First Description of Coronary Artery Ostial Atresia With Fistulous Origin From a Normal Right Ventricle
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CASE REPORT
First Description of Coronary Artery Ostial Atresia With Fistulous Origin From a Normal Right Ventricle Olawale Olabiyi • Debra Kearney • Rajesh Krishnamurthy • David Morales Antonio G. Cabrera
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Received: 6 March 2012 / Accepted: 27 June 2012 Ó Springer Science+Business Media, LLC 2012
Abstract Anomalous origins of both the left and right coronary arteries are rare but have been well documented when both arteries arise from the pulmonary trunk (Angelini et al., Circulation 105:2449–2454, 2002). An anomalous coronary arterial origin from the pulmonary arteries usually involves the left coronary artery (ALCPA) and less frequently the right coronary artery (ARCPA). At least three cases have been reported in which the right coronary artery arose abnormally from the left ventricle (LV), but none have been reported in which both coronary arteries took their origin from the right ventricle (Ippisch and Kimball, J Am Soc Echocardiogr 23:222.e1–222.e2, 2010; Okuyama et al., Jpn Heart J 36:115–118, 1995; Culbertson et al., Pediatr Cardiol 16:73–75, 1995). Ostial atresia with anomalous origin of a coronary artery from the right ventricle has been described only in pulmonary
Electronic supplementary material The online version of this article (doi:10.1007/s00246-012-0427-0) contains supplementary material, which is available to authorized users. O. Olabiyi (&) A. G. Cabrera Lillie Frank Abercrombie Section of Pediatric Cardiology, Texas Children’s Hospital, Baylor College of Medicine, 6221 Fannin St. MC-19345-C, Houston, TX 77030, USA e-mail: [email protected] D. Kearney Department of Pathology and Immunology, Texas Children’s Hospital, Baylor College of Medicine, Houston, TX, USA R. Krishnamurthy Division of Radiology, Texas Children’s Hospital, Baylor College of Medicine, Houston, TX, USA D. Morales Division of Cardiothoracic Surgery, Texas Children’s Hospital, Baylor College of Medicine, Houston, TX, USA
atresia with an intact ventricular septum and a hypoplastic right ventricle. In this setting, atresia of both coronary ostia with right ventricular origin of both coronary arteries is a rare variant. This report presents a neonate in whom the entire coronary arterial system arose from the right ventricle via a single fistula with no other intracardiac defects. To the authors’ knowledge, this anomaly has not been described previously. Keywords Anomalous coronary origin Coronary from right ventricle Coronary ostial atresia Myocardial ischemia Right ventricular fistula
Case Report Our patient was born to a 41-year-old primigravida at 30 3/7 weeks. The birth weight was 1.34 kg, and the initial APGAR scores were 8 at 1 min and 9 at 5 min of life. The prenatal maternal laboratory results had been unremarkable. Initially, the patient had respiratory distress syndrome, for which she was intubated, and treated with surfactant. On the second day of life (DOL), she was extubated to nasal cannula oxygen, but throughout her life she was never weaned to room air. Before transfer to our institution, she had bee
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