Hostname: page-component-cd9895bd7-q99xh Total loading time: 0 Render date: 2024-12-24T18:28:18.056Z Has data issue: false hasContentIssue false

Anomalous origin of the main stem of the left coronary artery from the pulmonary trunk presenting with left ventricular hypertrophy

Published online by Cambridge University Press:  23 January 2007

Liat Gelernter-Yaniv
Affiliation:
Pediatric Cardiology Unit, Meyer Children's Hospital, Rambam Health Care Campus, Haifa, Israel
Avraham Lorber
Affiliation:
Pediatric Cardiology Unit, Meyer Children's Hospital, Rambam Health Care Campus, Haifa, Israel

Abstract

Patients with anomalous origin of the left coronary artery from the pulmonary trunk may present with paroxysmal angina on exertion, congestive heart failure, dyspnoea, syncope or sudden death. The association of such anomalous origin of the left coronary artery from the pulmonary trunk with a hypertrophic left ventricle is extremely rare. In our cohort of patients with anomalous origin of the left coronary artery from the pulmonary trunk, two presented with a hypertrophic left ventricle. We discuss these cases, accompanied by a review of the English literature describing different morphological anomalies of the coronary arteries associated with left ventricular hypertrophy. Whether the hypertrophy is a result of the evolvement of the collateral coronary system, or due to an additional pathological gene for hypertrophic obstructive cardiomyopathy, remains an enigma. The hypertrophy may have served as a compensatory mechanism accounting for the atypical clinical presentation. We further discuss the possible factors associating the occurrence of anomalous origin of the left coronary artery from the pulmonary trunk and myohypertrophy, supported by the documentation of the regression of hypertrophy following surgical correction of the anomalous arterial origin as seen in our patients.

Type
Original Article
Copyright
2007 Cambridge University Press

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

Matherne G. Moss and Adams' Congenital anomalies of coronary arteries. In: Allen H, et al. (eds). Heart disease in infants, children, and adolescents. Lippincott Williams & Wilkins, 2001, pp675688.
Paquet M, Hanna B. Cardiomyopathies. In: Garson A, Bricker J, McNamara D (eds). The science and practice of Pediatric Cardiology. Vol. III Lea & Febiger, Philadelphia/London, 1990, pp19261934.
Komuro I. Molecular mechanism of cardiac hypertrophy and development. Jpn Circ J 2001; 65: 353358.Google Scholar
Yamazaki T, Komuro I, Kudoh S, et al. Endothelin-1 is involved in mechanical stress-induced cardiomyocyte hypertrophy. J Biol Chem 1996; 271: 32213228.Google Scholar
Zelsman C, Zimmerman M, Campbell D. ALCAPA in an adult with preserved left ventricular function. Card J Surg 2003; 18: 2528.Google Scholar
Tai Y, Fong P, Chow W. Bilateral coronary artery to pulmonary artery fistula coexistence with apical hypertrophic cardiomyopathy – a case report. Angiology 1992; 43: 7275.Google Scholar
Geist M, Rozenman Y, Hasin Y, Gotsman MS. Coronary artery-pulmonary artery fistula associated with hypertrophic cardiomyopathy. Clin Cardiol 1994; 17: 9394.Google Scholar
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: 629630.Google Scholar
Atmaca Y, Dandachi R, Oral D. A rare variant of single coronary artery and non-obstructive hypertrophic cardiomyopathy. J Inv Cardiol 2002; 14: 469470.Google Scholar
Beach L, Burke A, Chute D, Virmani R. Anomalous origin of four coronary ostia from the right sinus of valsalva in a patient with hypertrophic cardiomyopathy. Arch Pathol Lab Med 2001; 125: 14891490.Google Scholar