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Osteogenesis imperfecta (OI) is a rare genetic disorder characterized by defects in type I collagen that can pose serious complications during pregnancy. The aim was to evaluate maternal and fetal outcomes in pregnant women with OI.This was a retrospective cohort study, using the Healthcare Cost and Utilization Project Nationwide Inpatient Sample. We examined the records of pregnant women with OI during the period 2003 to 2011. We evaluated antenatal complications and method of delivery among 295 women with OI, using unconditional logistic regression.Of the total 7 287 994 births in our cohort, we encountered 295 deliveries among women with OI. The prevalence was 4 per 1 00 000 deliveries per year over the study period. Births to women with OI were more likely to be complicated by antepartum hemorrhage (odds ratio (OR) 2.01, 95% confidence interval (CI) 1.04 to 3.91), placenta abruption (OR 2.50, 95% CI 1.24 to 5.03), intrauterine growth restriction and small-for-gestational-age infants (OR 2.42, 95% CI 1.42 to 4.14), congenital malformation (OR 7.33, 95% CI 4.20 to 12.78) and preterm birth (OR 2.24, 95% CI 1.63 to 3.06). Seventy-five percent of women with OI delivered by cesarean section, and they had an increased rate of tubal sterilization at delivery (OR 1.67, 95% CI 1.18 to 2.36). No differences in rates of stress fracture and maternal death were found.These findings suggest that there are increased risks to both mother and fetus in pregnancies complicated by OI.