Central neurocytomas are typically benign tumors that have high local control rates after gross total resection. Radiotherapy for residual or recurrent central neurocytomas is controversial. We report a 30-year-old male with a central neurocytoma in the lateral ventricle. The tumor was subtotally resected through a transcallosal approach, and subsequently treated with gamma knife radiosurgery. The tumor had shrunken markedly by 2 months after radiosurgery and remained unchanged during the one year follow-up period. Gamma knife radiosurgery may be an option for effective treatment of patients with residual or recurrent central neurocytomas.