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Aortic coarctation associated with alveolar capillary dysplasia and misalignment of the pulmonary veins.

Abstract
After surgical repair of an aortic coarctation a term infant presented with severe pulmonary hypertension and cyanosis unresponsive to treatment including extracorporeal membrane oxygenation. The atypical clinical course became apparent once the accompanying diagnosis of congenital alveolar capillary dysplasia with misalignment of the pulmonary veins had been established at autopsy. In infants with congenital heart defects and with refractory pulmonary hypertension unexplainable on anatomic findings, a lung biopsy at the time of cardiac repair should be considered to avoid further therapies that would not alter the uniformly fatal course of this rare lung disorder.
AuthorsAndreas Gamillscheg, Gerfried Zobel, Ekkehard Spuller, Friedrich Reiterer, Albrecht Beitzke
JournalPediatric cardiology (Pediatr Cardiol) Vol. 29 Issue 1 Pg. 191-4 (Jan 2008) ISSN: 0172-0643 [Print] United States
PMID17874220 (Publication Type: Case Reports, Journal Article)
Topics
  • Aortic Coarctation (epidemiology, surgery)
  • Dilatation, Pathologic
  • Extracorporeal Membrane Oxygenation
  • Fatal Outcome
  • Female
  • Humans
  • Infant, Newborn
  • Lymphatic Vessels (pathology)
  • Persistent Fetal Circulation Syndrome (etiology, therapy)
  • Pulmonary Alveoli (pathology)
  • Pulmonary Veins (abnormalities)

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