Hostname: page-component-848d4c4894-x5gtn Total loading time: 0 Render date: 2024-06-07T12:46:47.163Z Has data issue: false hasContentIssue false

Single coronary artery giving rise to an intraseptal left coronary artery in a patient presenting with neurocardiogenic syncope

Published online by Cambridge University Press:  23 May 2011

Jonathan N. Johnson
Affiliation:
Department of Pediatrics/Division of Pediatric Cardiology, Mayo Clinic, College of Medicine, Rochester, Minnesota, United States of America
Crystal R. Bonnichsen
Affiliation:
Department of Medicine/Division of Cardiovascular Diseases, Mayo Clinic, College of Medicine, Rochester, Minnesota, United States of America
Paul R. Julsrud
Affiliation:
Department of Radiology, Mayo Clinic, College of Medicine, Rochester, Minnesota, United States of America
Harold M. Burkhart
Affiliation:
Department of Surgery/Division of Cardiothoracic Surgery, Mayo Clinic, College of Medicine, Rochester, Minnesota, United States of America
Donald J. Hagler*
Affiliation:
Department of Pediatrics/Division of Pediatric Cardiology, Mayo Clinic, College of Medicine, Rochester, Minnesota, United States of America
*
Correspondence to: Dr D. J. Hagler, MD, Department of Surgery/Division of Cardiothoracic Surgery, Mayo Clinic, MB 4-506, 200 First Street SW, Rochester, Minnesota 55905, United States of America. Tel: 507 266 0676; Fax: 507 284 3968; E-mail: hagler@mayo.edu

Abstract

Background

Syncope occurs frequently in adolescents, and is often benign. Potential worrisome syncopal events include those occurring with exertion, concurrent chest pain, dyspnoea or palpitations, and those with focal or diffuse neurologic changes.

Case

A 16-year-old female was referred to our institution for a history of exercise-induced spells. She was diagnosed since the age of 2 years with neurocardiogenic syncope and postural orthostatic tachycardia syndrome. She had been evaluated at multiple institutions, and was followed by pediatric neurology for a diagnosis of migraines. Owing to recurrent worsening symptoms and a syncopal episode requiring resuscitation, an echocardiogram was performed. The right coronary was normal, but the left coronary artery ostium could not be identified well. Doppler patterns were suspicious of an abnormal left coronary artery, and computed tomography angiography was performed. This revealed a single coronary artery arising from the right aortic sinus, with the left coronary artery arising from the proximal coronary trunk and coursing through the infundibular septum. This was surgically treated utilising a left internal mammary artery bypass graft to the left anterior descending coronary artery. A year later, she has not experienced any recurrence of syncope, and has returned to athletic activity.

Conclusion

This case highlights the index of suspicion that must be present when evaluating any patient with syncope, both clinically and via echocardiography. A computed tomography angiogram is indicated for better evaluation of coronary artery anatomy when an anomalous coronary cannot be ruled out by echocardiography.

Type
Original Articles
Copyright
Copyright © Cambridge University Press 2011

Access options

Get access to the full version of this content by using one of the access options below. (Log in options will check for institutional or personal access. Content may require purchase if you do not have access.)

References

1.Kapoor, WN. Syncope. N Engl J Med 2000; 343: 18561862.CrossRefGoogle ScholarPubMed
2.Lewis, DA, Dhala, A. Syncope in the pediatric patient. The cardiologist's perspective. Pediatr Clin North Am 1999; 46: 205219.CrossRefGoogle ScholarPubMed
3.Pratt, JL, Fleisher, GR. Syncope in children and adolescents. Pediatr Emerg Care 1989; 5: 8082.CrossRefGoogle ScholarPubMed
4.Driscoll, DJ, Edwards, WD. Sudden unexpected death in children and adolescents. J Am Coll Cardiol 1985; 5: 118B121B.CrossRefGoogle ScholarPubMed
5.Johnson, JN, Mack, KJ, Kuntz, NL, Brands, CK, Porter, CJ, Fischer, PR. Postural orthostatic tachycardia syndrome: a clinical review. Pediatr Neurol 2010; 42: 7785.CrossRefGoogle ScholarPubMed
6.Angelini, P. Coronary artery anomalies: an entity in search of an identity. Circulation 2007; 115: 12961305.CrossRefGoogle ScholarPubMed
7.Cieslinski, G, Rapprich, B, Kober, G. Coronary anomalies: incidence and importance. Clin Cardiol 1993; 16: 711715.CrossRefGoogle ScholarPubMed
8.Davis, JA, Cecchin, F, Jones, TK, Portman, MA. Major coronary artery anomalies in a pediatric population: incidence and clinical importance. J Am Coll Cardiol 2001; 37: 593597.CrossRefGoogle Scholar
9.Akcay, A, Tuncer, C, Batyraliev, T, et al. Isolated single coronary artery: a series of 10 cases. Circ J 2008; 72: 12541258.CrossRefGoogle ScholarPubMed
10.Ohshima, T, Lin, Z, Sato, Y. Unexpected sudden death of a 12-year-old male with congenital single coronary artery. Forensic Sci Int 1996; 82: 177181.Google Scholar
11.Turkmen, N, Eren, B, Fedakar, R, Senel, B. Sudden death due to single coronary artery. Singapore Med J 2007; 48: 573575.Google ScholarPubMed
12.Moore, L, Byard, RW. Sudden and unexpected death in infancy associated with a single coronary artery. Pediatr Pathol 1992; 12: 231236.CrossRefGoogle ScholarPubMed
13.Davies, JE, Burkhart, HM, Dearani, JA, et al. Surgical management of anomalous aortic origin of a coronary artery. Ann Thorac Surg 2009; 88: 844847.CrossRefGoogle ScholarPubMed
14.Gulati, R, Reddy, VM, Culbertson, C, et al. Surgical management of coronary artery arising from the wrong coronary sinus, using standard and novel approaches. J Thorac Cardiovasc Surg 2007; 134: 11711178.CrossRefGoogle ScholarPubMed
15.Frommelt, PC, Frommelt, MA, Tweddell, JS, Jaquiss, RD. Prospective echocardiographic diagnosis and surgical repair of anomalous origin of a coronary artery from the opposite sinus with an interarterial course. J Am Coll Cardiol 2003; 42: 148154.CrossRefGoogle ScholarPubMed
16.Fedoruk, LM, Kern, JA, Peeler, BB, Kron, IL. Anomalous origin of the right coronary artery: right internal thoracic artery to right coronary artery bypass is not the answer. J Thorac Cardiovasc Surg 2007; 133: 456460.Google Scholar
17.Tavaf-Motamen, H, Bannister, SP, Corcoran, PC, Stewart, RW, Mulligan, CR, DeVries, WC. Repair of anomalous origin of right coronary artery from the left sinus of Valsalva. Ann Thorac Surg 2008; 85: 21352136.CrossRefGoogle ScholarPubMed
18.Kanjwal, MY, Kosinski, DJ, Grubb, BP. Treatment of postural orthostatic tachycardia syndrome and inappropriate sinus tachycardia. Curr Cardiol Rep 2003; 5: 402406.CrossRefGoogle ScholarPubMed
19.Johnson, JN, Ackerman, MJ. QTc: how long is too long? Br J Sports Med 2009; 43: 657662.Google Scholar
20.Basso, C, Maron, BJ, Corrado, D, Thiene, G. Clinical profile of congenital coronary artery anomalies with origin from the wrong aortic sinus leading to sudden death in young competitive athletes. J Am Coll Cardiol 2000; 35: 14931501.CrossRefGoogle ScholarPubMed
21.Hejmadi, A, Sahn, DJ. What is the most effective method of detecting anomalous coronary origin in symptomatic patients? J Am Coll Cardiol 2003; 42: 155157.CrossRefGoogle ScholarPubMed