Spontaneous regression of breast lymphoproliferative disorders after withdrawal of methotrexate in rheumatoid arthritis patients with Epstein-Barr virus infection: a case report and review of the literature

• Published in: Journal of medical case reports Vol. 16; no. 1; p. 49
• Main Authors: Ogawa, Ayumi, Nakagawa, Tsuyoshi, Kumaki, Yuichi, Hosoya, Tokuko, Oda, Goshi, Mori, Mio, Fujioka, Tomoyuki, Kubota, Kazunori, Onishi, Iichiro, Uetake, Hiroyuki
• Format: Journal Article
• Language: English
• Published: England BioMed Central Ltd 07.02.2022; BioMed Central; BMC
• Subjects: Antigens; Arthritis; Arthritis, Rheumatoid - drug therapy; Biopsy; Breast lymphoma; Case Report; Case reports; Drug withdrawal; Epstein-Barr virus; Epstein-Barr Virus Infections - complications; Epstein-Barr Virus Infections - diagnosis; Epstein–Barr virus (EBV); Female; Health aspects; Herpesvirus 4, Human; Humans; Immunoglobulin G; Infections; Laboratories; Lymphatic diseases; Lymphoma; Lymphoproliferative Disorders - chemically induced; Lymphoproliferative Disorders - drug therapy; Methotrexate; Methotrexate - adverse effects; Methotrexate-associated lymphoproliferative disorders (MTX-LPD); Middle Aged; Neoplasm Recurrence, Local; Other iatrogenic immunodeficiency-associated lymphoproliferative disorders (OIIA-LPD); Patients; Prognosis; Rheumatoid arthritis; Rheumatoid factor; Tumors; United States > US; Breast lymphoma; Other iatrogenic immunodeficiency-associated lymphoproliferative disorders (OIIA-LPD); Epstein–Barr virus (EBV); Methotrexate-associated lymphoproliferative disorders (MTX-LPD)
• ISSN: 1752-1947; 1752-1947
• DOI: 10.1186/s13256-022-03274-1

Lymphoproliferative disorder (LPD) has been shown to occur after treatment with methotrexate (MTX). Currently, MTX-LPD has become widely recognized, but its mechanism and prognostic factors remain unclear. We report the first case of Epstein-Barr virus (EBV)-associated MTX-LPD of the breast. A 63-year-old Asian woman with long-term rheumatoid arthritis presented to our facility with intermittent fever. A physical examination revealed a 3-cm lump in her left breast. She had been taking MTX for the past 15 years. Laboratory studies revealed slightly elevated levels of EBV-viral capsid antigen antibody immunoglobulin G and EBV nuclear antibody. Contrast-enhanced computer tomography revealed a mass in the left breast, a subcutaneous nodule in the abdomen, a mass in the left lung, and a nodule in the left retroperitoneum. The definitive diagnosis was consistent with MTX-LPD merging into an EBV-positive, diffuse large B-cell lymphoma. Six months following the withdrawal of MTX, the breast mass had markedly shrunk and the patient remained in good health for 1 year with no evidence of relapse of LPD. MTX-LPD rarely occurs in the breast, and it is difficult to diagnose because there have only been six reported cases of breast MTX-LPD reported in the literature. EBV-positive MTX-LPD tends to regress spontaneously after MTX withdrawal, and our case also had similar results. It is important to make an appropriate diagnosis of MTX-LPD of the breast based on imaging and pathology to determine the appropriate treatment protocol for this rare disorder.