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Pulmonary Vein Stenosis in Infants: A Systematic Review, Meta-Analysis, and Meta-Regression
Carl H Backes1, Erin Nealon2, Aimee K Armstrong3
1Department 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.
Insights
Infants with pulmonary vein stenosis (PVS) have a high mortality rate of 58.5%. Outcomes worsen with more severe or bilateral PVS, highlighting the need for better research.
Area of Science:
- Pediatric Cardiology
- Neonatal Health
- Vascular Biology
Background:
- Pulmonary vein stenosis (PVS) is a rare but serious condition in infants.
- Understanding the outcomes and prognosis of PVS is crucial for early intervention and management.
- Primary PVS in infants presents unique challenges due to its congenital nature and potential for rapid progression.
Purpose of the Study:
- To quantify the outcomes of infants diagnosed with pulmonary vein stenosis (PVS).
- To determine the mortality incidence in infants with primary PVS.
- To identify factors influencing mortality in this vulnerable population.
Main Methods:
- A systematic literature search was conducted across MEDLINE (PubMed), Scopus, and Web of Science up to February 1, 2017.
- Included studies focused on infants (<1 year) with primary PVS, defined as absence of prior interventions.
- Data were pooled using an inverse variance heterogeneity model, with mortality as the primary outcome.
Main Results:
- Forty-eight studies comprising 185 infants were analyzed.
- The pooled mortality incidence for infants with PVS was 58.5%.
- Higher mortality was observed in infants with 3-4 vein stenoses (83.3%) versus 1-2 (36.1%) and in those with bilateral (78.7%) versus unilateral disease (26.0%).
Conclusions:
- Estimates of prognosis for primary PVS in infancy remain uncertain due to variability in study quality and outcomes.
- Multicenter, interdisciplinary collaborations are essential to standardize measurements and improve understanding.
- Further research is needed to address the limitations of current data and improve clinical outcomes for infants with PVS.
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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