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Primary pulmonary vein stenosis among premature infants with single-vessel disease

Eli Zettler1, Brian K Rivera1, Corey Stiver2

  • 1Center for Perinatal Research, The Abigail Wexner Research Institute at Nationwide Children's Hospital, Columbus, OH, USA.

Journal of Perinatology : Official Journal of the California Perinatal Association
|September 29, 2020
PubMed

Insights

Conservative management of single-vessel primary pulmonary vein stenosis (PPVS) in preterm infants showed disease progression in half of cases. Early risk stratification is crucial for targeted therapies in these infants.

Area of Science:

  • Pediatric Cardiology
  • Neonatology
  • Vascular Biology

Background:

  • Single-vessel primary pulmonary vein stenosis (PPVS) is a rare condition affecting preterm infants.
  • Conservative management, involving active surveillance and deferred treatment, is a common initial approach.
  • Understanding outcomes in this cohort is essential for optimizing care.

Purpose of the Study:

  • To describe the outcomes of preterm infants with single-vessel PPVS initially managed conservatively.
  • To identify factors associated with disease progression versus stabilization.
  • To inform risk stratification and therapeutic strategies.

Main Methods:

  • Retrospective cohort study of infants <37 weeks' gestation diagnosed with single-vessel PPVS between 2009-2019.
  • Classification into disease progression or stabilization groups.
  • Analysis of cardiopulmonary outcomes and Kaplan-Meier survival analysis.

Main Results:

  • Twenty infants were included; 10 showed disease progression and 10 remained stable.
  • All infants in the progressive group developed severe stenosis or atresia (100%), versus 0% in the stable group (P<0.01).
  • Severe pulmonary hypertension was more frequent in the progressive group (50% vs 0%, P=0.03), with lower survival (P<0.01).

Conclusions:

  • Conservative management of single-vessel PPVS in preterm infants carries a significant risk of progression.
  • Risk stratification appears feasible, potentially guiding individualized treatment approaches.
  • Further research is needed to refine therapeutic strategies based on risk profiles.
Abstract

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