Methotrexate-Associated Lymphoproliferative Disorder with Intraocular Involvement Improved with Intravitreal Methotrexate and Chemotherapy: A Case Report

Introduction: Although methotrexate-associated lymphoproliferative disorders (MTX-LPDs) often develop in extranodal sites, such as the lungs and skin, their involvement in the eyes is quite rare. We report a case of MTX-LPD with subretinal involvement. Case Presentation: A 66-year-old woman had a 14-year history of rheumatoid arthritis and had been receiving MTX for 8 years. Multiple erythematous nodules had developed on both lower extremities during MTX administration, and the patient was diagnosed with diffuse large B-cell lymphoma not otherwise specified (DLBCL-NOS). Since the skin masses disappeared within a couple of months after MTX withdrawal, MTX-LPD was diagnosed. Although no recurrence of LPD was observed for 1 year, she suddenly presented with decreased visual acuity in the right eye. Fundus examination showed a swelling of the optic disc, vitreous opacity, and multiple widespread, subretinal, yellow-white, elevated lesions. Since the cytokine profile of the vitreous fluid indicated that interleukin (IL)-10/6 ratio was >1, relapse of MTX-LPD in the eye was diagnosed. Intravitreal injection of MTX (IVMTX) successfully reduced the LPD and led to improved visual acuity. However, a new mass developed in the left cerebellum. Open biopsy confirmed relapse of DLBCL-NOS, and six cycles of chemotherapy resulted in complete remission (CR). She has had no recurrence for 16 months after the last administration of IVMTX in the eye and other regions. Conclusions: Ocular involvement was observed in patients with MTX-DLBCL-NOS. Although IVMTX is effective for intraocular involvement, careful observation should be considered even after CR is achieved.