OBJECTIVES: To assess the cost-effectiveness of benzonidazole versus no specific treatment for Chagas disease in Brazil. METHODS: A Markov model was constructed to compare the differences in costs and effects between a cohort of patients treated with benzonidazole and other cohort of patients to whom no specific treatment was offered for Chagas' disease. For the patients treated with benzonidazole, a 60% treatment effectiveness rate was used in the base case analysis, in accordance with the world health organization report. A third payer lifetime perspective was adopted for a patient infected with Chagas disease who, following the model proposed by Schofield (1991), was infected with 15 years and dies with 52 years, on average. The Markov cycle duration adopted was 1 year. For cost data, an expert Delphi panel was performed with Brazilian experts on treating Chagas disease. A 3% annual discount rate was adopted for both costs and benefits, to reflect social preference. RESULTS: The mean cost per patient treated with benzonidazole was R$ 1,325, while the mean cost per patient to whom no specific treatment was given was R$ 2,281. The death risk of patients treated with benzonidazole was 51% smaller than the risk of death in patients to whom no specific treatment was given. Due to a high variability of the effectiveness rate found in the literature, a sensitivity analysis was conducted to test the robustness of the model. Varying the rate between 30% and 80%, the mean cost per patient treated with benzonidazole found was between R$ 2,010 and R$ 869, which is below the cost of offering no treatment for the patients. The ICER for benzonidazole versus no treatment was – R$ 3,033 per life year gained (LYG). CONCLUSION: Benzonidazole reduced medical resource utilization for patients halved death risk. Findings suggest Benzonidazole to be a cost-saving alternative for the Brazilian setting.