There have been a number of case reports of antibiotic-associated psychosis in the literature that have not been systematically reviewed. Urinary tract infections (UTIs) are among the most common bacterial infections and have also been associated with acute psychosis. We performed a systematic review of cases of antibiotic-associated acute psychosis during treatment of a UTI and evaluated the strength of the association for each case. We identified reports by searching PubMed, PsychINFO, and Web of Knowledge, and the reference lists of identified reports. We systematically evaluated the quality of the causal relationship between antibiotic treatment of UTI and psychosis. Fourteen articles (describing 15 different cases) met the inclusion criteria. The primary findings were as follows: (1) a majority (60%) of reported cases were "highly suggestive" of a potential causal relationship between antibiotic treatment and psychosis, including 3 cases with a recurrence of psychosis after rechallenge with the same antibiotic; (2) 3 different classes of antibiotics were implicated in this association, including fluoroquinolones, penicillins, and trimethoprim-sulfamethoxazole; (3) for most of the reported cases, both the onset and resolution of psychosis occurred within 1 week of initiation and discontinuation of the antibiotic, respectively; (4) approximately half of the cases did not require treatment with antipsychotics; and (5) affected men were significantly more likely to have a psychiatric history. Our findings suggest that acute psychosis is a potential adverse effect of antibiotic treatment of UTI, although the mechanism(s) underlying this association remains unclear.