OBJECTIVES: Although the economic burden of brain tumors is potentially important, little is known about the cost of these cancers in Canada. The objective of this study was to estimate the current economic burden associated with primary brain cancers in Canada. METHODS: We used a prevalence-based cost-of-illness framework to estimate the annual cost due to primary brain tumors in the Canadian population. In order to retrieve clinical epidemiological and healthcare resources utilization data, relevant studies were identified with MEDLINE and EMBASE databases and Canadian Cancer statistics databases were consulted. The analysis was carried out from a societal perspective. Costs were extracted and converted to 2015 prices using the Canadian health component of the consumer price index. For each cost category, the estimated quantity for each resource was multiplied by the unit cost and then summed to get the total costs attributable to brain tumors in Canada. RESULTS: Prevalence of primary brain tumors in Canada was estimated to 2,289 prevalent cases. Costs related to productivity losses for primary brain tumors represented 46% of the total costs. Direct costs were estimated to 54% of the total costs, of which the costs associated with chemotherapy represented 52% of all direct costs, while hospitalization, imagery, medical visits, radiotherapy, surgery and concomitant treatments represented 30%, 5%, 5%, 4%, 3% and 1% of all direct costs respectively. The total cost of brain tumors in Canada was estimated at CAN$321,119,700 and the average cost per patient was CAN$140,288.  CONCLUSIONS: The primary brain cancers are scarce but have high societal and economic impacts. This economic burden of primary brain cancers provides a comprehensive overview of the costs associated with these conditions.