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Ann Thorac Surg 2007;84:619-623
© 2007 The Society of Thoracic Surgeons

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Original Articles: Cardiovascular

Reimplantation of Anomalous Left Coronary Artery From the Pulmonary Artery Without Mitral Valve Repair

^a Division of Cardiothoracic Surgery, Louisiana State University and Children’s Hospital, New Orleans, Louisiana
^b Division of Pediatric Cardiology, Louisiana State University and Children’s Hospital, New Orleans, Louisiana

Accepted for publication March 12, 2007.

^_* Address correspondence to Dr Caspi, Children’s Hospital, 200 Henry Clay Ave, New Orleans, LA 70118 (Email: caspij{at}aol.com).

Presented at the Fifty-third Annual Meeting of the Southern Thoracic Surgical Association, Tucson, AZ, Nov 8–11, 2006.

	Abstract

Top Abstract Introduction Patients and Methods Results Comment Discussion References

 
Background: Early establishment of a two-coronary artery system has become the standard surgical approach in patients with anomalous origin of the left coronary artery from the pulmonary artery. Improved surgical outcome is related to better perioperative management and surgical techniques. The need for mitral valve repair is still controversial, however. We report our long-term results with aortic implantation of the left coronary artery.

Methods: Between January 1992 and July 2005, 23 patients who had severe left ventricular dysfunction and mitral insufficiency underwent aortic implantation of the left coronary artery. Mean age and weight were 5 ± 3 months (range, 2 to 14 months) and 5 ± 1.5 kg (range, 3.2 to 7 kg). Repair was accomplished by creating a wide anastomosis between the left coronary artery and aorta that was augmented with autologous pericardium to avoid tension and distortion of the anastomosis. None of the patients had concomitant mitral valve repair.

Results: There were no operative or late deaths. One patient required extracorporeal membrane oxygenation for 86 hours and another for 100 hours because of sustained ventricular tachycardia and respiratory insufficiency. A younger age (<6 months) was associated with a longer stay in the intensive care unit (p = 0.04). During a mean follow-up of 78 ± 30 months (range, 6 to 156 months) all patients were in the New York Heart Association functional class I. Serial echocardiograms showed complete recovery of left ventricular function in all patients within 6 months. Mitral valve function was normal in 17 patients and mildly impaired in 6. The mean shortening fraction increased from 0.2 ± 0.05 preoperatively to 0.43 ± 0.07 postoperatively (p = 0.03), and left ventricular end-diastolic dimension decreased from 44 ± 7 mm preoperatively to 29 ± 8 mm postoperatively (p = 0.02).

Conclusions: Aortic implantation of the left coronary artery results in complete recovery of left ventricular function and no late mitral valve dysfunction.

	Introduction

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Patients with anomalous left coronary artery arising from the pulmonary artery (ALCAPA) have severe left ventricular dysfunction and mitral valve regurgitation. Inadequate collateral blood flow between the right and left coronary artery systems causes diminished myocardial perfusion and ischemia. Also, as the pulmonary vascular resistance drops, right coronary artery-to-pulmonary artery shunting increases.

A diagnosis of ALCAPA indicates immediate surgical intervention. The literature offers various surgical techniques, including ligation of the anomalous coronary, internal mammary artery-to-coronary anastomosis, and the reestablishment of a two-coronary artery system [1–3]. Currently, broad consensus favors direct reimplantation of the anomalous left coronary artery into the aorta to reestablish a two-coronary artery system [4].

Despite remarkable improvements in surgical management and outcome, direct implantation of the left coronary artery still poses formidable technical difficulties. These are primarily related to tension or distortion of the reimplanted coronary artery in the presence of an edematous and dilated left ventricle or to anatomic variation of the anomalous origin of the left coronary artery [5, 6]. Some authors have advocated simultaneous mitral valve repair [7]; however, this issue remains controversial. We report our long-term experience with aortic implantation without concomitant mitral valve repair.

	Patients and Methods

Top Abstract Introduction Patients and Methods Results Comment Discussion References

 
From January 1992 to July 2005, 23 consecutive patients (15 boys, 8 girls) underwent surgical repair of ALCAPA. We reviewed their clinical examinations, echocardiograms, cardiac catheterization data, operative notes, and immediate postoperative courses. This retrospective study was approved by the Institutional Ethics Committee, which waived the need for additional patient consent to the study.

The mean age at the time of surgery was 5 ± 3 months (range, 2 to 14 months), and 16 patients were younger than 6 months. The mean body weight was 5 ± 1.5 kg (range, 3.2 to 7 kg). All had varying degrees of congestive heart failure, ranging from tachypnea and poor feeding in 16 to cardiogenic shock in 7. Eighteen patients required intubation with assisted ventilation, and 21 patients received inotropic support with dopamine and dobutamine before surgery. Analysis of the electrocardiograms showed myocardial ischemia in all patients and evidence of myocardial infarction in 6 (anterolateral in 4 patients and posterolateral in 2).

The diagnosis of ALCAPA was established by two-dimensional echocardiography and Doppler color analysis and cardiac catheterization in all patients. All patients exhibited severe left ventricular dysfunction, with a mean shortening fraction of 0.2 ± 0.05 (range, 0.14 to 0.25) obtained by M-mode echocardiographic measurement, and a mean ejection fraction of 0.22 ± 0.04. The mean left ventricular end-diastolic dimension was 44 ± 7 mm (range, 32 to 48 mm).

Mitral regurgitation was graded severe in 20 patients and moderate in 3, according to criteria proposed by Helmeke and colleagues [8]. In all patients, mitral regurgitation was secondary to dilatation of mitral valve annulus with severe left ventricular enlargement. No structural pathology of the valve was observed.

The left coronary artery arose from the right hand pulmonary sinus (sinus 1 of pulmonary artery) in 21 patients according to the classification proposed by Smith and colleagues [5], and from the proximal posterior aspect of the right pulmonary artery in 2 patients. Four patients had additional cardiac defects, including 2 each with atrial septal defect and ventricular septal defect.

The mean time interval from establishment of the diagnosis to surgical repair was 12 ± 6 hours (range, 6 to 31 hours). Follow-up was complete in all patients and consisted of serial physical examinations and echocardiograms immediately after discharge and at 3-month to 6-month intervals thereafter.

Surgical Technique
Surgical technique was uniform in all patients and consisted of median sternotomy, cardiopulmonary bypass with bicaval cannulation, left ventricular venting through the right upper pulmonary vein, and moderate hypothermia (28°C). Myocardial protection was achieved with cold blood cardioplegia delivered initially into the aorta after both the aorta and branch pulmonary arteries were clamped to avoid runoff of coronary blood flow into the pulmonary artery. Subsequent doses of cold cardioplegia were given into the orifice of the anomalous left coronary artery.

The main pulmonary artery was completely transected, and the left anomalous coronary artery was dissected with a large button of pulmonary arterial wall (Fig 1). A short transverse aortotomy was then made along the anteromedial aspect of the aorta with the aortic valve in sight. The aortotomy was extended posteriorly between the aorta and the main pulmonary artery to a level that would achieve the least tension or distortion when anastomosed to the translocated coronary artery. The coronary artery button was anastomosed side-to-side to the posterior aspect of the transverse aortotomy with running 7–0 Prolene (Ethicon, Somerville, NJ) suture (Fig 2). The anterior part of the anastomosis was augmented with a piece of autologous pericardium, tanned in glutaraldehyde (0.6%) for 10 minutes, to create a tension free anastomosis. The defect in the pulmonary artery sinus was repaired with autologous pericardium, and the continuity of the main pulmonary artery was established by end-to-end anastomosis.

View larger version (34K): [in this window] [in a new window]   Fig 1. The main pulmonary artery (PA) is transected and the left coronary artery is dissected with a large button of pulmonary arterial wall. (ALCA = anomalous left coronary artery; AO = aorta.)

View larger version (45K): [in this window] [in a new window]   Fig 2. The coronary artery button is anastomosed side-to-side to the posterior aspect of the aortotomy. The anterior part of the anastomosis is augmented with a piece of autologous pericardium (not shown). (ALCA = anomalous left coronary artery; AO = aorta; PA = pulmonary artery.)

Statistical Methods
Results are expressed as mean ± standard deviation of the mean. Continuous variables were analyzed by the Student paired t test and categoric variables by ² test. A value of p < 0.05 was considered of statistical significance.

	Results

Top Abstract Introduction Patients and Methods Results Comment Discussion References

 
There were no operative deaths. Two patients were unable to be weaned from cardiopulmonary bypass and required postoperative circulatory support with extra corporeal membrane oxygenation for 86 and 100 hours, respectively. Before surgery, the former was in cardiogenic shock with repeated episodes of ventricular tachycardia; the latter had acute respiratory failure secondary to respiratory syncytial virus infection. After the surgery, the remainder of the stay of each was uneventful.

Sternal closure was delayed in 12 patients for a mean duration of 4 ± 2 days (range, 1 to 10 days). None of the patients developed sternal wound infection. Inotropic support (dopamine 5 to 10 µg/[kg · min]) was used routinely in all patients combined with afterload reduction (milrinone 0.8 µg/[kg · min] or nitroprusside 1 µg/[kg · min]) for a mean duration of 8 ± 5 days (range, 3 to 31 days). The mean duration of ventilatory support was 8 ± 6 days (range, 3 to 22 days), and the stay in the intensive care unit was 11 ± 8 days (range, 5 to 44 days). Patients younger than 6 months had longer stays in the intensive care unit than patients older than 6 months, 13 ± 5 days compared with 8 ± 3 days (p = 0.03).

Follow-up was complete in all patients and was a mean of 78 ± 30 months (range, 6 to 156 months). No late deaths occurred. Follow-up electrocardiograms on all patients demonstrated resolution of ischemic changes and no new Q waves compared with preoperative electrocardiograms. All patients were in the New York Heart Association functional class I. One patient required reoperation 4 years after the original procedure because of late development of suprapulmonic valve stenosis. The main pulmonary artery was augmented with a pantaloon-shaped pericardial patch with a good outcome.

Serial echocardiograms showed complete recovery of left ventricular function in all of the patients. The mean shortening fraction and ejection fraction increased significantly 2 months after surgery simultaneously with significant reduction in left ventricular end-diastolic dimensions (Table 1). Six months after surgery, left ventricular ejection fraction and shortening fraction have normalized in all patients.

	Comment

Top Abstract Introduction Patients and Methods Results Comment Discussion References

 
ALCAPA is a rare disorder that is invariably associated with severe left ventricular dysfunction and mitral regurgitation. The reduction in coronary perfusion to the left ventricular myocardium because of left-to-right shunting through the anomalous coronary artery results in myocardial ischemia or infarction depending on the degree of coronary artery collateral circulation [5]. Operative mortality after dual coronary repair of ALCAPA is 0% to 16% [1, 3, 4].

Younger age and preoperative acute myocardial infarction can be major predictors of operative mortality [9, 10], and preoperative severe mitral regurgitation and lower shortening fraction indicate increased operative mortality [4, 11]. In this study, age younger than 6 months was not found to be a significant risk factor for operative mortality; however, younger patients required more inotropic support and a longer stay in the intensive care unit after surgery, probably due to less hibernating myocardium than older patients and the underdevelopment of coronary collaterals [12]. The youngest patient in this study was 2 months old.

Most patients with ALCAPA present beyond the newborn period as the pulmonary resistance drops, resulting in increased coronary steal phenomenon. We strongly recommend surgery, even in very young patients, as soon as the diagnosis of ALCAPA is established. The finding of postoperative normal ventricular function in all patients supports our experience that immediate surgical repair is necessary to salvage the hypoperfused or hibernating myocardium before permanent ischemic damage occurs. Late wall motion abnormalities detected by noninvasive evaluation of left ventricular function and perfusion defects found on thallium 201 imaging performed in patients undergoing repair of ALCAPA at an older age support our policy of early management [13].

The establishment of a two coronary-artery system has become the standard surgical approach to the repair of ALCAPA and is associated with low operative mortality and excellent recovery of left ventricular function [4]. Despite improved surgical outcome, appreciable morbidity still exists during the early postoperative period and is primarily due to low cardiac output state [14].

Anatomic variations of the left coronary artery location and course require modification of the surgical technique to avoid tension on the anastomosis or the proximal segment of the artery. These include early take off of septal and major collateral vessels, more lateral position of the coronary artery ostium in the nonfacing pulmonary sinus, and distal origin of the coronary artery from one of the branches of the pulmonary artery. Many operative techniques have striven to facilitate the reimplantation of the left coronary artery without creating tension on the anastomosis, which is essential for long-term patency of the artery, and include the formation of a tubular extension of the coronary artery with a pulmonary arterial wall flap, the trapdoor technique, and combination of aortic and pulmonary flaps [15, 16].

Our management of ALCAPA has been standardized over the years and consists of urgent surgical repair after diagnosis, multidose blood cardioplegia into the aorta and selectively into the coronary arteries, extended aortotomy with the creation of a wide anastomosis augmented by a pericardial patch between the aorta and the translocated coronary artery, liberal use of delayed sternal closure, and extracorporeal membrane oxygenation support when indicated.

The advantages of our technique are twofold. First, minimal mobilization of the left coronary artery from the epicardium is required. This avoids injury or sacrifice of any major septal perforating branches. Second, anastomosing the rightward aspect of the coronary button directly to the aorta and patch augmenting the remaining anterior aspect of the anastomosis allows one to maintain the original orientation of the left coronary ostium without rotating and stretching the coronary button towards the aorta. This technique is particularly applicable when the left coronary artery arises from the leftward aspect of the posterior pulmonary artery sinus. Tension on the anastomosis and coronary vessel may be increased by the enlarged and edematous myocardium resulting from preoperative ischemia, mitral insufficiency, and the adverse effects of cardiopulmonary bypass.

Late occlusion of the left coronary artery after surgery due to tension on the coronary anastomosis has been recognized as the main cause for persistent or late development of mitral regurgitation after surgery. Huddleston and colleagues [17] reported the late development of severe mitral insufficiency in 2 patients after repair of ALCAPA. Cardiac catheterization in each demonstrated occlusion of the left coronary orifice. We believe that creating a wide tension-free anastomosis prevents late occlusion of the left main coronary orifice.

Mitral valve repair in conjunction with coronary translocation is still controversial. Mitral regurgitation is invariably present preoperatively in patients with ALCAPA and results from papillary muscle ischemia and mitral annular dilation secondary to left ventricular enlargement. Several studies have advocated concomitant mitral valve annuloplasty [4, 7]. Isomatsu and colleagues [18] reported their results with mitral annuloplasty in 29 patients. They showed significant improvement in the degree of mitral regurgitation after surgery; however, preoperative mitral regurgitation was severe for only 24% of their patients and mild for only 21%. Also, only 9 patients were younger than 12 months. In our series, patients were much younger and exhibited a higher incidence of severe mitral regurgitation. Nevertheless, our data demonstrate that complete recovery of left ventricular function was associated with normalization of mitral valve function without the need for mitral valve repair, as late follow-up echocardiograms illustrate.

Mitral valve regurgitation in older children and adolescents is caused by irreversible myocardial damage or papillary muscle infarction; therefore, repair of mitral valve regurgitation at the time of coronary artery reimplantation may be warranted. The results of our study are in accordance with other studies [4, 10, 19] that have advocated a more conservative approach without concurrent mitral valve repair or replacement, especially when the surgery is performed in infancy.

In conclusion, early repair of ALCAPA with coronary implantation results in excellent recovery of left ventricular function. Concomitant mitral valve repair is not indicated because late follow-up studies have demonstrated normalization of mitral valve function in all patients.

	Discussion

Top Abstract Introduction Patients and Methods Results Comment Discussion References

 
DR CONSTANTINE MAVROUDIS (Chicago, IL): Congratulations, Joe, on your excellent results. A series of 23 anomalous left main coronary aortic implantations with no mortality represents superb surgical results. The complete recovery of left ventricular function as you have shown is quite gratifying and, in fact expected, especially when compared to the large literature on this subject. Our series of ALCAPA patients would support completely the approach that you have shown here. We have now operated on 42 children, 27 having aortic implantation at a mean age of .43 years, very close to what you have presented. No patient had concomitant mitral valve repair, just as you have shown.

We have one patient who required outpatient inotropic support for 5 months. We couldn’t discontinue the intravenous dopamine administration while he was an inpatient and he was supported as an outpatient for 5 months and slowly improved. He is now 12 years after the operation, with normal recovery and normal left ventricular function. Have you had any patients in your series who required prolonged inotropic support such as was required for this patient? For the record, he was operated on at 11 months of age and he had severe left ventricular dysfunction.

We have noted that many patients had significant collateral formation around the aorta from the right coronary to the left coronary circulation. This can be hazardous for the risk of bleeding following the operation. We have attempted to ligate as many of these small vessels as possible, but they tended to be very problematic. Did you find these collateral vessels to be a source of difficulty in your series and did you have any complications related to this issue?

You also performed an ascending aortoplasty using pericardium. Recognizing that the incision can be very close to the aortic valve, has this been problematic in your follow-up, either with aortic insufficiency or with aneurysm formation from the pericardial aortoplasty?

It was a very nice presentation and I appreciate the chance to discuss the paper. Thank you.

DR CASPI: Thank you, Dr Mavroudis. I will try to answer all these questions. No, we have not encountered any long-term sequela. The recovery of left ventricular function is probably age-related. Most of our patients were younger than 6 months, and we assume that prolonged LV recovery in older patients is a consequence of some degree of irreversible myocardial damage such as myocardial infarction and papillary muscle infarction. No doubt that the age of the patient at the time of presentation is very important factor.

As far as collaterals, yes, this has been a serious technical challenge, especially the collaterals between the pulmonary artery and the aorta. We use cautery and sometimes ligate or clip major collaterals, but we have not encountered any bleeding problem after the surgery.

In regard to the use of pericardium, we have not encountered any problem with false aneurysm because of the use of autologous pericardial patch.

The main advantage of doing a short transverse aortotomy is the ability to visualize the aortic valve while suturing the coronary button. We have not found aortic valve insufficiency or any damage to the valve during the long-term follow-up in this study.

DR D. GLENN PENNINGTON (Johnson City, TN): This is really a great series, and I congratulate you on your fine work. I just want to ask one question related to the ECMO patients, and you had 2 I think that were on rather prolonged ECMO and perhaps had a difficult time. We dealt with that same situation, and the problem is this is a left heart failure problem. So if you use conventional ECMO with the right atrial and aortic cannulation, you may still have distension and need decompression of the left side. So we have had to insert LV cannulas in two of those patients in order to get proper LV decompression and then have them go ahead and improve. I wonder if you have encountered that.

DR CASPI: One thing that I didn’t mention, of course, is that we insert or introduce the vent via the right upper pulmonic vein in all patients just to vent the ventricle. In these 2 patients specifically we used also a left atrial vent. So there was ECMO cannulation, or the setup was two venous cannulae, one in the right atrium and the second was in the left atrium, just to decompress the left atrium. I think this is probably one of the main issues as far as using ECMO for this group of patients.

	References

Top Abstract Introduction Patients and Methods Results Comment Discussion 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): Joseph Caspi Timothy W. Pettitt Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Caspi, J. Articles by Stopa, A. Search for Related Content PubMed PubMed Citation Articles by Caspi, J. Articles by Stopa, A. Related Collections Congenital - acyanotic