• Pediatr Crit Care Me · May 2010

    Case Reports

    Omphalocele and alveolar capillary dysplasia: a new association.

    • Luella C Gerrits, Amerik C De Mol, Johan Bulten, Frans H Van der Staak, and Arno F J Van Heijst.
    • Department of Neonatology, Radboud University Nijmegen Medical Centre, Nijmegen, The Netherlands.
    • Pediatr Crit Care Me. 2010 May 1;11(3):e36-7.

    ObjectiveFirst report of an infant with coexistent omphalocele and alveolar capillary dysplasia.DesignDescriptive case report.SettingNeonatal intensive care unit of a tertiary care children's hospital.PatientWe describe a term infant with omphalocele and respiratory insufficiency attributable to pulmonary hypertension. The patient was placed on extracorporeal membrane oxygenation, but the pulmonary hypertension persisted. After 10 days on extracorporeal membrane oxygenation, a lung biopsy was performed. It showed alveolar capillary dysplasia. Because of the lethal prognosis, extracorporeal membrane oxygenation was withdrawn and the patient expired.ConclusionsThis is the first description of an association between omphalocele and alveolar capillary dysplasia. In newborns with omphalocele who have severe respiratory insufficiency and pulmonary hypertension, alveolar capillary dysplasia should be considered.

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