Due to the spread of antimicrobial-resistant bacteria and poor penetration of many antimicrobial drugs across the blood-brain barrier following intravenous administration, treatment of central nervous system (CNS) infections is challenging, especially infections caused by carbapenem-resistant organisms (CRO). Intraventricular (IVT) infusion of antimicrobial drugs could be a choice. This report aims to describe a patient with CNS infection caused by carbapenem-resistant Acinetobacter baumannii (CRAB) which was successfully treated with IVT combined with intravenous (IV) colistin sulfate.A case of CNS infection caused by CRAB after a craniocerebral injury was presented. The patient was treated with IVT together with IV colistin sulfate. Moreover, literature on the regimens and safety of colistin sulfate were also reviewed and summarized.Intraventricular (50,000 U, qd/100,000 U, qd) combined with IV (500,000 U, q12h/500,000 U, q8h) colistin sulfate was given to the patient, and the CNS infection was successfully controlled. The patient was finally transferred back to a local hospital for rehabilitation treatment. No nephrotoxicity or neurotoxicity was observed during the therapy.IV combined with IVT colistin sulfate is effective in the treatment of CNS infections caused by CRAB. IVT concomitant IV colistin sulfate might be a therapeutic option worth considering in the treatment of CNS infections caused by CRO.© 2023 Yang et al.