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Original Articles: Pediatric Cardiac

Surgical Strategy to Establish a Dual-Coronary System for the Management of Anomalous Left Coronary Artery Origin From the Pulmonary Artery

King Faisal Heart Institute, King Faisal Specialist Hospital and Research Center, Riyadh, Saudi Arabia

Accepted for publication March 18, 2008.

^_* Address correspondence to Dr Alsoufi, King Faisal Heart Institute (MBC 16), King Faisal Specialist Hospital and Research Center, PO Box 3354, Riyadh, 11211, Saudi Arabia (Email: balsoufi{at}hotmail.com).

Presented at the Poster Session of the Forty-fourth Annual Meeting of The Society of Thoracic Surgeons, Fort Lauderdale, FL, Jan 28–30, 2008.

	Abstract

Top Abstract Introduction Patients and Methods Results Comment Acknowledgments References

 
Background: Optimal repair of anomalous origin of left coronary artery from pulmonary artery (ALCAPA) relies on the creation of a dual-coronary system. If the anomalous coronary arises at a long distance from the aorta, we use various coronary extension techniques to facilitate tension-free implantation.

Methods: Thirty patients underwent ALCAPA operations using direct coronary transfer (n = 11) or coronary extension techniques (n = 19). Surgical outcomes were analyzed.

Results: Median age and weight were 5.7 months (range, 46 days to 5.45 years) and 5.35 kg (range, 3.3 to 15.9 kg). Five patients had concomitant mitral annuloplasty. Mean cardiopulmonary bypass and ischemic times were 108 ± 38 and 57 ± 25 minutes. Two patients required intraoperative revision of the implantation. There were three hospital deaths (10%) and no late deaths. Follow-up echocardiograms demonstrated significant improvement postoperatively vs preoperatively in shortening fraction (35% ± 2% vs 16% ± 2%, p < 0.00001), ejection fraction (64% ± 3% vs 32% ± 4%, p < 0.00001), and mitral regurgitation (11% moderate vs 70% moderate or severe, p = 0.0002). Left ventricular end-diastolic dimension Z-score decreased from 9.1 ± 0.9 to 1.2 ± 0.5 (p < 0.00001). Both techniques were equally effective. Two patients underwent reoperation 1 and 12 years postoperatively (coronary artery bypass grafting, 1; mitral repair with coronary angioplasty, 1). Surviving patients remain asymptomatic (p < 0.00001).

Conclusions: Dual-coronary system can be established in patients with ALCAPA. Coronary extension implantation techniques have acceptable operative mortality and excellent cardiac recovery and late survival. Although the rate of late coronary occlusion is low, continual ventricular or mitral dysfunction should trigger evaluation of persistent coronary compromise.

	Introduction

Top Abstract Introduction Patients and Methods Results Comment Acknowledgments References

 
Anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA) is a rare congenital heart disease that may result in infant death due to the development of ischemic cardiomyopathy [1]. Several surgical techniques have been implemented to repair this lesion. Operative approaches that establish a dual-coronary supply to the heart have been associated with improved outcomes compared with approaches that result in one source for coronary circulation [1–13].

Different techniques have been described to achieve a dual-coronary system, including the creation of intrapulmonary tunnel and coronary transfer. At our institution we have adopted a surgical policy to establish a dual-coronary supply since 1991. Because an intrapulmonary tunnel was associated with several complications, coronary transfer has been our surgical strategy of choice [14]. When a significant gap existed between the anomalous coronary artery and the aorta, we used several previously described modified implantation techniques to facilitate tension-free transfer of distant coronaries [14–16]. Our surgical strategy has evolved with time to use those techniques in all ALCAPA patients. In the current series, we review our experience with those modified implantation techniques and assess postoperative cardiac recovery and long-term clinical outcomes.

	Patients and Methods

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Inclusion Criteria
From July 1991 to July 2007, 30 consecutive children with ALCAPA underwent surgical correction at the King Faisal Specialist Hospital and Research Center in Riyadh, Saudi Arabia. The patients were identified using the surgical database. Clinical, operative, and outcome data were abstracted from the medical records. Approval of this study was obtained from the Research Ethics Board at our institution, and individual consent was waived.

Patient Characteristics
During the study period, 30 children (12 boys, 18 girls) with ALCAPA underwent surgical treatment. The median age at the time of operation was 5.7 months (range, 46 days to 5.45 years), and 16 were aged younger than 6 months. The median weight was 5.35 kg (range, 3.3 to 15.9 kg).

Upon clinical presentation, 12 patients required intensive care unit admission for inotropic support and management of severe congestive heart failure, and 4 were intubated preoperatively. Overall, 23 patients had history of congestive heart failure, whereas 7 were asymptomatic and were diagnosed based on the presence of a heart murmur.

Preoperative Echocardiography Data
The preoperative mean shortening fraction was 0.16 ± 0.02 and the mean ejection fraction was 0.32 ± 0.04, and 8 patients (27%) had a preoperative ejection fraction of less than 0.20. Mean preoperative left ventricular end-diastolic diameter was 4.0 ± 0.14 cm, with a mean Z score 9.1 ± 0.9. The preoperative degree of mitral valve regurgitation (MR) was none in 1 patient, mild in 6, mild to moderate in 2, moderate in 15, moderate to severe in 4, and severe in 2. Left ventricular wall motion abnormalities were present in 24 patients.

Operative Technique
All procedures were performed though a midline sternotomy. Cardiopulmonary bypass (CPB) was established with standard aortic and bicaval venous cannulation. The left ventricle was decompressed by venting through the right superior pulmonary vein. Mild hypothermia (32° to 34°) was used. Once CPB was initiated, the right and left pulmonary arteries were snared to prevent myocardial ischemia secondary to steal of coronary blood flow into the pulmonary artery.

The aorta was cross-clamped, and the heart was arrested with antegrade blood cardioplegia infused into the ascending aorta and into the main pulmonary artery. Additional doses of cardioplegia were infused directly into the orifice of the left coronary artery if needed.

Several techniques have been used in our series. Earlier in our experience, when the anomalous left coronary artery originated from the posterior pulmonary sinus, the coronary ostia was excised with a wide cuff of pulmonary arterial wall to form a coronary button. The coronary button was widely mobilized to allow a tension-free anastomosis. Similarly, an incision was made at the posteromedial wall of the aorta for the anastomosis. The resultant defect in the pulmonary artery was patched with autologous pericardium (Fig 1).

View larger version (26K): [in this window] [in a new window]   Fig 1. Direct coronary transfer for treatment of anomalous left coronary artery from the pulmonary artery. (Left panel) The pulmonary artery is transected. The coronary artery is excised with a wide cuff of pulmonary arterial wall to form a coronary button. (Middle panel) The coronary button is widely mobilized to allow a tension-free anastomosis. (Right panel) An incision is made at the posteromedial wall of the aorta to serve as the site of the anastomosis of the anomalous coronary artery to the aorta.

View larger version (31K): [in this window] [in a new window]   Fig 2. Coronary implantation with autologous flap extensions from aorta posteriorly and pulmonary artery anteriorly. (Left panel) The pulmonary artery is transected. The anomalous left coronary artery is excised as a button together with a tongue of tissue from the anterior pulmonary artery wall. This tongue is used to form the anterior half of the planned tunnel. (Middle panel) The aortic incision is fashioned to create a flap to form the posterior half of the tunnel. The aortic flap is first sutured to the lower margin of the coronary button. (Right panel) The tongue of pulmonary artery wall is then sutured to complete the tunnel as well to close the aortotomy.

View larger version (28K): [in this window] [in a new window]   Fig 3. Coronary implantation with autologous flap extension from pulmonary artery sinuses. (Left panel) The pulmonary artery is transected. Two transverse parallel incisions, one proximal and the other distal to the anomalous coronary artery orifice, are made and extended an equal distance on both sides of the coronary orifice. (Middle panel) The isolated segment of the pulmonary artery containing the origin of the anomalous artery at its center is 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. (Right panel) With the use of a 5-mm aortic punch, an incision is made at the posteromedial wall of the aorta to serve as the site of the anastomosis of the lengthened coronary artery to the aorta.

Mitral valve repair was performed in 5 patients with either severe regurgitation or mitral dysfunction that seemed to be more organic than functional. Our repair technique involved annuloplasty along the posterior leaflet using a polypropylene suture, reinforced with a strip of autologous pericardium, in two layers of running mattress fashion. In addition, papillotomy was performed in 1 patient, and division of secondary chordae from the posterior mitral leaflet was needed in 2 patients to improve the mobility of the retracted leaflet.

Weaning from CPB was done with the use of inotropic support. The sternum was left open in 12 patients, with delayed chest closure performed in the intensive care unit after the swelling had subsided.

Mean CPB and cardiac ischemic times were 108 ± 38 and 57 ± 25 minutes, respectively, for the whole group. These times were somewhat longer when coronary lengthening implantation techniques were used compared with simple direct coronary transfer technique: mean CPB duration was 99 minutes for direct transfer vs 122 minutes for coronary lengthening procedures (p = 0.12), and mean cardiac ischemic duration was 50 minutes for direct transfer vs 65 minutes for coronary lengthening procedures (p = 0.09).

Follow-Up
Late outcomes were determined from recent office visits at King Faisal Specialist Hospital and Research Center or from direct correspondence with the patient's family. The mean follow-up duration was 4.2 ± 4.5 years (range, 2 days to 14.6 years).

Statistical Analysis
All the data were analyzed with SAS 9 software (SAS Institute Inc, Cary, NC). Data are presented as frequency, median with range, or mean ± standard deviation, as appropriate, with the number of nonmissing values indicated. Characteristics and outcomes of patients in the preoperative period vs those at the last follow-up were compared using paired t tests and ² or the Fisher exact test, as appropriate. Unrelated two-group comparisons were done with unpaired, two-tailed t tests for continuous variables and the ² or Fisher exact test for categoric data.

	Results

Top Abstract Introduction Patients and Methods Results Comment Acknowledgments References

 
Operative Mortality
Three patients (10%) died postoperatively of multisystem organ failure (n = 1), ventricular fibrillation (n = 1), and cardiogenic shock after a persistent low cardiac output state (n = 1). One patient in our series received postoperative support with extracorporeal membrane oxygenation (ECMO) and eventually died of multisystem organ failure and inability to wean off ECMO assistance. One patient had sudden cardiac arrest due to ventricular fibrillation 12 days postoperatively and did not receive ECMO; whereas the other patient's operation was in 1991, before wide use of ECMO for postoperative cardiac support at our institution.

Multiple demographic, operative, and era variables were entered into both univariate and multivariable regression analyses to identify factors associated with operative mortality. None of the assessed variables was significantly associated with hospital death, likely due to the small sample size (Table 1).

Clinical Status and Echocardiography Assessment
At the most recent follow-up, all survivors had normal clinical exercise ability and were in New York Heart Association function class I except for 1 patient who was admitted to another hospital for pneumonia a few months postoperatively and had a respiratory arrest resulting in ischemic brain injury.

Follow-up echocardiography in survivors demonstrated a mean shortening fraction of 0.35 ± 0.2 postoperatively compared with 0.16 ± 0.02 preoperatively (p < 0.00001) and an ejection fraction of 0.64 ± 0.3 postoperatively compared with 0.32 ± 0.04 preoperatively (p < 0.00001). Mitral regurgitation was absent to mild in 24 survivors and was moderate in 3 (11%) compared with 70% of patients who had moderate or severe MR preoperatively (p = 0.0002). The left ventricular end-diastolic dimension Z-score decreased from 9.1 ± 0.9 preoperatively to 1.2 ± 0.5 at the latest follow-up (p < 0.00001).

No difference was noted in the echocardiographic recovery between patients who underwent direct coronary transfer vs those who underwent coronary lengthening implantation techniques. In most patients, systolic function, left ventricular dilatation, and MR had recovered by 6 months postoperatively.

	Comment

Top Abstract Introduction Patients and Methods Results Comment Acknowledgments References

 
Surgical management of ALCAPA has evolved over the years into the currently generally accepted standards that include urgent surgical repair soon after diagnosis, the establishment of a dual-coronary artery system, vigilant myocardial protection with administration of multiple doses of blood cardioplegia into the aorta and selectively into the coronary arteries while preventing steal into the pulmonary arteries, creation of a wide anastomosis between the anomalous artery and the aorta, liberal use of delayed sternal closure, and ECMO support when indicated [1–13].

We discuss the results of the modified implantation techniques that we have adopted for the surgical treatment of ALCAPA at our institution.

Implantation Technique
Several techniques have been used by different surgeons to reestablish a dual-coronary system for the treatment of ALCAPA [1–16]. Direct anomalous coronary transfer and implantation into the aorta provides the simplest management option. It is the most frequently adopted surgical technique and has gained popularity through increased experience with coronary artery transfer techniques adapted from the arterial switch operation (Fig 1). The reported mortality rate for direct reimplantation is 0% to 16% [1–13]. Nonetheless, direct coronary transfer has major limitations. When the anomalous coronary arises from the posterior pulmonary sinus close to the commissure between the posterior and the nonfacing sinus, or less frequently when it arises from the nonfacing sinus, there is a long distance between the coronary artery orifice and aorta. In these cases, direct coronary transfer can result in tension applied to the aortocoronary anastomosis resulting in increased incidence of stenosis and obstruction with the risk of persistent ventricular ischemia and development of postoperative cardiac arrest.

Coronary artery bypass grafting using a saphenous vein, the internal mammary artery, or the subclavian artery is associated with major shortcomings such as kinking, stenosis, and late occlusion and is usually not recommended in infants and small children [1].

Creation of an intrapulmonary artery tunnel with aortopulmonary window (Takeuchi operation) has been recommended as the treatment of choice in infants by some surgeons [7, 13]. This procedure has been associated with numerous complications such as the development of supravalvular pulmonary stenosis, baffle leaks creating a coronary–pulmonary artery fistula, and aortic valve insufficiency [7, 13] Overall reported operative mortality has been 0% to 23%, with the reported late reoperation or catheter intervention rate as high as 30% [7, 13].

Several techniques have been described in the literature to avoid any tension on the implanted anomalous coronary artery [14–16]. Sese and colleagues [14] recommended the use of a flap of pulmonary artery wall with a corresponding flap from the aorta to lengthen the implanted coronary artery (Fig 2). We adopted this modification selectively early in our practice in cases of distant coronaries; and later on, our strategy evolved to use this technique in all children with ALCAPA.

This technique allows adequate lengthening of the implanted vessel to bridge the distance between the anomalous coronary and the aorta and to avoid tension on the aortocoronary anastomosis and prevent the development of obstruction that may be amplified in the immediate postoperative period by the dilated left ventricular and the tense pulmonary artery. Extensive dissection of the anomalous coronary artery is not necessary in this technique, which minimizes bleeding complications and the risk of distortion. This procedure preserves the growth potential of the implanted artery because viable native tissues are used. Although this tunnel had to be revised twice intraoperatively in our experience due to inaccurate orientation of the autologous flaps, this was the result of a learning curve and was associated with excellent recovery in both patients after proper implantation. The risk of distortion should not be any different than that in direct coronary transfer when performed by experienced surgeons. We have not noted any evidence of distortion to the aortic valve on follow-up echocardiograms.

Nonetheless, we have noted two late postoperative failures, one at about 8 months and one at about 12 years, as described in the Results section. As noted in many prior publications, most of the patients show significant resolution of their ventricular dilatation, systolic function, and MR between 4 and 8 months postoperatively [1, 9, 10]. Failure to do so should trigger evaluation of persistent coronary ischemia, as was the case in our first reoperation. In addition, late development of MR or ventricular dysfunction, as was the case in our second reoperation, should also encourage further investigation to rule out coronary compromise. No late failures occurred in the patients in our series who received direct coronary transfer, and late stenosis after coronary lengthening techniques does not seem to be higher than that reported in other series mainly using direct coronary transfer in the treatment of ALCAPA [1–13] (Fig 4).

View larger version (18K): [in this window] [in a new window]   Fig 4. Freedom from reoperation after anomalous left coronary artery from the pulmonary artery implantation stratified by implantation technique for direct coronary transfer (dashed line) and coronary extension (solid line) techniques.

Mortality and Risk Factors for Death
Published mortality after dual-coronary repair of ALCAPA is 0% to 16% [1–12]. Hospital mortality after ALCAPA repair was 10% in our series. Previous reports have identified several risk factors for operative mortality such as younger age, operations done before ECMO availability, decreased preoperative left ventricular function, and severity of preoperative MR [1, 3, 9, 10]. Although all deaths in our series were in infants, age was not identified as a risk factor. Similarly, the degree of ventricular dysfunction or MR was not a significant factor in our risk analysis. More important, neither the implantation technique nor the site of origin of the anomalous left coronary artery was associated with hospital death. Nonetheless, our small cohort size and the multiple variables involved limit the power of the study to identify clinically significant risk factors.

Fate of the Mitral Valve
The management of the regurgitant mitral valve at the time of ALCAPA repair varies between institutions and remains controversial. Many surgeons suggest that the improvement in coronary perfusion, ventricular dilation, and papillary muscle dysfunction associated with successful ALCAPA surgery will result in improvement in MR, and therefore, mitral valve repair is not recommended [3, 8, 9–12]. On the other hand, other surgeons recommend surgical repair of severe MR at the time of the ALCAPA procedure owing to the high incidence of persistent MR after coronary reimplantation [2, 7]. Moreover, several reports have shown that the improvement in the degree of MR is generally slower than that of ventricular dysfunction [1, 9, 10]. In our experience, the ALCAPA operation was associated with significant improvement of MR. Nonetheless, because our philosophy towards mitral valve repair has evolved with time, several patients in our series with significant preoperative MR underwent concomitant mitral valve repair at time of their ALCAPA operation, with considerable improvement. Several others with significant MR who did not undergo mitral repair also showed considerable improvement of regurgitation. Therefore, our series is not suitable to address the issue of mitral valve repair requirement. Our current approach is to perform concomitant mitral valve repair in patients with severe MR or in whom preoperative echocardiographic assessment of valve dysfunction reveals a likely organic rather than functional pathology.

Most important, 2 patients who continued to show evidence of persistent or recurrent MR were found to have obstruction of the implanted coronary, and therefore continual MR and ventricular dysfunction should prompt further evaluation of coronary ischemia.

Conclusion
A dual-coronary system in the surgical management of ALCAPA can be established in all patients regardless of the site of origin of the anomalous coronary artery. Coronary extension implantation techniques allow tension-free anastomosis with minimal risk of distortion and a low rate of late occlusion. Regardless of the implantation technique, the ALCAPA operation is associated with acceptable operative mortality, excellent cardiac recovery, and late survival. Although the rate of late coronary occlusion is low, continual ventricular dysfunction or mitral regurgitation should prompt evaluation of persistent coronary compromise.

	Acknowledgments

Top Abstract Introduction Patients and Methods Results Comment Acknowledgments References

 
The illustrations in this article were created by one of the authors but were adapted from similar work by Rachid Idriss that can be seen in reference 1.

	References

Top Abstract Introduction Patients and Methods Results Comment Acknowledgments 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): Bahaaldin Alsoufi Fareed Khouqeer Charles C. Canver Zohair Al-Halees Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Alsoufi, B. Articles by Al-Halees, Z. Search for Related Content PubMed PubMed Citation Articles by Alsoufi, B. Articles by Al-Halees, Z. Related Collections Congenital - acyanotic