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Importance Orofacial cleft (OFC) is one of the most common congenital malformations, with a wide variation in incidence worldwide. However, population-based studies on the incidence of OFC in North America are lacking. Objectives To examine the incidence of OFC in Ontario, Canada, and to compare risk factors and mortality associated with children with OFC vs children without OFC. Design, Setting, and Participants This population-based retrospective cohort study used health administrative data from the province of Ontario, Canada. Childre