A 4-month-old female patient admitted because of hydrocephalus. She was premature infant with germinal matrix hemorrhage at gestational period 27 wks. A ventriculoperitoneal shunt with a low-pressure type valve was placed. Follow-up CT scan obtained 3 months after VP shunt placement, revealed a new Lt. middle cranial fossa cyst. The girl was readmitted to hospital at 7 months after VP shunt placement, complaining of lethargy and vomiting. A CT scan was demonstrated a increase in size of a cyst, She subsequently underwent a left-sided pterional craniotomy with partial excision of membrane and cyst fenestration into the basal cisterns. Follow-up CT scan after 18 months revealed reaccumulation of cyst, the girl was reoperated on. After 2 months, the size of cyst was not decreased. Finally, She underwent a cystoperitoneal shunt. Follow-up CT scan after 5 months was demonstrated disappeared cyst and reexpansion of brain parenchyma. We report the development of a symptomatic sylvian fissure arachnoid cyst developed after ventriculoperitoneal shunt.