The treatment of multidrug-resistant Acinetobacter baumannii infections is a serious problem in neonatal Intensive Care Units. Colistin has been used successfully in the treatment of these infections. Due to the limited penetration of colistin into the cerebrospinal fluid, central nervous system infection due to A. baumannii necessitates intrathecal or intraventricular application. Here, we describe our experience with intravenous and intraventricular colistin administration in a premature infant (33 gestational weeks) with shunt infection and brain abscess. No side-effects related to the drug were observed. To the best of our knowledge, our patient is the first premature infant to undergo intraventricular colistin administration. Although the central nervous system infection in our patient was treated successfully, it is obvious that detailed prospective studies are required regarding colistin usage in neonates and especially in premature infants.