Other Iatrogenic Immunodeficiency-associated Lymphoproliferative Disorder, Hodgkin Type, following Epstein-Barr Viral Hepatitis in a Patient with Rheumatoid Arthritis

Abstract

A 59-year-old man with an 18-year history of rheumatoid arthritis who had been treated with steroids, methotrexate, and infliximab presented with a high-grade fever, cervical lymphadenopathy, and hepatosplenomegaly. Epstein-Barr virus (EBV) hepatitis was diagnosed based on the liver histology and EBV antibody titer. The symptoms improved temporarily, but five months later, the fever, skin rash, jaundice, and thrombocytopenia relapsed. Bone marrow and liver biopsies demonstrated infiltration with Reed-Sternberg cells. Based on these findings, the patient was diagnosed with other iatrogenic immunodeficiency-associated lymphoproliferative disorder (OIIA-LPD), Hodgkin lymphoma type. This case followed a rare clinical course, in that acute hepatitis preceded the diagnosis of OIIA-LPD.

Epstein-Barr Virus–Related Lymphoproliferative Disorders–Associated Polymyositis in a Patient With Rheumatoid Arthritis

“…We here report a case of polymyositis with EBV-LPD due to MTX and infliximab treatment with spontaneous regression after the discontinuation of these agents. Although a few reports have described an association of EBV-related LPD with MTX and infliximab therapy, the involvement of skeletal muscles has never been noted in previous cases 1–3 . The causal link between infliximab and LPD remains uncertain in the present case because LPD regressed with the cessation of both MTX and infliximab.…”

“…Although a few reports have described an association of EBV-related LPD with MTX and infliximab therapy, the involvement of skeletal muscles has never been noted in previous cases. [1][2][3] The causal link between infliximab and LPD remains uncertain in the present case because LPD regressed with the cessation of both MTX and infliximab. For the treatment of LPD associated with immunosuppressive therapy, the discontinuation of the immunosuppressive agents should be the first choice after careful histologic examination for EBV in the peripheral tissues and its genome in the peripheral blood cells.…”