Patrick D Evers1, Daksha Ranade2, Mark Lewin1, Bhawna Arya3. 1. Division of Cardiology, Department of Pediatrics, University of Washington School of Medicine and Seattle Children's Hospital, Seattle, Washington. 2. Department of Research Informatics, University of Washington School of Medicine and Seattle Children's Hospital, Seattle, Washington. 3. Division of Cardiology, Department of Pediatrics, University of Washington School of Medicine and Seattle Children's Hospital, Seattle, Washington. Electronic address: bhawna.arya@seattlechildrens.org.
Abstract
BACKGROUND: Coarctation of the aorta (CoA) is difficult to diagnose by fetal echocardiogram (F-Echo), often requiring multiple F-Echos during gestation and neonatal echocardiograms (N-Echos) after birth. Furthermore, CoA is the most common ductal-dependent lesion missed on routine physical exam. OBJECTIVES: We sought to determine the most cost-effective diagnostic approach in caring for infants in whom an initial F-Echo is concerning for CoA. METHODS: Four paradigms for management after initial F-Echo could not rule out CoA were compared, with a single paradigm involving additional F-Echos: (1) multiple F-Echos for diagnostic clarity and performance of N-Echo on neonates with remaining high suspicion for CoA on F-Echos (prenatal-multiple), (2) no further F-Echo and performance of N-Echo on neonates with high suspicion for CoA on initial F-Echo (postnatal-selective), (3) no further F-Echo and performance of N-Echo on all neonates (postnatal-all), and (4) no further F-Echo or N-Echo with reliance on routine physical exam to identify afflicted infants (postnatal-none). Decision analysis models were constructed. Probabilities dictating clinical course and costs were calculated using our institution's study population. The utility-state values were derived from existing literature. The measure of effectiveness was quality-adjusted life years. To represent societal perspectives, cost was defined as hospital reimbursement payments. RESULTS: From 2007 to 2014 at our institution, 92 patients were diagnosed with CoA and met the inclusion criteria for this study. These patients presented to care either through prenatal diagnosis (n = 31), postnatal examination findings while clinically well (n = 41), or after clinical deterioration in extremis (n = 20), with one patient subsequently dying. Presenting in extremis was associated with a 20% increase in the cost of their subsequent care and with a 51% increase in length of hospital stay. Postnatal-none was the least effective paradigm but also the least costly, thus forming the baseline model. Of the three other diagnostic approaches modeled, Postnatal-all was the cost-effective paradigm, maximizing utility due to avoidance of high-cost/low-utility disease states such as presentation in extremis and death. Prenatal-multiple was the next most effective but was also the most expensive. CONCLUSIONS: Echocardiography is the screening gold standard in avoiding the devastating clinical manifestations of a missed CoA. When a diagnosis of CoA cannot be ruled out on initial F-Echo, the most cost-effective approach is performance of N-Echo on all neonates with no further prenatal evaluation.
BACKGROUND:Coarctation of the aorta (CoA) is difficult to diagnose by fetal echocardiogram (F-Echo), often requiring multiple F-Echos during gestation and neonatal echocardiograms (N-Echos) after birth. Furthermore, CoA is the most common ductal-dependent lesion missed on routine physical exam. OBJECTIVES: We sought to determine the most cost-effective diagnostic approach in caring for infants in whom an initial F-Echo is concerning for CoA. METHODS: Four paradigms for management after initial F-Echo could not rule out CoA were compared, with a single paradigm involving additional F-Echos: (1) multiple F-Echos for diagnostic clarity and performance of N-Echo on neonates with remaining high suspicion for CoA on F-Echos (prenatal-multiple), (2) no further F-Echo and performance of N-Echo on neonates with high suspicion for CoA on initial F-Echo (postnatal-selective), (3) no further F-Echo and performance of N-Echo on all neonates (postnatal-all), and (4) no further F-Echo or N-Echo with reliance on routine physical exam to identify afflicted infants (postnatal-none). Decision analysis models were constructed. Probabilities dictating clinical course and costs were calculated using our institution's study population. The utility-state values were derived from existing literature. The measure of effectiveness was quality-adjusted life years. To represent societal perspectives, cost was defined as hospital reimbursement payments. RESULTS: From 2007 to 2014 at our institution, 92 patients were diagnosed with CoA and met the inclusion criteria for this study. These patients presented to care either through prenatal diagnosis (n = 31), postnatal examination findings while clinically well (n = 41), or after clinical deterioration in extremis (n = 20), with one patient subsequently dying. Presenting in extremis was associated with a 20% increase in the cost of their subsequent care and with a 51% increase in length of hospital stay. Postnatal-none was the least effective paradigm but also the least costly, thus forming the baseline model. Of the three other diagnostic approaches modeled, Postnatal-all was the cost-effective paradigm, maximizing utility due to avoidance of high-cost/low-utility disease states such as presentation in extremis and death. Prenatal-multiple was the next most effective but was also the most expensive. CONCLUSIONS: Echocardiography is the screening gold standard in avoiding the devastating clinical manifestations of a missed CoA. When a diagnosis of CoA cannot be ruled out on initial F-Echo, the most cost-effective approach is performance of N-Echo on all neonates with no further prenatal evaluation.