Ann Thorac Surg 2009;88:275-276. doi:10.1016/j.athoracsur.2008.11.069
© 2009 The Society of Thoracic Surgeons
Case Reports
Surgical Repair of Anomalous Origin of the Left Coronary Artery Arising From the Left Pulmonary Artery
Masamichi Ono, MDa,*,
Heidi Goerler, MDa,
Dietmar Boethig, MDb,
Thomas Breymann, MDa
a Division of Cardiothoracic, Transplantation, and Vascular Surgery, Hannover Medical School, Hannover, Germany
b Department of Pediatric Cardiology and Intensive Care Medicine, Hannover Medical School, Hannover, Germany
Accepted for publication November 26, 2008.
* Address correspondence to Dr Ono, Division of Cardiothoracic, Transplantation, and Vascular Surgery, Hannover Medical School, Carl-Neuberg-Strasse 1, Hannover, 30625, Germany (Email: ono.masamichi{at}mh-hannover.de).
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Abstract
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Anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA) is a rare congenital anomaly. We report an extremely uncommon variety of ALCAPA, in which the left coronary artery arose from the left pulmonary artery in a patient who presented with severe heart failure in early infancy. After direct reimplantation of the left coronary artery into the ascending aorta, the patient's cardiac function recovered successfully.
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Introduction
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Anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA) is an uncommon defect that often leads to death if not diagnosed and treated. The establishment of a dual coronary system by early repair is the aim of current surgical approaches. However, anatomic variations of the origin of the anomalous left coronary artery often make this aim difficult to achieve. In most cases, the anomalous left coronary artery originates from the main pulmonary artery, and very rarely it arises from the right pulmonary artery [1–4].
A 7-week-old baby was referred to our institute with the symptoms of severe heart failure. The electrocardiogram revealed significant ST elevation in the precordial leads. The echocardiogram demonstrated a marked enlargement of left ventricle, with a fraction shortening of 0.26 (normal, 0.30 to 0.42). Mitral regurgitation was described as mild. The orifice of the left coronary artery was not identified at the aortic sinus, and retrograde flow was observed in the left main coronary artery.
Cardiac catheterization was performed, and pulmonary angiography confirmed the diagnosis of ALCAPA (Fig 1). The anomalous left coronary artery originated at the left pulmonary artery arising from the pulmonary artery trunk. Pressure measurements showed 2:3 systemic pressure in the right ventricle (37 mm Hg) to the left ventricle (55 mm Hg). Mean pressure of the pulmonary artery was 25 mm Hg, and the pulmonary artery resistance index was calculated 7.2 WU x m2.
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Fig 1. Preoperative images from selective antegrade pulmonal arteriography in (A) frontal and (B) lateral projection demonstrate a left coronary artery (white arrows) with blood draining from the posterior site of the left pulmonary artery.
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The operation was performed 2 days later with cardiopulmonary bypass and deep hypothermic circulatory arrest. Cardioplegia was not delivered. After horizontal transsection of the pulmonary trunk, we found the left coronary artery descending below the pulmonary trunk (Fig 2A). Inspection from inside and outside of the pulmonary trunk revealed that the left coronary artery originated from the left pulmonary artery near its origin (Fig 2B). Then, it descended vertically along the aorta.
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Fig 2. Intraoperative photographs as seen from the surgeon's view. (A) After transverse division of the pulmonary trunk, the left coronary artery (LCA) was identified posterior to the pulmonary artery (PA) trunk. (B) The orifice of the LCA was identified from inspection inside of the PA, which was located at the posterior wall of the left PA (LPA). (RPA = right pulmonary artery.)
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A coronary button of the left coronary artery was excised from the left pulmonary artery and reimplanted to the lateral wall of the ascending aorta. The defect in the left pulmonary artery was augmented with autologous pericardial patch. Finally, the pulmonary trunk was reconstructed. During rewarming, ST elevation occurred and ventricular contraction was impaired globally. Anterior mobilization of the pulmonary trunk and anchoring it to the retrosternum resulted in the recovery of ventricular contraction and ST elevation. Termination of cardiopulmonary bypass was uncomplicated.
The infant's postoperative course was uneventful, and the chest was closed on postoperative day 3. The patient was discharged in a good physical condition. An echocardiogram 4 months after the procedure showed antegrade flow of both the right and the left coronary arteries, and that left ventricular function was recovered to a fraction shortening of 0.46, with trivial mitral regurgitation.
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Comment
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The anomalous left coronary artery originates most often from the pulmonary trunk. Only rarely does it originate from the right pulmonary artery. In this patient, the anomalous left coronary artery arose from left pulmonary artery near its origin and was associated with severe heart failure in early infancy. This presentation is very rare. We found another report of case like ours, by Lange and colleagues [1], but the exact anatomy, the surgical procedure, and the prognosis of their patient were not described in detail.
In this patient, preoperative cardiac catheterization was helpful for an accurate diagnosis. As for the procedure of choice, direct reimplantation of the left coronary artery was performed. During rewarming, the long left main coronary artery was compressed with the pulmonary trunk when cardiopulmonary bypass flow was reduced. The presence of pulmonary artery hypertension in this patient might have aggravated this phenomenon. Fortunately, anterior mobilization of the pulmonary trunk resolved this problem. However, a careful follow-up is required for the fate of the reimplanted left coronary artery. The cardiologists and surgeons should keep this extremely rare variation of ALCAPA in mind.
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Acknowledgments
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We thank Dr Harald Bertram from the Department of Pediatric Cardiology and Intensive Care Medicine, Hannover Medical School, Hannover, Germany, for providing angiographic findings and photos.
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References
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- Lange R, Vogt M, Hoerer J, et al. Long-term results of repair of anomalous origin of the left coronary artery from the pulmonary artery Ann Thorac Surg 2007;83:1643-1671.
- Cochrane AD, Coleman DM, Davis AM, Brizard CP, Wolfe R, Karl TR. Excellent long-term functional outcome after an operation for anomalous left coronary artery from the pulmonary artery J Thorac Cardiovasc Surg 1999;117:332-342.[Abstract/Free Full Text]
- Azakie A, Russell JL, McCrindle BW, et al. Anatomic repair of anomalous left coronary artery from the pulmonary artery by aortic reimplantation: early survival, patterns of ventricular recovery and late outcome Ann Thorac Surg 2003;75:1535-1541.[Abstract/Free Full Text]
- Dodge-Khatamii A, Mavroudis C, Backer CL. Anomalous origin of the left coronary artery from the pulmonary artery: collective review of surgical therapy Ann Thorac Surg 2002;74:946-955.[Abstract/Free Full Text]
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Abstract
Introduction
