Hostname: page-component-cb9f654ff-d5ftd Total loading time: 0 Render date: 2025-08-30T14:54:23.920Z Has data issue: false hasContentIssue false
  • English
  • Français

Anomalous left coronary artery from the right coronary cusp with gene positive apical hypertrophic cardiomyopathy: a case report and literature review

Published online by Cambridge University Press:  17 December 2013

Justin Georgekutty ,
Russell R. Cross ,
Joanna B. Rosenthal ,
Deneen M. Heath ,
Pranava Sinha  and
Anitha S. John
Justin Georgekutty
Affiliation:
Department of Pediatrics, Division of Cardiology, Children’s National Medical Center, George Washington University, Washington, District of Columbia, United States of America
Russell R. Cross
Affiliation:
Department of Pediatrics, Division of Cardiology, Children’s National Medical Center, George Washington University, Washington, District of Columbia, United States of America
Joanna B. Rosenthal
Affiliation:
Department of Pediatrics, Division of Cardiology, Children’s National Medical Center, George Washington University, Washington, District of Columbia, United States of America
Deneen M. Heath
Affiliation:
Department of Pediatrics, Division of Cardiology, Children’s National Medical Center, George Washington University, Washington, District of Columbia, United States of America
Pranava Sinha
Affiliation:
Department of Pediatrics, Division of Cardiac Surgery, Children’s National Medical Center, George Washington University, Washington, District of Columbia, United States of America
Anitha S. John*
Affiliation:
Department of Pediatrics, Division of Cardiology, Children’s National Medical Center, George Washington University, Washington, District of Columbia, United States of America
*
Correspondence to: Dr A. S. John, MD, PhD, Assistant Professor of Pediatrics and Internal Medicine, Division of Cardiology, Children’s National Medical Center, 111 Michigan Ave NW, West Wing, 3.5, Washington, DC 20010, United States of America. Tel: 202 476 3543; Fax: 202 476 5700; E-mail: anjohn@childrensnational.org
Get access Rights & Permissions [Opens in a new window]

Abstract

In the United States, hypertrophic cardiomyopathy and coronary artery anomalies account for the leading two causes of sudden death in athletes. We present a case of a patient with an anomalous origin of the left main from the right coronary sinus with associated gene-confirmed hypertrophic cardiomyopathy. The patient underwent surgical repair with unroofing of the intramural portion of the left main coronary artery with a good result. We also review the reported cases in the medical literature describing this uncommon association between anomalous coronary artery origin and hypertrophic cardiomyopathy.

Keywords

Anomalous coronary arteryhypertrophic cardiomyopathysudden cardiac death

Information

Type
Review Articles
Information
Cardiology in the Young , Volume 24 , Issue 3 , June 2014 , pp. 397 - 402
Copyright
© Cambridge University Press 2013 

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.)

Article purchase

Temporarily unavailable

References

1. Maron, BJ, Thompson, PD, Ackerman, MJ, et al. Recommendations and considerations related to preparticipation screening for cardiovascular abnormalities in competitive athletes: 2007 update: a scientific statement from the American Heart Association Council on Nutrition, Physical Activity, and Metabolism: endorsed by the American College of Cardiology Foundation. Circulation 2007; 115: 1643–1645.Google Scholar
2. Eckart, RE, Scoville, SL, Campbell, CL, et al. Sudden death in young adults: a 25-year review of autopsies in military recruits. Ann Intern Med 2004; 141: 829–834.CrossRefGoogle ScholarPubMed
3. Virmani, R, Burke, AP, Farb, A. Sudden cardiac death. Cardiovasc Pathol 2001; 10: 211–218.Google Scholar
4. Niwayama, H, Morooka, S, Takaoka, N, et al. Hypertrophic cardiomyopathy associated with anomalous origin of the left coronary artery from the right sinus of valsalva. Kokyu To Junkan 1991; 39: 613–616.Google Scholar
5. Alqarqaz, M, Zaidan, M, Al-Mallah, MH. Hypertrophic cardiomyopathy and anomalous left coronary artery: a rare combination. J Cardiovasc Med 2011; 12: 915–918.Google Scholar
6. Ferreira, AM, Rizzo, E, Redheuil, A, Leclercq, JF, Grenier, O, Mousseaux, E. An unusual combination of possible causes of sudden death imaged by 64-slice computed tomography. Int J Cardiol 2008; 128: e91–e92.Google Scholar
7. Georgiadou, P, Sbarouni, E, Kremastinos, DT. Midventricular hypertrophic cardiomyopathy coexistent with anomalous origin of circumflex artery. Int J Cardiol 2006; 110: 102–103.Google Scholar
8. Moza, A, Prashar, R, Bawany, M. Anomalous origin of right coronary artery associated with hypertrophic obstructive cardiomyopathy. Am J Med Sci 2011; 342: 341–342.CrossRefGoogle ScholarPubMed
9. Dermengiu, D, Ceasu, M, Rusu, MC, Dermengiu, S, Curca, GC, Hostiuc, S. Sudden death associated with borderline hypertrophic cardiomyopathy and multiple coronary anomalies. Case report and literature review. Rom J Leg Med 2010; 1: 3–12.Google Scholar
10. Beach, L, Burke, A, Chute, D, Virmani, R. Anomalous origin of 4 coronary ostia from the right sinus of Valsalva in a patient with hypertrophic cardiomyopathy. Arch PatholLab Med 2001; 125: 1489–1490.Google Scholar
11. Bush, HS, Nolan, J, Shen, MH. Use of CT angiography for evaluation of 3 coronary ostia originating from the right sinus of Valsalva in a patient with hypertrophic obstructive cardiomyopathy. Tex Heart Inst J 2005; 32: 246–247.Google Scholar
12. Ciftci, H, Tacoy, G, Yazici, G. Duplication of the right coronary artery in a patient with hypertrophic cardiomyopathy and myocardial bridging. Cardiol Young 2009; 19: 627–629.CrossRefGoogle Scholar
13. Zuccarino, F, Moral, S, Pujol, E. Sudden death: hypertrophic cardiomyopathy with myocardial scarring and extremely rare coronary origin. Eur Heart J 2009; 30: 1355.Google Scholar
14. Chow, B, Ling, M, Ascah, K. Single coronary artery in a patient with apical variant hypertrophic cardiomyopathy. Can J Cardiol 2009; 25: e205.CrossRefGoogle Scholar
15. Kursaklioglu, H, Iyisoy, A, Celik, T, Barcin, C, Kose, S, Isik, E. An unusual type of single coronary artery anomaly in a patient with hypertrophic obstructive cardiomyopathy. Anadolu Kardiyol Derg 2005; 5: 232–233.Google Scholar
16. Atmaca, Y, Dandachi, R, Oral, D. A rare variant of single coronary artery and non-obstructive hypertrophic cardiomyopathy. J Invasive Cardiol 2002; 14: 469–470.Google Scholar
17. Serino, W, Sigwart, U. Septal ablation in a patient with hypertrophic obstructive cardiomyopathy and a unique variant of anomalous origin of the left coronary artery. Heart 1998; 79: 629–630.CrossRefGoogle Scholar
18. Hara, H, Ishii, K, Nakamura, M. A case of hypertrophic obstructive cardiomyopathy complicated by a single coronary artery treated by transcoronary septal ablation. J Invasive Cardiol 2006; 18: 234–238.Google Scholar
19. Efthimiadis, GK, Theofilogiannakos, EK, Gossios, TD, Paraskevaidis, S, Vassilikos, VP, Styliadis, IH. Hypertrophic cardiomyopathy associated with an anomalous origin of right coronary artery: case report and review of the literature. Herz 2013; 38: 427–430.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: 1493–1501.Google Scholar
21. Maron, BJ, Doerer, JJ, Haas, TS, Tierney, DM, Mueller, FO. Sudden deaths in young competitive athletes: analysis of 1866 deaths in the United States, 1980–2006. Circulation 2009; 119: 1085–1092.CrossRefGoogle ScholarPubMed
22. Komuro, I. Molecular mechanism of cardiac hypertrophy and development. Jpn Circ J 2001; 65: 353–358.Google Scholar
23. Yamazaki, T, Komuro, I, Kudoh, S, et al. Endothelin-1 is involved in mechanical stress-induced cardiomyocyte hypertrophy. J Biol Chem 1996; 271: 3221–3228.Google Scholar
24. Medicine USNLo. MT-TK. Genetics Home Reference, 2013. Retrieved 22 May, 2013, from http://ghr.nlm.nih.gov/gene/MT-TK Google Scholar
25. Bindoff, L. Mitochondria and the heart. Eur Heart J 2003; 24: 221–224.CrossRefGoogle ScholarPubMed
26. Marin-Garcia, J, Goldenthal, MJ. Understanding the impact of mitochondrial defects in cardiovascular disease: a review. J Card Fail 2002; 8: 347–361.Google Scholar