Abstract
BackgroundLymphoproliferative 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.Case presentationWe 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.ConclusionMTX-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.
Highlights
Lymphoproliferative disorder (LPD) has been shown to occur after treatment with methotrexate (MTX)
It is important to make an appropriate diagnosis of MTX-associated lymphoproliferative disorder (MTXLPD) of the breast based on imaging and pathology to determine the appropriate treatment protocol for this rare disorder
LPD that arises in patients treated with immunosuppressive drugs, including biologic drugs for autoimmune diseases, is categorized as “other iatrogenic immunodeficiency-associated lymphoproliferative disorder (OIIA-LPD)”, other than in post-transplant lymphoproliferative disorder, according to the World Health Organization (WHO) Classification of Tumors of Hematopoietic and Lymphoid Tissues, 4th edition (2008)
Summary
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 had similar results. It is important to make an appropriate diagnosis of MTXLPD of the breast based on imaging and pathology to determine the appropriate treatment protocol for this rare disorder
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