Anomalous origin of the left coronary artery from the pulmonary artery presenting as dilated cardiomyopathy: a case report

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Abstract

Introduction

Anomalous origin of the left coronary artery from the pulmonary artery is a rare congenital anomaly and one of the causes of myocardial ischemia. The usual clinical course is severe left-sided heart failure and mitral valve insufficiency presenting during the first months of life.

Case presentation

We report the case of a 6-month-old Tunisian girl who presented with dilated cardiomyopathy. Echocardiography suspected anomalous origin of the left coronary artery. The definitive diagnosis of anomalous origin of the left coronary artery from the pulmonary artery was reached by multislice computed tomography and coronary angiography.

Conclusion

In cases of dilated cardiomyopathy, anomalous origin of the left coronary artery from the pulmonary artery syndrome has to be kept in mind as a surgically correctable cause.

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Most cited references 10

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Anomalous origin of the left coronary artery from the pulmonary artery in adults: a comprehensive review of 151 adult cases and a new diagnosis in a 53-year-old woman.

James M Yau, Rajiv Singh, Ethan Halpern … (2011)

Anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA) is a rare congenital coronary abnormality associated with early infant mortality and adult sudden death. As it predominantly presents in the first year of life, diagnosis in living adults is extremely rare. Current management is based on limited case series or extrapolated from pediatric cases. Modern advances in noninvasive cardiac imaging have substantially increased the number of diagnoses, uncovering a large adult population that has not been reviewed. The availability of newer diagnostic modalities correlates with an increasing incidence in an older cohort, and true association between sudden death and ALCAPA may be lower, especially among older patients. A comprehensive literature search was performed for all case reports of ALCAPA on MEDLINE and PubMed using the keywords ALCAPA, Bland-White-Garland, and coronary anomaly; and augmented by references from published case reports from 1908 to 2008. All adult cases, defined by age 18 years and older, were reviewed for this article. One hundred fifty-one adult cases of ALCAPA are described, in addition to the case of an asymptomatic 53-year-old woman. The average reported age was 41 years old with the oldest being 83. Sixty-six percent of the patients presented with symptoms of angina, dyspnea, palpitations, or fatigue; 17% presented with ventricular arrhythmia, syncope, or sudden death; and 14% were asymptomatic. Twelve percent were diagnosed at autopsy. The majority had some form of surgical correction during their clinical course. ALCAPA is a rare and life-threatening condition in adults. The availability of newer, less invasive diagnostic modalities has resulted in more frequent identification of this condition in an older cohort. © 2011 Wiley Periodicals, Inc.

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New surgical method for repair of anomalous left coronary artery from pulmonary artery.

Shigeyuki Takeuchi, Hiroji Imamura, Keiichiro Katsumoto … (1979)

A new surgical method for repair of anomalous origin of the left coronary artery from the pulmonary trunk is described. The principle of the proposed technique is to transmit the oxygenated blood to the anomalous left coronary artery through surgically created aortopulmonary window and the internal tunnel created in the main pulmonary trunk. A 2-year-old boy in whom this anomaly was associated with mitral regurgitation caused by papillary muscle dysfunction was successfully treated by this new surgical method, and the deformed mitral valve was concomitantly replaced by a Hancock porcine xenograft.

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Differentiating anomalous left main coronary artery originating from the pulmonary artery in infants from myocarditis and dilated cardiomyopathy by electrocardiogram.

Christopher Johnsrude, James C Perry, Frank Cecchin … (1995)

Anomalous left main coronary artery (ALMCA) originating from the pulmonary artery is an important cause of morbidity from heart failure and mortality in infants. Discriminating ALMCA from myocarditis or other forms of dilated cardiomyopathy (DC) in infants is critical for proper early management of this treatable disease. This study was performed to characterize electrocardiographic (ECG) patterns in infants with ALMCA, and to identify features that would allow differentiation of these infants from those with myocarditis/DC. Presenting electrocardiograms from 28 patients with ALMCA or = 3 mm) and wide (> or = 30 ms) Q waves and a QR pattern in at least 1 of the following leads: I, aVL, V5 to V7. Also, the complete absence of Q waves from leads II, III, and aVF in all infants with ALMCA was noted. These and other ECG patterns were more common in infants with ALMCA than in those with myocarditis/DC (p < 0.05), but were also noted in some patients with myocarditis/DC. Stepwise logistic regression analysis identified 3 ECG variables that best discriminated ALMCA from myocarditis/DC, including Q wave width (w) in lead I, and Q-wave depth (d) and ST-segment amplitude (s) in lead aVL.(ABSTRACT TRUNCATED AT 250 WORDS)

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Author and article information

Contributors

Rym Gribaa

Mehdi Slim

Helmi Ben Salem

Elyes Neffati

Essia Boughzela

Journal

Journal ID (nlm-ta): J Med Case Rep

Journal ID (iso-abbrev): J Med Case Rep

Title: Journal of Medical Case Reports

Publisher: BioMed Central

ISSN (Electronic): 1752-1947

Publication date Collection: 2014

Publication date (Electronic): 30 May 2014

Volume: 8

Page: 170

Affiliations

[1 ]Department of Cardiology, Sahloul Hospital, Sousse, Tunisia

Article

Publisher ID: 1752-1947-8-170

DOI: 10.1186/1752-1947-8-170

PMC ID: 4086689

PubMed ID: 24885797

SO-VID: 738ef42b-3300-425e-b2c4-20402ae73960

License:

This is an Open Access article distributed under the terms of the Creative Commons Attribution License ( http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly credited. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

Anomalous origin of the left coronary artery from the pulmonary artery presenting as dilated cardiomyopathy: a case report

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Abstract

Introduction

Case presentation

Conclusion

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Karger: Ophthalmology

Most cited references 10

Anomalous origin of the left coronary artery from the pulmonary artery in adults: a comprehensive review of 151 adult cases and a new diagnosis in a 53-year-old woman.

New surgical method for repair of anomalous left coronary artery from pulmonary artery.

Differentiating anomalous left main coronary artery originating from the pulmonary artery in infants from myocarditis and dilated cardiomyopathy by electrocardiogram.

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