A 55-year-old male patient suffered from left intracranial hemorrhage received craniotomy and removal of intracerebral hematoma. Ventriculoperitoneal shunt was implanted for the complication of hydrocephalus. The skull was put back later because of stable status. He developed scalp necrosis and, therefore several times of debridement was done. The skull was taken away under the suspicion of osteomyelitis. However, he had vomiting, somnolence, urinary incontinence and unable to walk two months after this procedure. Computed tomography showed slit ventricles in right hemisphere. The shunt was then removed and his clinical conditions improved. Slit ventricle syndrome is commonly seen in children, rather than aged persons, with hydrocephalus treated with shunt surgery. We presented this unusual case, which may help clinicians in dealing with such kind of patients.