A 41-year-old woman suffered fever, speech disturbance and confusion four months after transsphenoidal surgery for pituitary macroadenoma. Meanwhile, the patient had been well except for transient asymptomatic postoperative pneumocephalus without cerebrospinal fluid (CSF) rhinorrhoea. Brain computed tomographic scan revealed a peripherally enhancing pineal abscess which superimposed on a preexisting pineal cyst. CSF findings showed elevated leukocyte count and positive bacterial culture. Three weeks of intensive antibiotic therapy and surgical evacuation of the pineal abscess ensued. The patient showed restricted postoperative recovery with moderate hypothalamic dysfunction. A unique case of delayed-onset postoperative pineal abscess which complicates the preexisting asymptomatic pineal cyst is presented and the possible explanation for this is speculated with review of the available literatures.