Introduction: Methotrexate is an essential drug in the treatment of children with acute lymphoblastic leukemia. A complication of methotrexate includes neurotoxicity-it presents with signs and symptoms ranging from headache to strokelike symptoms. Case report: An 11-year-old girl with lymphoblastic lymphoma, who presented a history of leukoencephalopathy, which mimicked stroke one week after receiving high doses of intravenous methotrexate. The patient's symptoms completely improved within two days without sequels. Ten days after a new administration of methotrexate presented right-side hemiparesis and dysarthria. Again, with full resolution. Brain magnetic resonance scan was indispensable for diagnosis. The patient was discharged home without neurological symptoms. Conclusions: Neurotoxicity associated with methotrexate is transient and most patients continue to receive this therapy without intercurrences. It is important that healthcare professionals recognize this entity.