ABSTRACT.Purpose: To investigate the cost and effects of risk factor guided screening strategies for retinopathy of prematurity. Methods: Clinical data from the Netherlands Retinopathy of Prematurity study (NEDROP study) that included all infants screened for ROP and born in 2009 were used to assess the cost and effects of several screening strategies for ROP using different criteria: (1) gestational age (GA), (2) birthweight (BW), (3) combined GA-BW and (4) combined GA-BW and presence of risk factors. Two treatment strategies were evaluated: the infants actually treated in the NEDROP study (n = 17) and all infants detected with severe ROP (n = 29). Results: The most efficient screening strategy to include all infants treated for both treatment strategies is to screen all infants with a GA of 30 weeks or less and a BW of 1250 g or lower together with infants with a GA of 30-32 weeks and a BW of 1250-1500 g with at least one risk factor. The marginal cost ranged from €43 848 to € 226 914 per additional infant with improved vision. Conclusion: The current Dutch guideline may be improved: the same effectiveness can be obtained for lower costs. Releasing the precondition that no infants with severe ROP might be missed will lead to lower costs, but this will also lead to a lower number of infants with improved visual acuity. The costs of detecting all infants with severe ROP seem acceptable for society when the QALY gain and savings from a societal perspective resulting from improved vision are taken into account.