Cerebral venous sinus thrombosis (CVST) is an uncommon but recognized complication of treatment for leukemia. Our goal was to determine the long-term neurocognitive outcomes in childhood cancer survivors who had CVST during therapy. Nine patients were identified from an institutional database. All had experienced CVST in the setting of L-asparaginase therapy in combination with other chemotherapeutic agents. Four patients completed neuropsychological evaluation. Their neurological examinations were normal. Neuropsychological testing showed that the participants performed well, with average to above-average scores on cognitive and behavioral testing. Three exhibited difficulties on a visual-motor integration task and 1 had difficulty with fine-motor dexterity, nonverbal memory, emotional control, shifting attention, and anxiety. Overall, by patient and parent report, the survivors had few problems. CVST is a known complication associated with treatment for leukemia and non-Hodgkin lymphoma, most commonly observed if asparaginase is used in combination with other chemotherapeutic agents. Although subtle difficulties were noted in survivors on neuropsychological testing, survivors themselves were not aware of the deficits. Further evaluation of leukemia survivors with a history of CVST is needed to assess for deficits and to understand whether further intervention is necessary.