OBJECTIVES: The main objective is to assess resource utilization and cost of schizophrenia as well as cost drivers using a prospective cohort that followed a representative sample of 288 French patients. METHODS: This study is the first comprehensive prospective study on cost of schizophrenia.  Patients were randomly selected from three defined catchment areas located in the North, Centre and South of France. Unit costs were based on the most valid available data, mainly health insurance and French public sources. Costs were defined based on society perspective. Direct cost included treatment provided in inpatient services, intermediate facilities, outpatient visits and medication costs. Indirect costs were considered as productivity loss for unemployed or sick leave  patients in working age. RESULTS: Average patient direct costs yield €3534, break down as 39.3% for inpatient treatment, 37.7% for day clinic, and 16.1% for medication. The remaining includes visits to psychiatrists, GP’s, psychologists and other physicians. Assuming 1% prevalence of schizophrenia, the estimation of annual direct costs yields €1, 581, 111, 600, and indirect cost of schizophrenia linked to productivity loss in France yields €2, 214, 488, 006. Several cost drivers were identified: relapse during the follow-up period, positive symptoms of schizophrenia, and depressive symptoms at baseline predict higher costs, while satisfaction with their health or negative symptoms of schizophrenia at baseline was linked with lower costs. CONCLUSIONS: This study highlights the heavy societal burden of schizophrenia in France, of which hospitalization (day and full time) services account for 77%. Any attempt to anticipate and prevent hospitalization might have significant effect on the cost of schizophrenia. Such intervention is likely to be cost effective.