A Case of Methotrexate-Associated Lymphoproliferative Disorder of the Maxillary Gingiva and a Literature Review of Oral Cases of the Disease

Abstract

Methotrexate （MTX） is widely used to treat rheumatoid arthritis. However, it has been suggested that MTX-treated rheumatoid arthritis patients are at risk of lymphoma. Recently, the occurrence of this lymphoproliferative disorder in the oral cavity has been reported as an MTX-associated lymphoproliferative disorder （MTX-LPD）. We report a case of MTX-LPD combined with ulceration and jaw necrosis in a 72-year-old male, who had received MTX for 7 years for rheumatoid arthritis. He was referred to our clinic to receive treatment for pain in the posterior maxillary gingiva. Computed tomography showed bone resorption around the root of the maxillary premolar, and bone sequestration was suspected. At first, we diagnosed the patient with osteomyelitis, but the pathological diagnosis was MTX-LPD.
We reviewed this case and 23 previously reported cases of oral MTX-LPD that occurred in Japan. The oral MTX-LPD developed outside of the lymph nodes in 40-50％ of cases. Ulceration was seen in 20 cases, 23 cases involved no or slight swelling, the maxillary and/or mandibular gingiva was affected in 22 cases, and 14 cases involved bone resorption or bone defects. Many cases were clinically diagnosed as osteomyelitis, malignant tumors, or chronic periodontitis. When oral MTX-LPD is suspected, the patient’s medical history should be taken, and a systemic examination should be performed.