Eli Zettler1, Brian K Rivera1, Corey Stiver2, Brian Boe2, Clifford Cua2, Molly K Ball3, Charles V Smith4, Jonathan L Slaughter1,3,5,6, Bernadette Chen1,3,5, Ryan Callahan7, Carl H Backes8,9,10,11. 1. Center for Perinatal Research, The Abigail Wexner Research Institute at Nationwide Children's Hospital, Columbus, OH, USA. 2. The Heart Center at Nationwide Children's Hospital, Columbus, OH, USA. 3. Division of Neonatology, The Ohio State University Wexner Medical Center, Columbus, OH, USA. 4. Seattle Children's Research Institute, University of Washington School of Medicine, Seattle, WA, USA. 5. Department of Pediatrics, The Ohio State University College of Medicine, Columbus, OH, USA. 6. Division of Epidemiology, The Ohio State University College of Public Health, Columbus, OH, USA. 7. Department of Cardiology, Boston Children's Hospital and Harvard Medical School, Boston, MA, USA. 8. Center for Perinatal Research, The Abigail Wexner Research Institute at Nationwide Children's Hospital, Columbus, OH, USA. Carl.BackesJr@nationwidechildrens.org. 9. The Heart Center at Nationwide Children's Hospital, Columbus, OH, USA. Carl.BackesJr@nationwidechildrens.org. 10. Division of Neonatology, The Ohio State University Wexner Medical Center, Columbus, OH, USA. Carl.BackesJr@nationwidechildrens.org. 11. Department of Pediatrics, The Ohio State University College of Medicine, Columbus, OH, USA. Carl.BackesJr@nationwidechildrens.org.
Abstract
OBJECTIVES: Describe outcomes among preterm infants diagnosed with single-vessel primary pulmonary vein stenosis (PPVS) initially treated using conservative management (active surveillance with deferral of treatment). STUDY DESIGN: Retrospective cohort study at a single, tertiary-center (2009-2019) among infants <37 weeks' gestation with single-vessel PPVS. Infants were classified into two categories: disease progression and disease stabilization. Cardiopulmonary outcomes were examined, and a Kaplan-Meier survival analysis performed. RESULTS: Twenty infants were included. Compared to infants in the stable group (0/10, 0%), all infants in the progressive group had development of at least severe stenosis or atresia (10/10, 100%; P < 0.01). Severe pulmonary hypertension at diagnosis was increased in the progressive (5/10, 50%) versus the stable group (0/10, 0%; P = 0.03). Survival was lower among infants in the progressive than the stable group (log-rank test, P < 0.01). CONCLUSION: Among preterm infants with single-vessel PPVS, risk stratification may be possible, wherein more targeted, individualized therapies could be applied.
OBJECTIVES: Describe outcomes among preterm infants diagnosed with single-vessel primary pulmonary vein stenosis (PPVS) initially treated using conservative management (active surveillance with deferral of treatment). STUDY DESIGN: Retrospective cohort study at a single, tertiary-center (2009-2019) among infants <37 weeks' gestation with single-vessel PPVS. Infants were classified into two categories: disease progression and disease stabilization. Cardiopulmonary outcomes were examined, and a Kaplan-Meier survival analysis performed. RESULTS: Twenty infants were included. Compared to infants in the stable group (0/10, 0%), all infants in the progressive group had development of at least severe stenosis or atresia (10/10, 100%; P < 0.01). Severe pulmonary hypertension at diagnosis was increased in the progressive (5/10, 50%) versus the stable group (0/10, 0%; P = 0.03). Survival was lower among infants in the progressive than the stable group (log-rank test, P < 0.01). CONCLUSION: Among preterm infants with single-vessel PPVS, risk stratification may be possible, wherein more targeted, individualized therapies could be applied.
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