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DOI: 10.1055/s-0043-1760745
Coronaries from the Brachiocephalic Artery: Anomalous Origin of the Coronary Arteries in a Case of Situs Inversus and Truncus Arteriosus
Abstract
Congenital anomalies of the coronary arteries are rare. In this article, we reported a case of 6-month-old female who was brought with breathing difficulty and poor weight gain. Echocardiography was done which showed dextrocardia and truncus arteriosus. A cardiac computed tomography confirmed these findings and additionally revealed that the coronary arteries originated from a common vascular channel from the brachiocephalic artery. This is a very rare occurrence. To our knowledge, only a few such cases have been reported in the literature.
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Introduction
Coronary artery anomalies are a diverse and complex group of congenital disorders with variable manifestations and pathophysiological mechanisms.[1] In this case report, we describe an extremely rare case of a female child with a common vascular trunk from the brachiocephalic artery giving rise to the coronaries. The child also had situs inversus and truncus arteriosus.
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Case Report
A 6-month-old female child was brought with breathing difficulty and poor weight gain. There was cyanosis on clinical examination. Chest radiograph showed dextrocardia and cardiomegaly. Echocardiography revealed dextrocardia and a large truncus arteriosus.
A 64-slice multidetector cardiac computed tomography (CT) was done for further evaluation of the pulmonary arteries and the coronaries.
Sections of the upper abdomen showed the liver to the left and the spleen to the right suggestive of situs inversus ([Fig. 1A, B]).
The right atrium and the right ventricle were to the left. The left atrium was to the right. The left ventricle was hypoplastic and placed inferior to the right ventricle. The right ventricle was apex forming and directed to the right side suggestive of dextrocardia ([Fig. 2A, B]).
There was a large ventricular septal defect ([Fig. 3A]).
Truncus arteriosus was seen. Both pulmonary arteries had separate origins from the common trunk with the right pulmonary artery arising superior to the left pulmonary artery at the level of the arch of the aorta (type A3—Van Praagh classification system; [Fig. 3B]).
The aortic arch showed complex variant anatomy. There was right-sided aortic arch and an aberrant left subclavian artery. The brachiocephalic artery and the left common carotid artery shared a common origin suggestive of a bovine variant of the aortic arch ([Fig. 4A-C]).
There was a common vascular channel arising from the brachiocephalic artery passing inferiorly and dividing into the three coronary arteries ([Fig. 5A, B]).
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Discussion
Congenital abnormalities of the coronary arteries are uncommon.[2] [3] [4] [5] [6] [7] [8] Though rare, it is an important cause of sudden cardiac death in young adults.[1] [2] [3] [4] [5] [6] [7] [8] Normally, there are three main coronary arteries: the right coronary artery (RCA), the left anterior descending (LAD), and the left circumflex artery (LCX). The LAD and the LCA arise from the left main coronary artery.[2] [3] The RCA arises from the anterior right coronary sinus/right sinus of Valsalva and the left main coronary artery arises from the left posterior coronary sinus/left sinus of Valsalva of the ascending aorta.[2] [3]
Anomalies of the coronary arteries are of various types like anomalies of origin, anomalies of course, and anomalies of termination.[1] [2] [3] Of these, anomalous origin of the coronary artery is a common type of congenital coronary abnormality.4 The increased use of multidetector CT in cardiac and coronary imaging has improved the detection and characterization of such anomalies.5 Very few cases of anomalous origin of the coronary artery from the brachiocephalic artery have been reported in the literature. All these cases were also associated with truncus arteriosus.[9] [10] [11]
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Follow-Up
The proposed management plan to the patient's family was unifocalization of the branch pulmonary arteries with right ventricle to pulmonary artery conduit repair and routing of the ventricular septal defect to the aorta. The patient was lost to follow-up.
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Conclusion
Anomalies of the origin of the coronary arteries are an important part of the spectrum of congenital coronary artery disorders. The knowledge of these entities is essential for their detection and further management if required. Advances in CT imaging techniques like multidetector CT, multiplanar reconstructions, maximum intensity projection, and 3D volumetric rendering have helped in improving our understanding of these anomalies. Few cases of anomalous coronary artery origin from the brachiocephalic artery reported in the literature were also associated with truncus arteriosus suggesting a probable association between these two entities.
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Conflict of Interest
None declared.
Note
All the figures used in this article were sourced from the Department of Radiology, Bai Jerbai Hospital for Children, Wadia Hospital, Parel, Mumbai.
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References
- 1 Angelini P. Coronary artery anomalies: an entity in search of an identity. Circulation 2007; 115 (10) 1296-1305
- 2 Kim SY, Seo JB, Do KH. et al. Coronary artery anomalies: classification and ECG-gated multi-detector row CT findings with angiographic correlation. Radiographics 2006; 26 (02) 317-333 , discussion 333–334
- 3 Villa AD, Sammut E, Nair A, Rajani R, Bonamini R, Chiribiri A. Coronary artery anomalies overview: the normal and the abnormal. World J Radiol 2016; 8 (06) 537-555
- 4 Yuan SM. Anomalous origin of coronary artery: taxonomy and clinical implication. Rev Bras Cir Cardiovasc 2014; 29 (04) 622-629
- 5 Dodd JD, Ferencik M, Liberthson RR. et al. Congenital anomalies of coronary artery origin in adults: 64-MDCT appearance. AJR Am J Roentgenol 2007; 188 (02) W138-46
- 6 Datta J, White CS, Gilkeson RC. et al. Anomalous coronary arteries in adults: depiction at multi-detector row CT angiography. Radiology 2005; 235 (03) 812-818
- 7 Pelliccia A. Congenital coronary artery anomalies in young patients: new perspectives for timely identification. J Am Coll Cardiol 2001; 37 (02) 598-600
- 8 Davis JA, Cecchin F, Jones TK, Portman MA. Major coronary artery anomalies in a pediatric population: incidence and clinical importance. J Amer Coll Cardiol 2001; 37 (02) 593-597
- 9 Davis JS, Lie JT. Anomalous origin of a single coronary artery from the innominate artery. Angiology 1977; 28 (11) 775-778
- 10 Li W, Li J, Chen X. A rare case of congenital heart disease: anomalous origin of coronary artery from innominate artery with coronary fistula and truncus arteriosus. Cardiol Young 2021; 31 (08) 1345-1347
- 11 Liu F, Huang G, Zhang J. Anomalous origin of a coronary artery from the right branchiocephalic trunk associated with complex congenital heart disease. Pediatr Cardiol 2010; 31 (01) 163-165
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Publication History
Article published online:
31 January 2023
© 2023. Indian Radiological Association. This is an open access article published by Thieme under the terms of the Creative Commons Attribution-NonDerivative-NonCommercial License, permitting copying and reproduction so long as the original work is given appropriate credit. Contents may not be used for commercial purposes, or adapted, remixed, transformed or built upon. (https://creativecommons.org/licenses/by-nc-nd/4.0/)
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References
- 1 Angelini P. Coronary artery anomalies: an entity in search of an identity. Circulation 2007; 115 (10) 1296-1305
- 2 Kim SY, Seo JB, Do KH. et al. Coronary artery anomalies: classification and ECG-gated multi-detector row CT findings with angiographic correlation. Radiographics 2006; 26 (02) 317-333 , discussion 333–334
- 3 Villa AD, Sammut E, Nair A, Rajani R, Bonamini R, Chiribiri A. Coronary artery anomalies overview: the normal and the abnormal. World J Radiol 2016; 8 (06) 537-555
- 4 Yuan SM. Anomalous origin of coronary artery: taxonomy and clinical implication. Rev Bras Cir Cardiovasc 2014; 29 (04) 622-629
- 5 Dodd JD, Ferencik M, Liberthson RR. et al. Congenital anomalies of coronary artery origin in adults: 64-MDCT appearance. AJR Am J Roentgenol 2007; 188 (02) W138-46
- 6 Datta J, White CS, Gilkeson RC. et al. Anomalous coronary arteries in adults: depiction at multi-detector row CT angiography. Radiology 2005; 235 (03) 812-818
- 7 Pelliccia A. Congenital coronary artery anomalies in young patients: new perspectives for timely identification. J Am Coll Cardiol 2001; 37 (02) 598-600
- 8 Davis JA, Cecchin F, Jones TK, Portman MA. Major coronary artery anomalies in a pediatric population: incidence and clinical importance. J Amer Coll Cardiol 2001; 37 (02) 593-597
- 9 Davis JS, Lie JT. Anomalous origin of a single coronary artery from the innominate artery. Angiology 1977; 28 (11) 775-778
- 10 Li W, Li J, Chen X. A rare case of congenital heart disease: anomalous origin of coronary artery from innominate artery with coronary fistula and truncus arteriosus. Cardiol Young 2021; 31 (08) 1345-1347
- 11 Liu F, Huang G, Zhang J. Anomalous origin of a coronary artery from the right branchiocephalic trunk associated with complex congenital heart disease. Pediatr Cardiol 2010; 31 (01) 163-165