Actinomyces spp. in the marrow space It did not reveal tumor cells, including atypical lymphocytes, usually found in MTXLPD. The lesion progressed and caused massive bleeding three weeks after the withdrawal of MTX She visited the emergency department of our hospital. There was no recurrence of necrotic bone exposure or gingival swelling
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