IntroductionCommunity-acquired Escherichia coli ventriculitis is considered a rare condition. Central nervous system (CNS) infection due to gram-negative bacilli is usually associated with previous neurosurgical interventions. The recent publication of cases of Escherichia coli meningitis and ventriculitis suggests its prevalence may be underestimated by the literature.Case presentationA case of community-acquired Escherichia coli CNS infection on a 58 year old patient presenting with altered consciousness but without neck stiffness, nor significant past medical history is reported. Imaging and lumbar puncture findings suggested a complex case of meningitis with associated ventriculitis and vasculitis. Escherichia coli was later identified in cultures. Subsequent multi-organ support in Intensive Care was required. The patient was treated with a prolonged course of intravenous antimicrobials guided by microbiology, resulting in some neurological recovery. The main challenges encountered in the management of the patient were the lack of clear recommendations on the duration of treatment and the potential development of multi-resistant organisms.ConclusionBacterial central nervous system infections can have an atypical presentation, and an increasing number of cases of community-acquired ventriculitis have been reported. Early consideration should be given to use magnetic resonance imaging to help guide treatment. A long course of antibiotics is often required for these patients; however, the optimal duration for antimicrobial treatment is not well defined.