Carl H Backes1, Erin Nealon2, Aimee K Armstrong3, Clifford L Cua3, Courtney Mitchell4, Usha Krishnan5, Rachel D Vanderlaan6, Mi Kyoung Song7, Nicola Viola8, Charles V Smith9, Patrick I McConnell10, Brian K Rivera4, Jeffrey Bridge11. 1. Department of Pediatrics, Nationwide Children's Hospital and The Ohio State University, Columbus, OH; The Heart Center, Nationwide Children's Hospital, Columbus, OH; Department of Obstetrics and Gynecology, The Ohio State University, Columbus, OH; Center for Perinatal Research, The Research Institute at Nationwide Children's Hospital, Columbus, OH. Electronic address: carl.backesjr@nationwidechildrens.org. 2. Department of Pediatrics, Nationwide Children's Hospital and The Ohio State University, Columbus, OH. 3. Department of Pediatrics, Nationwide Children's Hospital and The Ohio State University, Columbus, OH; The Heart Center, Nationwide Children's Hospital, Columbus, OH. 4. Center for Perinatal Research, The Research Institute at Nationwide Children's Hospital, Columbus, OH. 5. Department of Pediatric Cardiology, Columbia University, New York, NY. 6. Division of Cardiac Surgery, University of Toronto, Hospital for Sick Children, Toronto, Ontario, Canada. 7. Department of Pediatrics, Seoul National University Children's Hospital, Seoul, Republic of Korea. 8. Department of Thoracic and Cardiovascular Surgery, University Hospital of Southampton, Southampton, United Kingdom; Department of Cardiothoracic Surgery, Children's Hospital of Southampton, Southampton, United Kingdom. 9. Center for Developmental Therapeutics, Seattle Children's Research Institute, University of Washington School of Medicine, Seattle, WA. 10. Department of Surgery, The Ohio State University, College of Medicine, Columbus, OH; Center for Pediatric Transplant Research, Nationwide Children's Hospital, Columbus, OH; Department of Cardiothoracic Surgery, Nationwide Children's Hospital, Columbus, OH. 11. Center for Innovation in Pediatric Practice, The Research Institute at Nationwide Children's Hospital, Columbus, OH.
Abstract
OBJECTIVE: To quantify outcomes of infants (<1 year of age) diagnosed with pulmonary vein stenosis (PVS). STUDY DESIGN: MEDLINE (PubMed), Scopus, and Web of Science were searched through February 1, 2017, with no language restrictions. Publications including infants diagnosed with primary PVS, defined as the absence of preceding intervention(s), were considered. The study was performed according to Meta-analysis of Observational Studies in Epidemiology guidelines, the Systematic Reviews, and Meta-Analysis checklist, and registered prospectively. The quality of selected reports was critically examined. Data extraction was independently performed by multiple observers with outcomes agreed upon a priori. Data were pooled using an inverse variance heterogeneity model with incidence of mortality the primary outcome of interest. RESULTS: Forty-eight studies of 185 infants were included. Studies were highly diverse with regards to the participants, interventions, and outcomes reported. The median (range) age at diagnosis was 5.0 (0.1-11.6) months. Pooled mortality was 58.5% (95% CI 49.8%-67.0%, I2 = 21.4%). We observed greater mortality incidence among infants with 3 or 4 vein stenoses than in those with 1 or 2 vein stenoses (83.3% vs 36.1%; P < .01). We observed greater mortality among infants with bilateral than unilateral disease (78.7% vs 26.0%; P < .01). CONCLUSIONS: Studies of primary PVS during infancy are highly variable in their methodological quality and estimates of clinical outcomes; therefore, estimates of prognosis remain uncertain. Multicenter, interdisciplinary collaborations, including alignment of key outcome measurements, are needed to answer questions beyond the scope of available data.
OBJECTIVE: To quantify outcomes of infants (<1 year of age) diagnosed with pulmonary vein stenosis (PVS). STUDY DESIGN: MEDLINE (PubMed), Scopus, and Web of Science were searched through February 1, 2017, with no language restrictions. Publications including infants diagnosed with primary PVS, defined as the absence of preceding intervention(s), were considered. The study was performed according to Meta-analysis of Observational Studies in Epidemiology guidelines, the Systematic Reviews, and Meta-Analysis checklist, and registered prospectively. The quality of selected reports was critically examined. Data extraction was independently performed by multiple observers with outcomes agreed upon a priori. Data were pooled using an inverse variance heterogeneity model with incidence of mortality the primary outcome of interest. RESULTS: Forty-eight studies of 185 infants were included. Studies were highly diverse with regards to the participants, interventions, and outcomes reported. The median (range) age at diagnosis was 5.0 (0.1-11.6) months. Pooled mortality was 58.5% (95% CI 49.8%-67.0%, I2 = 21.4%). We observed greater mortality incidence among infants with 3 or 4 vein stenoses than in those with 1 or 2 vein stenoses (83.3% vs 36.1%; P < .01). We observed greater mortality among infants with bilateral than unilateral disease (78.7% vs 26.0%; P < .01). CONCLUSIONS: Studies of primary PVS during infancy are highly variable in their methodological quality and estimates of clinical outcomes; therefore, estimates of prognosis remain uncertain. Multicenter, interdisciplinary collaborations, including alignment of key outcome measurements, are needed to answer questions beyond the scope of available data.
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