Desmoplastic cerebral astrocytoma of infancy is a massive cystic tumor, typically occurring in the cerebral hemisphere and a rare intracranial tumor that arises in subjects usually below the age of 18 months and shows a favorable prognosis. A 5.5-month-old boy was presented with a rapidly increasing head circumference and lethargy. A computerized tomography scan revealed a large, multicystic, partially enhancing right parieto-occipital mass with hydrocephalus. This patient was treated with a surgery and a firm plaque-like tumor along withmulticystic component was gross totally removed. The tumor mass was firmly attached to the dura and extended in fingerlike projections onto the cortical surface. The infant underwent no further therapy and has developed normally during 6 months after surgery. Desmoplastic cerebral astrocytoma of infancy is a rare intracranial tumor but recognition of this tumor should be appreciated because, despite its massive size and rapidly growing characterstics, this tumor often has a relatively benign clinical course.