Objective To systematically review the effects of cost sharing in health insurance schemes, so as to provide evidence for better designing cost sharing ratio in health insurance scheme. Methods The search terms were discussed, tested and then chosen by subject matter experts and search coordinators. The total 20 databases including comprehensive scope, health, economics, sociology, and grey literatures were searched to retrieve all the description or evaluation studies on the effects of cost sharing, such as health services utilization, financial burden or moral hazard. The information from the included studies was extracted into a pre-designed data extraction form, and then it was analyzed and summarized. Results A total of 73 studies were included, covering 17 countries like Australia, Canada, and China, etc. The results of statistical analyses showed that, a) Cost sharing methods were applied to every kind of health insurance scheme. The target population included general population, the elder, the poor, those with chronic disease and children, etc. The services covered clinic, hospitalization, mental health, prevention and drug; and b) The effects brought from cost sharing included: From full fee to cost sharing scheme, the enrollee in developing countries increased their health care utilization, and decreased their financial burden. From full coverage to cost sharing, the utilization of health services decreased in developed countries, but the cost of health insurance could not be reduced, and some undesirable effects were brought due to the decrease of both essential health service utilization and essential drugs compliance.