Methotrexate (MTX) intrathecal and intravenous administration is associated with neurotoxicity. We report a 15-year old girl diagnosed with large cell B lymphoma. On eighteen-day after intrathecal MTX presented stroke-like symptoms. Magnetic resonance was informed as a possible stroke in the right frontal lobe. The patient does not meet the criteria for fibrinolysis. MTX neurotoxicity was suspected, and theophylline was initiated. She showed complete recovery after three days of treatment. After describing the case, we review MTX neurotoxicity cases from our centre from January 2010 to December 2018. Also, we added to this data the previously published cases in the literature.