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Introduction: Stroke is an uncommon yet serious complication associated with cardiac catheterization in children with congenital heart disease. Our objective was to estimate the frequency and predictors of arterial ischemic stroke (AIS) in this specific setting.Methods: We performed a case-control study of children with congenital heart disease, who developed stroke in relation to cardiac catheterization performed at the Hospital for Sick Children between 1 January, 1992 and 1 January, 2014. Cases had new AIS (either symptomatic or silent) within 72 hours of cardiac catheterization and controls had cardiac catheterization but no new stroke within this time window. We estimated the frequency of AIS in children who underwent cardiac catheterization in this study period. Predictors of stroke that were tested include age, nature of intra-cardiac shunt, pre-procedural peripheral capillary oxygen saturation (SpO2), type of catheterization (diagnostic versus interventional), duration of procedure, right versus left heart protocol, procedural anticoagulation and major/minor complications. Pediatric Stroke Outcome Measure was used to assess the neurological outcome of cases.Results: Twenty eight children (mean age1.66years; 64% males) developed stroke among 19414 children who underwent cardiac catheterization during the study period. The frequency of cardiac catheterization related-stroke in children is 1.44/1000 cardiac catheterizations. Univariate analysis revealed that younger age (OR 1.22; 95% CI (1.02,1.45); p=0.030), low SpO2pre-catheterization (OR 1.08; 95% CI (1.03,1.12); p=0.004), lack of procedural anticoagulation with heparin (OR 3.57; 95% CI (1.04,11.76); p=0.044), and minor complications during catheterization (OR 7.15; 95% CI (1.51,33.79); p=0.013) were associated with stroke. Low SpO2pre-catheterization and lack of proceduralanticoagulation remained significant in multivariate analysis.Conclusion: The frequency of stroke after cardiac catheterization in children with congenital heart disease is 1.44/1000 cardiac catheterizations. Low SpO2 pre-catheterization and lack of procedural anticoagulation appear to be associated with a higher risk of stroke.