Stenotrophomonas maltophilia (formerly Xanthomonas maltophilia) is a Gram-negative bacillus increasingly associated with serious nosocomial infections. Here, we describe a 30-year-old male patient who developed meningitis associated with this organism after several neurosurgical procedures. A review of the literature revealed only 15 previous reports. Most cases were associated with neurosurgical procedures. Antimicrobial therapy is complicated by multiple drug resistance of the organism, and trimethoprim-sulfamethoxazole is the recommended agent for treatment. (C) 2008 International Society for Infectious Diseases. Published by Elsevier Ltd. All rights reserved.