Kidney transplantation is the preferred treatment for pediatric end stage renal disease (ESRD). Preemptive transplantation avoids the increased morbidity and mortality of dialysis. Yet, prior studies have not demonstrated significant graft or patient survival benefits for children transplanted preemptively versus non-preemptively. These previous studies were limited by small samples sizes and low rates of adverse events. Here, we compared graft failure and mortality rates, using Kaplan-Meier methods and Cox regression, among a large, national cohort of children with ESRD receiving preemptive versus non-preemptive kidney transplant between 2000 and 2012. Among 7,527 pediatric kidney transplant recipients in the United States Renal Data System, 1668 were transplanted preemptively. Over a median 4.8 years follow-up, 1314 experienced graft failure and over median 5.2 years of follow-up, 334 died. Dialysis exposure versus preemptive transplant conferred higher risk of graft failure (hazard ratio 1.32; 95% confidence interval: 1.10–1.56) and higher risk of death (hazard ratio 1.69; 95% confidence interval: 1.22–2.33) in multivariable analysis. Compared with children transplanted preemptively, children on dialysis for more than one year had 52% higher risk of graft failure and those on dialysis more than 18 months had 89% higher risk of death, regardless of donor source. Thus, preemptive transplantation is associated with substantial benefits in allograft and patient survival among children with ESRD, particularly when compared with children who receive dialysis for over one year. These findings support policies to promote early access to transplant and avoidance of dialysis for children with ESRD whenever feasible.