Methotrexate (MTX)-associated lymphoproliferative disorder (MTX-LPD) is a known adverse event of MTX and rarely originates in the central nervous system (CNS). There are few case reports related to CNS LPDs; therefore, an effective treatment has not been established. We report a case of primary CNS MTX-LPD treated with a new combination chemotherapy consisting of rituximab, procarbazine, methotrexate, and vincristine (R-MPV therapy). A 56-year-old woman presented with chief complaints of headache, memory disturbance, and gait disturbance. She had taken MTX for rheumatoid arthritis for 25 years. Magnetic resonance imaging demonstrated tumor lesions in the left frontal and occipital lobes. The tumor was removed via craniotomy and diagnosed as MTX-LPD. MTX therapy was discontinued, but no tumor regression was observed. Although R-MPV therapy was started, no obvious regression was obtained. The patient died after 4 courses of R-MPV therapy. If chemotherapy achieves limited effects for patients with CNS MTX-LPD, early introduction of radiotherapy or change to other chemotherapy should be considered.
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