The TRIVIA cohort for surgical management of tetralogy of Fallot: Merging population and clinical data for real-world scientific evidence

Abstract Background Guidelines for surgical management of tetralogy of Fallot (TOF) are often based on low-quality evidence due to the many challenges of congenital heart disease: heterogeneous cardiac anatomy, consequences from surgical interventions arising years later, and scarcity of hard outcomes. The overarching goal of the TRIVIA study is to evaluate the long-term impact of the surgical management strategies in tetralogy of Fallot. The specific objectives are: 1) to describe the long-term outcomes of TOF according to the native anatomy and the presence of genetic conditions, 2) to evaluate the long-term outcomes of surgical repair according to associated residual lesions, and 3) to evaluate the impact of pediatric pulmonary valve replacements on the long-term outcomes. Methods The TRIVIA study is a population-based cohort including all TOF subjects in the province of Quebec between 1980 and 2017. It links patient-level granular clinical data with long-term administrative healthcare data. We will evaluate mortality, cardiovascular interventions and hospitalizations for adverse cardiovascular events using survival Cox models and marginal mean/rates models for recurrent events, respectively. Multivariate multilevel models will correct for potential confounders and risk score matching will mitigate the potential of confounding by indication. Results The current TRIVIA cohort includes 1,001 eligible TOF subjects with complete lifelong follow-up for >98%. The median follow-up is 17.1 years, totaling >17,000 patient-years. Conclusion Universal health insurance data combined with granular clinical data enables the development of population-based cohorts, to which contemporary statistical methods are applied to address important research questions in congenital heart disease research.