In pediatric age group, two thirds of the ependymomas are located in posterior fossa and less frequently arise from supratentorial or spinal intramedullar region. Patient age, degree of resection and histopathologic subtype are among the prognostic criteria. Most of the patients with intracranial anaplastic ependymoma are candidate for radiotherapy. The role of chemotherapy remains controversial though benefits have been suggested in some studies. In the present study, 2 patients with anaplastic ependymoma, under the age of 3 years at the time of diagnosis are presented. Treatment was started with chemotherapy and radiotherapy was delivered after 36 months in both patients. Adjuvant chemotherapy and consecutive radiotherapy seem effective in patients with anaplastic ependymoma under the age of 3 years until more effective treatment methods are developed.