Objective: The proportion of patients with schizophrenia considered as being non-compliant with antipsychotic medications ranges from 20% to 89%. Non-compliance has been associated with increase relapse rates, more frequent and longer hospitalizations. Costs of schizophrenia in the U.S. vary from $32 to $65 billion, but contradictory results have been reported for the costs of non-compliance among these patients. The objective was to estimate the costs of non-compliance with antipsychotic medications among patients with schizophrenia. Methods: A literature review of studies published in the last decade was conducted. We utilized PubMed, Cochrane, EMBASE and CINADHL databases. The key terms included: (first: adherence, non-adherence, compliance, non-compliance), AND (second: schizophrenia, antipsychotics, atypical and typical antipsychotics, neuroleptics) AND (third: resource utilization, resource use, costs, economics, hospitalizations). Studies with data on costs of non-compliance or compliance were included. Two independent researchers reviewed the titles and abstracts and any differences were agreed by consensus. Costs were transformed in 2007 US dollars using the medical care price index. Results: Of 43 studies, six fulfilled the inclusion criteria, including five retrospective cohort studies that used Medicaid paid claims, and a cross-sectional study that used the United Kingdom Psychiatric Morbidity Survey. Most studies had <3 years of follow-up. A total of 52,512 participants were studied. The percentage of non-compliant patients ranged from 11% to 31%. A study found that users of atypical antipsychotics (72%) have higher compliance than users of typical antipsychotics (56%). Non-compliant patients spent, on average, ten more days in the hospital, and have 22% more rehospitalizations than compliant patients. Costs of care for non-compliant patients were on average $3,104 higher than for compliant patients. Conclusion: Patients non-compliant with antipsychotics had more and longer hospitalizations, and higher total costs than compliant patients. Non-compliance with antipsychotics has significant clinical and economic implications for patients with schizophrenia.