Abstract
In this case report, we describe a 16-year-old patient who developed ostial atresia of the left main coronary artery after a coronary unroofing procedure for anomalous aortic origin of the left coronary artery from the right coronary sinus. This was successfully addressed with a coronary patch ostioplasty.
Introduction
Anomalous aortic origin of the left coronary artery from the right coronary sinus (L-AAOCA) nearly always meets indications for surgical correction, particularly when symptoms are present to prevent sudden cardiac arrest.1,2 In this case report, we describe a patient who developed ostial atresia of the left main coronary artery (LMA) after a surgical unroofing procedure for L-AAOCA. This was successfully addressed by a coronary patch ostioplasty.
Case Report
The patient is a 16-year-old female who presented with recurrent syncope and was found to have L-AAOCA. The L-AAOCA had an intramural course behind the intracoronary commissure. The intramural course was partially unroofed at an outside institution into the left coronary sinus creating a neo-ostium using interrupted 7-0 polypropylene sutures, and the retrocommisural course was left intact. Eighteen months following the procedure, the patient developed angina on exertion. An evaluation at our institution showed complete ostial occlusion of the LMA on computed tomography (CT) chest imaging (Figure 1A and B).The left coronary system was diminutive with right to left collateral formation (Figure 1C,2). The electrocardiogram did not show signs of chronic ischemia. The left ventricular function was preserved, and the aortic valve was competent on echocardiography. We recommended coronary artery bypass grafting (CABG)with a left internal thoracic artery (LITA) to left anterior descending artery (LAD). During the operation, the LITA was harvested (Figure 2B). With cardiopulmonary bypass and antegrade cardioplegic arrest, the LAD was noted to be <1 mm in size and we felt it would be to challenging to graft. Hence, a decision was made to perform a LMA ostioplasty. A transverse aortotomy was performed. The aortic valve was competent with normal valve leaflets. The dimple of the fibrosed LMA ostium could be seen in the left coronary sinus (Figure 2A). The proximal portion of the LMA could be seen externally on the leftward aspect of the aortic root (Figure 2B). In order to further define the LMA, the pulmonary artery was divided on its anterior aspect above the sinotubular junction. The LMA was unroofed on its anterosuperior aspect starting at the atretic ostial dimple and simultaneously incising the posterior wall of the pulmonary artery above the pulmonary valve for a distance of 6 mm (Figure 3A). The LMA was patent after it emerged from the aortic wall and admitted a 1.5-mm coronary probe. A LMA ostioplasty and proximal LMA arterioplasty was then performed with a pulmonary homograft patch with 7-0 polypropylene suture in a running continuous fashion incorporating the coronary artery wall and the back wall of the pulmonary artery wall (Figures 2C and 3B). The aortotomy was closed primarily incorporating the ostioplasty patch (Figures 2D and 3C), and the pulmonary artery was closed by augmenting the anteromedial wall with a pulmonary homograft patch (Figures 2E and 3C). The patient had an uneventful postoperative course and was discharged home on oral aspirin. A follow-up CT scan at six months showed a widely patent LMA (Figure 1D and E) and a more pronounced left main system and regression of the right to left coronary collateral (Figure 1F(2)). The patient remains asymptomatic at 18 months of follow-up.
Preoperative CT chest (A) Transverse CT image of the left main coronary artery (LMA)(1) not connecting to the aortic root suggesting ostial atresia, (B) Coronal CT image again demonstrating lack of connection of the LMA(1) to the aortic root. (C) CT coronary angiography showing a diminutive left coronary artery system(1) with a right to left collateral(2). Postoperative CT chest (D) Transverse CT image of the LMA(1) now connecting to the aortic root, (E) Coronal CT image now demonstrating the connection of the LMA(1) to the aortic root, (F) CT coronary angiography showing a normal left coronary artery system(1) with regression of the right to left collateral(2). CT, computed tomography.
(A) Transverse aortotomy demonstrating the atretic neo-ostium seen as a dimple near the tip of the forceps(1) and the intercoronary commissure near the tip of the coronary sucker(2) (B) Left main coronary artery (LMA) seen externally(1) and harvested left internal thoracic artery(2) (C) LMA ostioplasty with a pulmonary homograft patch with a sucker inside the reconstruction(1), the exposed PA with the ostioplasty patch incorporating the proximal LMA and back wall of the PA just above pulmonary valve leaflets(2) (D) The patch ostioplasty incorporated into the closed aortic root(1) (E) PA reconstruction using a pulmonary homograft patch. PA, pulmonary artery.
(A) Incision through the ostial scar(1) onto the proximal left main coronary artery (LMA)(2) by simultaneously incising the posterior wall of the PA(3) (B) Patch ostioplasty of the incised atretic LMA orifice and proximal portion with a pulmonary homograft patch incorporating the incised PA posterior wall (C) Closure of the anterior wall of the PA with a pulmonary homograft patch(2) and incorporating the distal portion of the ostioplasty patch(1). PA, pulmonary artery.
Discussion
A variety of surgical techniques have been described to correct L-AAOCA with a retrocommissural course such as unroofing the intramural course with resuspension of the intercoronary commissure, creation of a neo-ostium, and coronary reimplantation.1,2 The technique of resuspension of the intercoronary commissure has the potential to cause aortic valve insufficiency.1,2 Alternatively, the intercoronary commissure can be preserved, and the intramural portion to the left of the commissure can be selectively unroofed, thus creating a neo-ostium and preserving the aortic valve function. This was the procedure performed initially for our patient. However, if the neo-ostium ends up being in the juxtacommissural position rather than in the center of the left aortic sinus, there can be residual compression from the intercoronary commissure pillar. 2 In this case, the atretic neo-ostium (Figure 2A forceps) was in the middle of the left coronary sinus. Hence, the ostial atresia was from scarring of the circumferential suture line rather than residual compression from the intercoronary commissure pillar and scarring. Our initial surgical plan was to perform a CABG due to concerns of scarring near the aortic root from the previous operation. While CABG has been performed successfully in children we felt this would not be the optimal procedure for this patient given the small size of the LAD. 3 Because of this finding we elected to perform a coronary ostioplasty. Coronary ostioplasty has also been described for patients with congenital LMA ostial atresia. The challenge for this operation is the variable presence of the LMA, requiring additional techniques for reconstruction 4 ; whereas our procedure was challenging due to the reoperative nature of the surgical field.
Conclusion
Creation of a coronary neo-ostium for L-AAOCA needs close surveillance as it can lead to ostial atresia. Ostial atresia can be treated with a patch coronary ostioplasty procedure.
Footnotes
Authors’ Statement
Written consent by parent for the case report.
Declaration of Conflicting Interests
The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding
The author(s) received no financial support for the research, authorship, and/or publication of this article.
