BACKGROUND: Parkinson disease (PD) is a chronic multifaceted neurodegenerative disorder of adult onset that affects quality of life and places a burden on patients, caregivers, and society. In early disease, dopaminergic therapy improves motor symptoms, but as the disease progresses, symptoms tend to increase in frequency and severity, even with best medical treatment (BMT). Deep brain stimulation (DBS) becomes an option for certain patients, but cost becomes an important issue. OBJECTIVE: We performed a systematic review of the literature of economic studies of the use of DBS in patients with PD, including costs studies or economic evaluations expressed as cost per improvement in quality life, decrease in dose of pharmacological treatments, and the decrease of caregiver burden. METHODS: We reviewed the following databases: Medline/PubMed, Embase, Cochrane Database of Systematic Reviews, LILACS, Cochrane Central Register of Controlled Trials, WHO International Clinical Trials Registry Platform ICTRP portal and ClinicalTrials.gov from 1980 to 2015. Costs have been converted or adjusted to 2016 US dollars (US$). RESULTS: Nine studies were identified. The average cost of DBS for a patient with PD in 5 years is US$186,244. The quality-adjusted life year was higher in DBS compared with BMT after at least 2 years of treatment, with an average incremental cost utility ratio of US$41,932 per additional quality-adjusted life year gained. Costs in the first year are higher with DBS because of direct costs related to the surgical procedure, the device, and the more frequent controls. Studies show better results with a longer time horizon (up to 5 years). CONCLUSION: DBS is a cost-effective intervention for patients with advanced PD, but it has a high initial cost compared with BMT. However, DBS reduces pharmacologic treatment costs and should also reduce direct, indirect, and social costs of PD on the long term.