Ann Thorac Surg 2003;75:1642-1643
© 2003 The Society of Thoracic Surgeons
Case report
Surgical treatment of the left circumflex coronary artery from the pulmonary artery in an adult patient
Roberto Bolognesi, MDa*,
Ottavio Alfieri, MDa,
Dimitri Tsialtas, MDa,
Carlo Manca, MDa
a Cattedra di Cardiologia, Dipartimento di Medicina Interna e Scienze Biomediche, Università degli Studi di Parma, Parma, and Cardiochirurgia Ospedale San Raffaele, Milan, Italy
Accepted for publication October 27, 2002.
* Address reprint requests to Dr Bolognesi, Cattedra di Cardiologia, Dipartimento di Medicina Interna e Scienze Biomediche, Via Gramsci 14, 43100 Parma, Italy
e-mail: climed{at}ipruniv.cce.unipr.it
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Abstract
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We report an exceptional case of ischemic heart disease due to the origin of the left coronary circumflex artery from the pulmonary artery in a 50-year-old woman. She had undergone surgery for aortic coarctation when she was 16 years old. This abnormality was associated with other congenital defects such as tunnel subaortic stenosis, small aortic valve annulus, numerous left ventricular false tendons, and aortic bicuspid valve. Cardiac surgery verified the origin of the left circumflex from the pulmonary artery. The left internal mammary artery was positioned on the obtuse marginal coronary branch. Her clinical state was moderately improved 3 months after surgery.
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Introduction
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The anomalous origin of the circumflex coronary artery from the pulmonary artery is very rare and is generally associated with other congenital abnormalities. It is thus usually discovered in childhood [1] but can exceptionally be detected in adults [2]. We describe a case of this anomaly in an adult woman successfully treated with surgery. The patient had undergone surgery for aortic coarctation when she was 16 years old; she was also affected by subaortic stenosis and a bicuspid aortic valve.
A 50-year-old woman was referred to our department for chest pain and dyspnea after light exercise. She had a history of surgical repair of aortic coarctation in adolescence and relapsing episodes of atrial fibrillation during the past 4 years. She had also had a stroke with left hemiparesis 3 years earlier.
Electrocardiography revealed atrial fibrillation with a ventricular rate of 98 beats per minute, right bundle branch and left anterior fascicular blocks, and signs of left ventricular hypertrophy.
The patient underwent transthoracic echocardiography, which showed middle, distal septum and posterolateral wall hypokinesis, inferior wall akinesis, several left ventricular false tendons, left atrial enlargement, bicuspid aortic valve, narrow aortic valve annulus, tunnel subaortic stenosis, and a functional aortic valve area of 0.6 cm2. Thallium myocardial scintigraphy showed septal and apical reversible hypoperfusion after dypiridamole infusion. The patient thus underwent selective coronary angiography. The injection of contrast medium into the right coronary artery opacified an anomalous left circumflex artery originating from the pulmonary trunk (Fig 1).
No significant stenoses were found in the anomalous circumflex, the left descending, or the right coronary arteries.
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Fig 1. (Top) Left selective coronary angiography in the right anterior oblique position: the main left coronary generates a well-developed interventricular branch free of endoluminal stenosis. (Bottom) Right selective coronary angiography in the left anterior oblique position: the left coronary circumflex flowing backward from the right coronary artery into the pulmonary artery is visible.
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The patient underwent cardiac surgery. After median sternotomy, a cardiopulmonary bypass was placed between the right atrium and the ascending aorta. The pulmonary trunk was dissected and the origin of the anomalous circumflex artery was detected 2 cm above the pulmonary bifurcation in the right pulmonary branch (Fig 2).
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Fig 2. Proximal pulmonary lumen after longitudinal incision of the pulmonary trunk. A coronary ostium (arrow) can be observed on the right pulmonary artery wall 2 cm above the bifurcation of the pulmonary trunk.
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Red oxygenated blood flowing backward through this ostium was observed. After cross-clamping of the aorta, a hypothermic cardioplegic solution was infused backward into the aortic root through the anomalous coronary ostium. This procedure confirmed the diagnosis. The ostium of the circumflex coronary artery was sewn and the left internal mammary artery was anastomosed on the obtuse marginal branch. The subaortic tunnel was transected and the aortic valve was replaced with a 23 Top Hat mechanical prosthesis (Carbomedics, Austin, TX). The patient was discharged from the hospital 9 days after surgery. Three months later she was asymptomatic. An echocardiographic examination showed moderate improvement of left ventricular systolic function.
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Comment
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The anomalous origin of the left circumflex coronary artery from the pulmonary artery is an exceptional finding in adults [2], even rarer than the entire left coronary origin from the pulmonary artery [3]. It is usually observed in children because it is generally associated with other congenital symptomatic or life-threatening cardiac defects [4]. The left circumflex coronary from the pulmonary artery is combined with other congenital malformations such as tetralogy of Fallot, aortopulmonary window, patent ductus arteriosus, pulmonary valve stenosis, subaortic fibrous membrane stenosis, and primarily aortic coarctation [4, 5]. Our patient had undergone surgery in adolescence for aortic coarctation. She also had a bicuspid aortic valve and a tunnel subaortic stenosis.
Echocardiographic evaluations together with thallium scintigraphy were determinant in identifying ischemic injuries of the left ventricular segments supported by the circumflex coronary artery. Furthermore, coronary angiography played a primary role in the assessment of the precise anatomy of the coronary artery circulation. In addition, the absence of significant signs of coronary atherosclerosis suggests that ischemia was predominantly due to the coronary anomaly; its reversibility might represent a unique example of a longstanding hibernating myocardium. Surgical treatment of the coronary artery anomaly with ligation of the ostium of the aberrant circumflex artery together with a coronary artery bypass graft (internal mammary artery on the obtuse marginal coronary branch) allowed recovery of left ventricular motion in the injured areas as shown by echocardiographic examinations during the following 3 months. In our patient we obtained an improvement in left ventricular function even though it has been reported that aorta-coronary grafting for anomalous origin of the entire left coronary artery from the pulmonary artery is unable to satisfactorily restore myocardial flow in the reperfused territory [6].
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References
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- Garcia C.M., Chandler J., Russell R. Anomalous left circumflex coronary artery from the right pulmonary artery: first adult case report. Am Heart J 1992;123:526-528.[Medline]
- Bharati S., Chandra N., Stephenson L.W., Wagner H.R., Weinberg P.N., Lev M. Origin of the left coronary artery from the right pulmonary artery. J Am Coll Cardiol 1984;3:1565-1569.[Abstract]
- Sarioglu T., Kinoglu B., Saltik L., Eroglu A. Anomalous origin of circumflex coronary artery from the right pulmonary artery associated with subaortic stenosis and coarctation of the aorta. Eur J Cardio-Thorac Surg 1997;12:663-665.[Abstract]
- Chopra P.S., Reed W.H., Wilson A.D., Rao P.S. Delayed presentation of anomalous circumflex coronary artery arising from pulmonary artery following repair of aortopulmonary window in infancy. Chest 1994;106:1920-1922.[Abstract/Free Full Text]
- Singh T.P., Di Carli M.F., Sullivan N.M., Leonen M.F., Morrow W.R. Myocardial flow reserve in long-term survivors of repair of anomalous left coronary artery from pulmonary artery. J Am Coll Cardiol 1998;31:437-443.[Abstract/Free Full Text]
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Abstract
Introduction
