Ependymoma is the third most common CNS tumor in children. Neuraxis dissemination at the time of diagnosis is rare and occurs in fewer than 10% of patients. Recent advances in neuroimaging, neurosurgery, and radiation therapy have improved disease control and functional outcomes for children with ependymoma. However, children under the age of 3 years with ependymoma and leptomeningeal spread historically have had worse outcomes. It is not clear if age alone, or a combination of risk factors such us unfavorable location, which may prevent gross total resection, and withholding radiation therapy may have contributed to poor outcomes in younger age groups. The reason for attempting GTR is based on results from retrospective and prospective studies, where the most important prognostic factor is extent of resection. These patients are not candidates for craniospinal irradiation. Therefore, new therapeutic approaches must be attempted. This is a case report of a posterior fossa ependymoma with leptomeningeal dissemination in a two-year-old child, successfully treated with dose intensive chemotherapy and intrathecal liposomal cytarabine.