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CASE REPORT
Korean J Pediatr 2012 August;55(8) :297-300.
Published online 2012 August 15.        doi:https://doi.org/10.3345/kjp.2012.55.8.297
Bronchial compression in an infant with isolated secundum atrial septal defect associated with severe pulmonary arterial hypertension
Sung-Hee Park (Park SH)1, So Young Park (Park SY)1, Nam Kyun Kim (Kim NK)1, Su-Jin Park (Park SJ)1, Han Ki Park (Park HK)2, Young Hwan Park (Park YH)2, Jae Young Choi (Choi JY)1
1Division of Pediatirc Cardiology, Congenital Heart Disease Center, Severance Cardiovascular Hospital, Department of Pediatrics, Yonsei University Collage of Medicine, Seoul, Korea
2Division of Cardiovascular Surgery, Congenital Heart Disease Center, Severance Cardiovascular Hospital, Department of Thoracic and Cardiovascular Surgery, Yonsei University Collage of Medicine, Seoul, Korea
Corresponding Author: Jae Young Choi ,Tel: +82-2-2227-4543, Fax: +82-2-312-9538, Email: cjy0122@yuhs.ac
Copyright © 2012 by The Korean Pediatric Society
ABSTRACT
Symptomatic pulmonary arterial hypertension (PAH) in patients with isolated atrial septal defect (ASD) is rare during infancy. We report a case of isolated ASD with severe PAH in an infant who developed airway obstruction as cardiomegaly progressed. The patient presented with recurrent severe respiratory insufficiency and failure to thrive before the repair of the ASD. Echocardiography confirmed volume overload on the right side of heart and severe PAH (tricuspid regurgitation [TR] with a peak pressure gradient of 55 to 60 mmHg). The chest radiographs demonstrated severe collapse of both lung fields, and a computed tomography scan showed narrowing of the main bronchus because of an intrinsic cause, as well as a dilated pulmonary artery compressing the main bronchus on the left and the intermediate bronchus on the right. ASD patch closure was performed when the infant was 8 months old. After the repair of the ASD, echocardiography showed improvement of PAH (TR with a peak pressure gradient of 22 to 26 mmHg), and the patient has not developed recurrent respiratory infections while showing successful catch-up growth. In infants with symptomatic isolated ASD, especially in those with respiratory insufficiency associated with severe PAH, extrinsic airway compression should be considered. Correcting any congenital heart diseases in these patients may improve their symptoms.
Keywords: Secundum atrial septal defect | Pulmonary arterial hypertension | Airway obstruction | Infant
 
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