AIM: To conduct a cost-utility analysis of the tele- retinopathy of prematurity (ROP) screening program against no screening. METHODS: A decision tree model was developed to identify and treat the infants with threshold ROP through the tele-screening system compared with no screening program from the societal perspective. We used the quality adjusted life years (QALY) index to measure the scenarios' effectiveness, which was discounted for the future years by 0.058. One hundred twenty-six randomly selected newborns with ROP required treatment were investigated to extract the treatment information. We considered the direct medical and non-medical costs in cost calculations analysed by the bottom-up approach. The figures of the model's inputs were calculated using the Monte Carlo simulation that generated 1000 random iteration of the data, and a one-way sensitivity analysis was performed on the model to cope with the potential uncertainties. RESULTS: The total and per capita needed the budget to establish a tele-ROP screening system were estimated at over 1.5 million and 35.13 USD, respectively. The total cost of identifying and treating an ROP case in tele-screening and no screening strategies were obtained as 108.72 and 63.52 USD, respectively, and their lifetime discounted QALY gained were calculated as 15.39 and 15.11, respectively. Therefore, incremental cost-effectiveness ratio (ICER) of tele-screening strategy against the competitive strategy was achieved as 161.43 USD. CONCLUSION: Tele-ROP screening program is one of the most cost-effective interventions in the Iranian health system and has a high priority to receive a budget for implementation.