AimsTo explore the relationship between preconception severe hypoglycemia (PSH) and pregnancy outcomes in pregnancies complicated with type 1 diabetes mellitus (T1DM).Materials and MethodsIn this multicenter prospective cohort study, women with pregestational T1DM were stratified by episodes of severe hypoglycemia within 1 year before conception: No PSH, sporadic PSH (1–6 times/year), and recurrent PSH (>6 times/year). We analysed the predictive ability of PSH for maternal and neonatal outcomes using log‐binomial regression models and receiver operating characteristic (ROC) curve.ResultsOf the 124 women studied, 37.1% experienced at least one episode of severe hypoglycemia preconception. In the multiple adjusted regression models, recurrent PSH was significantly associated with increased incidence of preeclampsia (RR 17.59, 95% CI: 2.89–150.62, p for trend = 0.007), preterm birth (RR 6.34, 95% CI: 1.22–40.63, p for trend = 0.027), neonatal hypoglycemia (RR 4.52, 95% CI: 1.14–17.16, p for trend = 0.017), neonatal hyperbilirubinemia (RR 4.12, 95% CI: 1.11–15.56, p for trend = 0.004), and composite neonatal outcome (RR 3.85, 95% CI: 1.01–19.61, p for trend = 0.003). In the ROC analysis, PSH predicted preeclampsia, preterm birth, neonatal hypoglycemia, neonatal hyperbilirubinemia, and composite neonatal outcome with areas under the ROC curve all ≥0.6.ConclusionsRecurrent preconception severe hypoglycemia is associated with increased risks of adverse outcomes in pregnant women with T1DM.