This report describes a rare case of postoperative hyperventilation attack after an endoscopic third ventriculostomy in a 46-year-old woman. About 60 min after the termination of the operation, an intractable hyperventilation started with respiratory rate of 65 breaths/min and $EtCO_2$, 16.3 mm Hg. Sedation with benzodiazepine, thiopental sodium, fentanyl, and propofol/remifentanil infusion was tried under a rebreathing mask at a 4 L/min of oxygen. With aggressive sedative challenges, ventilation pattern was gradually returned to normal during the 22 hrs of time after the surgery. A central neurogenic hyperventilation was suspected due to the stimulating central respiratory center by cold acidic irrigation solution during the neuroendoscopic procedure.