Brain abscess is rarely encountered in neonates and carries a high morbidity and mortality. Here we report a premature infant who developed systemic Escherichia coli (E. coli) infection resulting in multiple brain abscesses with intraventricular rupture at 3 months postnatal age. He was treated successfully with a combination of surgical and antimicrobial therapies. Related literature on brain abscess with special emphasis on intraventricular rupture is reviewed.