The sylvian aqueduct syndrome is a global rostral midbrain dysfunction induced by a transtentorial pressure gradient through the aquaeductus. Several months after ventriculoperitoneal shunt, a patient with hydrocephalus began experiencing a constellation of midbrain dysfunction symptoms, including bradykinesia, medial longitudinal fasciculus syndrome, third nerve palsy, and mutism. These were indicative of cerebral aqueduct syndrome. In addition, the patient showed posture-dependent underdrainage or overdrainage. All symptoms were resolved after distal catheter elongation and floating cranioplasty. We present a case of reversible parkinsonism, which developed in a patient with shunted hydrocephalus and aqueductal stenosis, and discuss the diagnosis and treatment of the sylvian aqueduct syndrome. We also review the literature to address problems of drainage and potential treatment modalities.