Background: Leptomeningeal inflammation caused by S. Maltophilia is rare. It is inextricably linked with prior antimicrobial therapy, prolonged ICU stays and antecedent neurosurgical intervention. Case description: We describe a case of a 5-year-old male child with posterior fossa medulloblastoma with obstructive hydrocephalus who underwent ventriculoperitoneal shunt followed by suboccipital midline craniotomy and later presented with S. Maltophilia meningitis. Conclusion: The overall mortality in the cases reviewed was 17 per cent. Inherent resistance to a wide array of antimicrobial agents with a simultaneously increasing number of cases poses a therapeutic challenge. Trimethoprim/sulfamethoxazole is recommended as empirical and as a definitive treatment in patients with S. Maltophilia infection. The optimal duration of therapy for S. Maltophilia meningitis is similar to the treatment of gram-negative bacillary meningitis, which is usually 2 weeks after the culture has been negative.