A 23-year-old male with non-communicating hydrocephalus caused by intraventricular hemorrhage from an arteriovenous malformation was treated by endoscopic third ventriculostomy (ETV) after repeated ventricular cerebrospinal fluid drainage and removal of ventriculoperitoneal shunt due to infection. Postoperatively, the patient developed robust rigidity and akinesia with mutism, suggesting secondary parkinsonism. These symptoms showed marked improvement after administration of levodopa, and were not aggravated after discontinuation. The pathophysiology of this case might involve dysfunction of the dopaminergic system caused by repeated compression and release of the paraventricular white matter. Careful perioperative management not leading to excessive dilation of the the ventricles should be considered for ETV for non-communicating hydrocephalus to avoid this unexpected complication.