Oral Methotrexate-related Lymphoproliferative Disease Presenting with Severe Osteonecrosis of the Jaw: A Case Report and Literature Review

Intern Med. 2018 Feb 15;57(4):575-581. doi: 10.2169/internalmedicine.8946-17. Epub 2017 Dec 8.

Abstract

Long-term methotrexate (MTX) treatment can cause MTX-related lymphoproliferative disorder (MTX-LPD). We experienced a case of MTX-LPD that was associated with severe osteonecrosis of the jaw mimicking medication-related osteonecrosis of the jaw. The patient was an 81-year-old woman with rheumatoid arthritis (RA) who was treated with MTX and bisphosphonate. After 7 years, she was referred to our department for the assessment of giant ulcer and exposure of the alveolar bone of the left maxilla. Histopathological and immunological analyses confirmed a diagnosis of MTX-LPD. At seven months after the cessation of MTX treatment, the ulcerative and necrotic lesions had markedly decreased in size. A 1-year follow-up examination showed no evidence of recurrence and good RA control.

Keywords: Epstein-Barr virus; lymphoma; medication-related osteonecrosis of the jaw; methotrexate-related lymphoproliferative disorder; rheumatoid arthritis.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Aged, 80 and over
  • Antirheumatic Agents / adverse effects*
  • Female
  • Humans
  • Jaw
  • Lymphoproliferative Disorders / chemically induced*
  • Lymphoproliferative Disorders / complications
  • Lymphoproliferative Disorders / diagnosis*
  • Methotrexate / adverse effects*
  • Osteonecrosis / diagnosis
  • Osteonecrosis / etiology*
  • Severity of Illness Index

Substances

  • Antirheumatic Agents
  • Methotrexate