Desmoplastic cerebral astrocytoma of infancy is a rare neoplasm with distinctive clinical and rad-iographic features. Almost all cases present in the first two years of life as a large, supratentorial, enhancing mass with a peripheral dural-based solid component and a central cystic component. After complete surgical excision, the course of this tumor is benign, and adjuvant chemotherapy or radiation therapy may not be required. We describe the case of a 9-month-old boy with a desmoplastic cerbral astrocytoma of infancy and review the associated literature.