The authors report on the first case of the intraventricular administration of tigecycline described in the Western literature. A 22-year-old man developed cerebrospinal fluid infection from an extremely drug-resistant strain of Acinetobacter baumannii as a complication of endoscopic transsphenoidal surgery for the removal of a giant pituitary adenoma. The patient was safely and successfully treated with the prolonged intraventricular administration of tigecycline. Here, the authors provide the schedule details that can be invaluable in treating meningitis from extremely drug-resistant bacterial strains. Intraventricular tigecycline can be a valuable tool against multidrug-resistant central nervous system infections.