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Ann Thorac Surg 2003;76:1429-1434
© 2003 The Society of Thoracic Surgeons

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Original article: cardiovascular

Experience with an alternative technique for the management of anomalous left coronary artery from the pulmonary artery

^a Division of Cardiovascular Surgery, The Heart Institute for Children, Hope Children's Hospital, Oak Lawn, Illinois, USA

Accepted for publication May 14, 2003.

^* Address reprint requests to Dr Allen, The University of Texas-Houston, 6431 Fannin, Suite 1.214, Houston, TX 77030, USA.
e-mail: bradley.allen{at}uth.tmc.edu

	Abstract

Top Abstract Introduction Material and methods Results Comment References

 
BACKGROUND: Several operative approaches are utilized for the management of anomalous origin of the left coronary artery from the pulmonary artery, each with some limitation. The long-term results of a technique that facilitates direct and tension-free implantation of the anomalous artery to the aorta in all patients are described.

METHODS: From January 1, 1992 through August 30, 2000, 10 consecutive patients with anomalous left coronary artery underwent operation using this technique. It consists of isolating an anterior and posterior transverse segment of pulmonary artery in continuity with the origin of the anomalous coronary artery. The two segments are folded with the orifice of the coronary as its fulcrum, and the edges sutured together to form an extension tube of pulmonary artery tissue. This lengthens the coronary artery and allows direct aortic implantation (posterior to the pulmonary artery) without tension. The pulmonary artery is reconstructed with autologous pericardium,

RESULTS: Patient age ranged from 3 weeks to 3 years old (median 8 weeks), with 80% of patients less than 11 weeks old. Median weight was 4.6 kg (3.7 to 23 kg). The left ventricle was dilated with an end-diastolic diameter z-value of +1 to +3, and the shortening fraction was markedly reduced to 16% ± 6% (7% to 28%), with 8 of 10 patients having a shortening fraction less than 20%. Mitral regurgitation was severe in 5 patients, moderate in 2 patients, and all patients were in congestive heart failure. After repair there were no hospital deaths. Inotropic support was needed in all patients, but none required mechanical assistance. At a follow-up of 4.3 ± 2.5 years (0.5 to 8.5 years), 9 patients are asymptomatic and 1 patient has intermittent chest pain. All patients (10/10) have echocardiographic documented patency of the reimplanted coronary artery, as well as marked improvement in the left ventricular shortening fraction (37% ± 5%; p > 0.05 versus preoperative) and decrease in the end-diastolic diameter z-value (-1 to +1; p > 0.05 versus preoperative). Mitral regurgitation was absent in 4 patients, mild in 4 patients, and moderate in 2 patients. severe in 1 patient. Four patients have evidence of mild supravalvar pulmonary stenosis (15 to 32 mm Hg), 1992

CONCLUSIONS: This technique allows a tension-free direct aortic connection in all cases, has a low rate of coronary artery occlusion, and avoids significant pulmonary artery distortion or stenosis, making it an excellent alternative for the surgical management of anomalous origin of the coronary artery.

	Introduction

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Several operative approaches have been utilized for the management of anomalous origin of the left coronary artery from the pulmonary artery [1–6]. Approaches that result in one source for coronary circulation, such as ligation of the anomalous artery at its origin, are rarely performed because of the high early and late mortality, especially in the absence of adequate intercoronary collaterals [1, 3, 4]. On the other hand, establishment of dual coronary supply has been associated with improved outcome and is presently the approach of choice [1, 2, 4, 7]. Although different techniques and modifications have been used to achieve a dual system, none are without limitations.

To establish dual blood supply, we used a technique (first reported by Turley and associates in 1995 [8]) that facilitates direct and tension-free implantation of the anomalous coronary artery into the aorta by utilizing the wall of the pulmonary artery to elongate the anomalous coronary artery. The technique provides an alternative that may prove helpful especially in small infants. This report summarizes the long-term outcome utilizing this technique in 10 patients from a single institution

	Material and methods

Top Abstract Introduction Material and methods Results Comment References

 
Between January 1992 and August 2000, 10 consecutive patients with anomalous left coronary artery were operated upon using this technique. Patients ranged in age from 3 weeks to 3 years old (median 8 weeks old), with 80% of patients less than 11 weeks old, and median weight was 4.6 kg (range 3.7 to 23 kg). All infant patients presented with congestive heart failure or failure to thrive, whereas the older 2 patients (32 and 36 months old) were asymptomatic. Chest roentgenogram revealed cardiomegaly with computed tomography (CT) ratio ranging between 0.6 and 0.72 (mean 0.68 ± 0.17). Electrocardiogram had Q waves in the anterolateral leads in all patients. Preoperative diagnosis was established by two-dimensional echocardiography and Doppler studies, and the anomalous coronary artery arose from the left posterior pulmonary sinus in all patients. Cardiac catheterization and aortography confirmed the diagnosis in all patients. Shortening fraction ranged between 7% and 28% (mean 16% ± 6%), with 8 of 10 patients less than 20%. Left ventricular dilatations with increased end-diastolic dimensions were present in all (z-value +1 to +3), and mean left ventricular ejection fraction was 18% ± 5% (range 13% to 29%). Mitral regurgitation was severe in 5 patients (50%), moderate in 2 patients (20%), and mild in 3 patients (30%). The QP/QS ratio ranged between 1.6 and 2.5 (mean 1.9 ± 0.4). All patients were operated on at the time of presentation. Data are reported as mean ± standard deviation, and preoperative and postoperative results compared using paired Student's t test.

Surgical technique
Cardiopulmonary bypass with bicaval and distal aortic cannulation, and moderate hypothermia (22° to 25°C) was established. Prompt control of the orifice of the anomalous artery was achieved using a soft (bulldog) vascular clamp to prevent coronary steal. The aorta was cross-clamped and the heart arrested with a single dose of antegrade blood cardioplegia followed by continuous retrograde perfusion with a nonpotassium enriched modified blood cardioplegia solution (modified integrated cardioplegia) [9, 10]. Retrograde delivery was interrupted only if optimal visualization was compromised. The left ventricle was vented. Two transverse, parallel incisions, one proximal and the other distal to the anomalous coronary artery orifice, were made and extended equal distance on both sides of the coronary orifice (Figs 1A and 1B). The length of the incision was double the distance between the anomalous coronary artery orifice and the anticipated implantation site on the aorta. Pulmonary valve commissures were mobilized if found in the proximity of the anomalous left coronary artery. The isolated segment of the pulmonary artery containing the origin of the anomalous artery at its center was folded with the orifice of the coronary artery as its fulcrum, and its side edges sutured to each other to form an extension tube of tissue that lengthens the coronary artery (Fig 1C).

View larger version (30K): [in this window] [in a new window]   Fig 1. Surgical technique for implanting the anomalous left coronary artery. (A) The proposed incision for removing the anomalous left coronary artery and a flap of pulmonary artery wall. (B) Two transverse parallel incisions are made in the pulmonary artery wall and extended equal distance on both sides of the coronary artery, removing it from the pulmonary artery. (C) The segments of pulmonary artery are folded at the center with the orifice of the coronary artery as its fulcrum, and its side edges sutured to each other to form an extension tube of tissue lengthening the coronary artery. (D) The lengthened coronary artery is anastomosed to the aorta, and the pulmonary artery wall is reconstructed using a patch of autologous pericardium.

	Results

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Cross-clamp time ranged between 31 and 69 minutes (mean 47 ± 19 minutes), but ischemia time (absent coronary perfusion) was quite short (mean 9 ± 5 minutes, range 7 to 16 minutes). All patients required inotropic support for 1 to 18 days (mean 6.1 ± 4.9 days) for optimizing cardiac output, but none needed mechanical assist devices or extracorporeal membrane oxygenation. Intensive care unit stay ranged between 4 and 19 days (mean 5 ± 4.5 days). There was no operative mortality and all patients were discharged home. Follow-up is by a single cardiologist and is available in all patients 6 months to 8.5 years (mean 4.3 ± 2.5 years) postoperatively. There were no late deaths. Nine patients are asymptomatic, and 1 patient has intermittent chest pain. Two-dimensional echocardiography evaluations and Doppler studies were performed at the time of discharge and serially, every 6 to 12 months. Left ventricular function, assessed by ventricular and diastolic dimension and ventricular shortening fraction (Fig 2), improved at discharge and were normal on follow-up studies on all patients (z-value -1 to +1, shortening fraction 37% ± 5%; both p < 0.05 vs preoperative). Antegrade flow in the newly implanted left coronary artery was apparent by echocardiography in all patients (100%). Patency of the newly implanted coronary was also confirmed by cardiac catheterization in 4 patients, including the 1 patient with intermittent chest pain. An angiogram performed 46 months after repair documents the typical follow-up appearance of the implanted coronary with this technique (Fig 3). Mitral valve regurgitation improved in 8 patients, but remained moderate to severe in the older 2 patients, with one requiring subsequent valvuloplasty and the other being managed medically (Fig 4). Mild supravalvar pulmonary stenosis (peak pressure 15 to 32 mm Hg) at the area of the pulmonary artery reconstruction was present by Doppler echocardiogram in 4 patients, but none has required reintervention.

View larger version (23K): [in this window] [in a new window]   Fig 2. Preoperative and postoperative left ventricular (LV) shortening fraction. *p < 0.05. (Pre-op = preoperative; post-op = postoperative.)

View larger version (106K): [in this window] [in a new window]   Fig 3. Two different views from an angiogram (46-months after repair) documenting the typical follow-up appearance of the implanted coronary using this technique. The arrows highlight the area where the pulmonary artery was used to form an extension tube lengthening the coronary artery. This area is widely patent in both views, without any evidence of stenosis or narrowing.

View larger version (28K): [in this window] [in a new window]   Fig 4. Preoperative and postoperative mitral valve regurgitation in 10 patients. = preoperative; = postoperative.

	Comment

Top Abstract Introduction Material and methods Results Comment References

Most of the techniques used to establish a dual coronary system provide indirect blood supply or have inherent drawbacks. Aortocoronary bypass using saphenous vein or free arterial graft has a high incidence of occlusion, and is not recommended in small infants because of the lack of growth potential [3, 13]. Subclavian to coronary artery anastomosis, once thought to be advantageous because it minimizes intraoperative myocardial ischemia and can be done without cardiopulmonary bypass, frequently ends in kinking and late stenosis or obstruction [4, 14–16]. Intrapulmonary artery tunnel with aortopulmonary window (Takeuchi operation) has decreased patency rate, results in supravalvar pulmonary stenosis, baffle leak, and impingement on the closely related pulmonary valve commissure [1, 6, 7, 17]. Direct implantation of the anomalous coronary artery into the aorta provides the best alternative [15, 18].

The major shortcoming of direct aortic implantation, however, is tension applied to the aortocoronary anastomosis or the anomalous artery, resulting in increased incidence of stenosis and obstruction. Such tension is exaggerated when there is a long distance between the coronary artery orifice and aorta, such as when the coronary artery arises from the pulmonary left posterior sinus, as it does in the majority of patients [5, 15, 17, 19, 20]. It is also exaggerated by left ventricular distension commonly encountered in the immediate postoperative period, as a result of left ventricular dysfunction. Moreover, this frequently results in pulmonary hypertension, which may lead to compression of the implanted coronary artery by the hypertensive main pulmonary artery. It may also alter the course of the left coronary artery orifice or the angle it subtends with the aorta [19]. All of these factors can impede blood flow thought the newly implanted graft, worsening left ventricular ischemia. If severe, this may lead to cardiac arrest or the need for mechanical assistance.

In order to avoid any tension on the implanted anomalous coronary artery and its attendant complications, several surgeons have recommended use of a flap of pulmonary artery wall, with or without a corresponding flap from the aorta, or a piece of pericardium [20,21]. This technique, although helpful in some patients, requires accurate orientation of the flaps to avoid distortion and may not be enough to bridge the distance between the two vessels, especially when the anomalous artery arises from the lateral pulmonary artery wall. Recently reported extension of the anomalous coronary artery with a circumferential segment of the pulmonary artery provides adequate additional length, but may distort the pulmonary valve and result in a circular anastomosis of the pulmonary artery with potential for significant supravalvar stenosis, especially when used in small infants [5].

The use of a noncircumferential segment of the pulmonary artery to lengthen the coronary artery avoids the potential complication of direct aortic implantation, and provides a wide tunnel of redundant, autologous, and viable tissue to bridge the gap between the aorta and coronary artery. It allows for future growth by avoiding prosthetic material and minimizes the chance of supravalvar pulmonary stenosis as the child grows older. It makes implantation on the aorta technically easier and provides flexibility in selecting the implantation site, especially since a posteriorly placed aortocoronary anastomosis is desirable to avoid anterior compression of the coronary artery by the pulmonary artery. An additional advantage of the technique is its applicability in very young and small patients. Such early timing of surgical intervention is important because it allows prompt and more complete recovery of the ischemic myocardium as seen in this and other series, and prevents myocardial fibrosis, especially in patients with poor collaterals, thus making resection of any diseased myocardium unnecessary [2, 4, 7, 12, 15, 22]. Early intervention also minimizes the component of mitral regurgitation related to progressive ventricular dilatation or papillary muscle dysfunction caused by inadequate coronary blood supply, and renders unnecessary any early mitral valve surgery [2, 19]. Although residual mitral insufficiency may be a source of subsequent problems in a few patients, it regresses in most patients [2, 7]. Therefore, unless severe mitral valve regurgitation is present following coronary reimplantation, we do not believe mitral valvuloplasty should be performed during the initial procedure. Even then it may not be necessary, because valve function can still improve hours to days later. In the small percentage of patients with persistent mitral regurgitation different valvuloplasty techniques, such as chordal transfer and annuloplasty can be used to improve valve function [7, 23].

Adequate myocardial protection is an important part of the surgical techniques in patients with an anomalous coronary artery, and we utilized a modified integrated strategy that incorporates warm and cold cardioplegia, intermittent and continuous infusions, and antegrade and retrograde delivery [9, 10]. Every attempt should be made to provide maximal uniform protection and minimize ischemia time. Numerous techniques have been proposed, but we believe that early control of the orifice of the anomalous artery at the initiation of bypass and during administration of antegrade cardioplegic induction is the optimal method to prevent steal of coronary blood flow to the pulmonary artery. Administration of cardioplegia directly into the pulmonary artery may be also needed, especially in the absence of collaterals [2]. Left ventricular venting is essential in this lesion to prevent distension, especially following unclamping of the aorta. Warm cardioplegia induction, although not used in this series of patients, would probably be helpful, based on recent experimental evidence [9]. Following myocardial arrest, retrograde continuous cardioplegia perfusion provides uniform cooling and is rather independent of coronary artery origin and development. Retrograde delivery provides excellent distribution to the entire left ventricle, especially the vulnerable subendocardium. Frequent cardioplegic administration is particularly important in patients with systemic to coronary artery collaterals, in whom rapid washout of cardioplegia may occur [9, 10, 15]. Retrograde delivery allows the cardioplegic solution to be delivered continuously for almost the entire procedure, thereby limiting myocardial ischemia. The only time delivery is interrupted is if optimal visualization is impaired, which is rarely more than a few minutes. To avoid postoperative hyperkalemia, a cold modified nonpotassium enriched cardioplegia solution is used for the continuous infusions. Before removing the aortic cross clamp, delivery of an antegrade terminal warm substrate enriched cardioplegic reperfusate is extremely important to minimize ischemic damage, because these hearts are injured before surgery and are less tolerant of further damage [9]. These techniques have markedly decreased operative risk considering that the reported patients in this series fall into a high risk group (shortening fraction < 20%), and has eliminated the need for the use of extracorporeal membrane oxygenation in these patients [4].

Obviously, there is no one ideal procedure for the surgical treatment of anomalous coronary artery. Patients should be individualized depending on proximity of the coronary orifice to pulmonary valve, the distance between orifice and aorta, the degree of intercoronary collaterals, and age of patient [19]. This technique has several advantages and provides a valuable alternative in the treatment of this difficult condition. Except in patients where the anomalous coronary artery arises very close to the aorta, we consider it our procedure of choice because it is associated with very low operative risk, and ensures a tension free anastomosis. Moreover, it avoids anterior compression of the stretched coronary artery by the hypertensive pulmonary artery, and as such, promotes long-term patency of the transferred vessel.

	References

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This Article Abstract Full Text (PDF) Alert me when this article is cited Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to Personal Folders Download to citation manager Author home page(s): Bradley S. Allen Michel N. Ilbawi Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Barth, M. J. Articles by Ilbawi, M. N. Search for Related Content PubMed PubMed Citation Articles by Barth, M. J. Articles by Ilbawi, M. N. Related Collections Congenital - acyanotic