The clinical and diagnostic findings and the factors influencing the neurologic and radiologic outcome of symptomatic ischemic stroke were evaluated in a group of 2,318 children with acute lymphoblastic leukemia (ALL) treated according to the AIEOP (Italian Association of Pediatric Hematology and Oncology) study protocols. In this multicentric retrospective study, a questionnaire was sent to each of the 43 AIEOP centers participating in the study. The questionnaire was designed to obtain information on the number, type, and time of occurrence of ischemic strokes, biologic and immunologic features of each case, as well as clinical data of the recruited patients. A prevalence of 0.47% was found. All ischemic strokes were sinovenous thrombosis (SVT). The most common neurologic presentations were diffuse neurologic signs and seizures. MRI with or without venography revealed SVT in 100% of cases; superficial SVT was diagnosed in the majority of cases. Antithrombotic drugs, in particular unfractioned heparin and low-molecular-weight heparin, were administered without bleeding complications. This series shows an excellent long-term neurologic outcome in children with SVT. However, a complete radiologic resolution was found in only 54% of cases; the involvement of deep cerebral venous sinuses was associated with an unfavorable imaging outcome.