This case report describes a 56-year-old woman with multi-drug-resistant Pseudomonas aeruginosa ventriculitis secondary to external ventricular drain insertion who was successfully treated with intrathecal colistin, intravenous colistin, and intravenous meropenem. A review of the English-language literature found six similar cases. While the patient did not experience side effects, self-limited paresthesias of the arm were noted in one case report. Intrathecal colistin should be considered as a therapy for ventriculitis when antibiotic choices are limited by resistant organisms. Patients should be monitored for neurological side effects during therapy.