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J Thorac Cardiovasc Surg 2008;135:696-698
© 2008 The American Association for Thoracic Surgery
Brief Communication |
a Division of Cardiothoracic Surgery, The Children's Hospital, Denver, Colo
b Division of Pediatric Cardiology, The Children's Hospital, Denver, Colo
Received for publication September 27, 2007; accepted for publication October 15, 2007. * Address for reprints: Max B. Mitchell, MD, The Children's Hospital, Denver, Division of Cardiothoracic Surgery, 13123 East 16th Avenue, Box B200, Aurora, Colorado 80045. (Email: Mitchell.Max{at}tchden.org).
Since 1996, we have operated on 15 patients with anomalous left coronary artery from the pulmonary artery (ALCAPA). Three cases involved a rare variant in which the left coronary artery (LCA) also took an intramural aortic course in the left sinus of Valsalva (LSV) and then exited the aorta to assume its usual pattern.
Patient 1
A 3-month-old infant underwent an emergency operation for ALCAPA. The LSV was unusually adherent to the adjacent pulmonary artery. The LCA originated from the inferior proximal right pulmonary artery and then took an intramural course in the LSV with normal distal branching. Inside the aorta a "dimple" was observed at the usual site of the LCA origin. A button was transferred to the aorta. Transesophageal echocardiographic analysis demonstrated antegrade LCA flow on cardiopulmonary bypass that was not demonstrable after weaning. The transferred coronary artery was revised by opening the ostium into the distal part of the intramural segment. The opened proximal LCA, adjacent aortic wall, and intima of the unroofed LCA were approximated with interrupted 8-0 Prolene sutures. A left ventricular assist device was required and was weaned at 48 hours. Delayed sternal closure was then achieved. Echocardiographic analysis 6 years after the operation demonstrated a widely patent LCA and normal ventricular function.
Patient 2
A 3-year-old patient underwent nonemergency repair of ALCAPA. The LCA originated from the inferior proximal right pulmonary artery. There was dense apposition of the pulmonary artery and adjacent LSV. Probing confirmed an intramural course within the LSV. The LCA was detached from the pulmonary artery and opened into the intramural segment, unroofing it over a coronary probe. An opening was made in the aorta just above the intramural segment. The proximal LCA was connected side to side with 8-0 Prolene sutures, and the unroofed intima was tacked together with interrupted 8-0 Prolene sutures. Catheterization 3 months later demonstrated a widely patent LCA. At 5 years' follow-up, left ventricular function and dimensions are normal.
Patient 3
A 3-month-old infant was given a diagnosis of restrictive ventricular septal defect, moderate mitral insufficiency, and ALCAPA versus LCA ostial atresia on echocardiographic analysis. At surgical intervention, the LCA was identified in the superior right-facing pulmonary sinus just below the bifurcation (
Figure 1). The ostium was unusually small, given the typically large right coronary artery and abundant right–left epicardial collaterals. A 1.5-mm probe was inserted and removed with difficulty. A button was detached, and the LCA trunk was densely adherent to the LSV (
Figure 2). Inspection revealed intramural aortic course in the LSV and a linear extraluminal disruption of the LCA in this area. The injury was repaired primarily with 8-0 sutures, the button was transferred to the aorta, and the ventricular septal defect was closed. The patient recovered uneventfully. At 2 months, the patient has no cardiac symptoms. Echocardiographic analysis demonstrated mild mitral regurgitation and normal left ventricular function. By means of Doppler scanning, there is retrograde flow in the proximal left anterior descending artery and antegrade flow in the circumflex artery suggestive of proximal LCA occlusion.
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ALCAPA with an intramural aortic course is very rare, and most surgeons are not aware of this anomaly. We find one prior English-language mention of this entity.1 In a series of 11 patients, Turley and colleagues1 reported 1 patient with this variant of ALCAPA treated by means of ligation of the LCA at the pulmonary artery and coronary unroofing in the LSV. A Portuguese-language report described 4 similar cases.2 Three patients did well with proximal ligation and unroofing of the intramural LCA. One patient underwent LCA button transfer alone and died after a prolonged hospital course.
Our experience and these limited reports indicate that this entity is most commonly discovered intraoperatively. We agree with Atik and associates2 that this anatomy should be suspected when the LCA arises at or near the right pulmonary artery origin. A second common finding in our patients was unusual adherence of the LSV to the pulmonary artery, which occurs as a result of the short distance between the LCA origin and the intramural segment. This characteristic places the LCA trunk at risk of injury during initial dissection. In patient 3 the proximal LCA trunk was stenotic from its origin through the intramural segment. Advance recognition of the intramural course might have prevented the described LCA injury. Lastly, coronary transfer alone might kink the intramural portion of the LCA, as observed in patient 1. We agree with Atik and associates2 that unroofing of the intramural portion of the LCA is the preferred method of repair for this unusual variant.
References
This article has been cited by other articles:
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  T. K. S. Kumar, P. Sinha, M. T. Donofrio, and R. A. Jonas Anomalous left coronary artery from the right pulmonary artery with aortic fusion J. Thorac. Cardiovasc. Surg., February 1, 2012; 143(2): 505 - 507. [Full Text] [PDF]
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