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J Thorac Cardiovasc Surg 1994;108:354-362
© 1994 Mosby, Inc.
SURGERY FOR CONGENITAL HEART DISEASE |
Berlin, Germany
Received for publication Nov. 2, 1993. Accepted for publication Jan. 28, 1994. Address for reprints: V. Alexi-Meskishvilli, MD, German Heart Institute Berlin, Department of Thoracic and Cardiovascular Surgery, Augustenburgerplatz 1, 13353 Berlin, Germany.
Abstract
Between January 1991 and June 1993, eleven children with anomalous origin of the left coronary artery from the pulmonary artery underwent direct aortic reimplantation of the left coronary artery at the German Heart Institute Berlin. The patients' ages ranged from 2.5 months to 10.5 years; six were infants. Three infants were intubated and their lungs ventilated before the operation, and one was resuscitated 2 days before the operation. The electrocardiograms of eight patients indicated deep Q waves. All but three of these patients had insufficient collaterals between the right and left coronary arteries. The entire group exhibited reduced left ventricular ejection fraction (minimum 15%) including mitral valve incompetence, which was moderate in six patients and severe in three. All six infants underwent emergency operations, and the remaining children, who were older, underwent elective operations involving moderate hypothermic perfusion and cold crystalloid cardioplegia. Aortic crossclamping time ranged from 22 to 79 minutes (mean 54 minutes). A two–coronary artery system was established in all patients by direct reimplantation of the anomalous left coronary artery into the ascending aorta. Three patients who also exhibited severe mitral valve incompetence underwent modified Kay mitral valve annuloplasty. A delayed sternal closure procedure (closure performed 1 to 10 days after the operation) was used on eight patients. A 10-month-old patient was successfully treated after the operation with a centrifugal left heart assist device and a 9-year-old patient received extracorporeal membrane oxygenation because of severe heart failure. No postoperative deaths occurred. Left ventricular end-diastolic volume decreased dramatically after the operation and returned to near normal values 1 to 9 months postoperatively. At the same time, the preoperatively depressed left ventricular ejection fraction returned to normal and mitral valve incompetence decreased or vanished in eight patients. Color Doppler echocardiograms (eleven patients) and coronary angiograms (three patients) indicated that the reimplanted left coronary artery was patent in all eleven patients during the follow-up period. Reimplantation of the left coronary artery into the ascending aorta is an effective method of establishing a two–coronary artery system in children with anomalous origin of the left coronary artery from the pulmonary artery. Mitral valve annuloplasty is recommended for patients who also have severe mitral valve incompetence. Prolonged assisted circulation must be used in cases of severe postoperative heart failure. (J THORACCARDIOVASCSURG1994;108:354-62)
Anomalous origin of the left coronary artery (LCA) from the pulmonary artery (PA) continues to present a challenge for cardiac surgeons. The methods proposed for creating a two-coronary system vary, but direct reimplantation of the anomalous LCA to the aorta has yielded greater success, especially in infants and children, 
1,2 and therefore has become the preferred procedure. This article presents the results of direct aortic reimplantations of the anomalous LCA in eleven patients at the German Heart Institute Berlin.
PATIENTS AND METHODS
The study population included all patients with anomalous origin of the LCA from the PA at our institution since the inception of the pediatric cardiac program in May 1986. None of the patients died before the operation. During the same time period, three adults (27, 35, and 56 years old) with anomalous origin of the LCA from the PA underwent successful establishment of a two-coronary system; the repair was achieved by aorta-coronary saphenous vein bypass in two patients and by a Takeuchi operation in one. The adult patients are not included in the subsequent analysis. Between January 1991 and June 1993, eleven children with this anomaly underwent direct reimplantation of the anomalous LCA to the aorta at the German Heart Institute Berlin (Table I). Seven of the patients were girls and four were boys. Their ages ranged from 2.5 months to 10.5 years. Six infants (range 2.5 to 10 months old; mean 4.9 months) underwent emergency operations because of symptoms of severe left ventricular (LV) dysfunction and congestive heart failure. Three were intubated and their lungs ventilated (1 to 5 days) before the operation because of cardiopulmonary instability; one was resuscitated 2 days before the operation. Five older children (range 3 to 11 years old; mean 5.9 years) underwent elective operations. The electrocardiograms of all the infants and two of the older children showed myocardial infarction patterns. Cardiomegaly (cardiothoracic ratio between 0.57 and 0.68; mean 0.63) was observed in ten patients, and one, a 9-year-old boy, had a cardiothoracic ratio of 0.35. With the exception of a cardiac murmur, this patient was completely free of symptoms. Nine patients had varying degrees of mitral valve incompetence (mild to moderate in six; severe in three). All of the patients underwent preoperative cardiac catheterization. A detectable left-to-right shunt at the PA level was found in two patients (9 and 10.5 years old). LV end-diastolic pressure was higher than normal in six of the nine patients in whom it was measured (Table I). Eight patients had insufficient collaterals between the right coronary artery (RCA) and LCA, which is defined as delayed passage of contrast medium from the RCA via collateral vessels into the LCA distribution and the absence of detectable arteriovenous shunt at the pulmonary trunk level. Echocardiographic examinations, which included determining LV end-diastolic volume and ejection fraction, were performed on most of the patients before the operation and serially 2 weeks to 9 months after the operation. The paired t test was used for comparing preoperative and postoperative echocardiographic data. The group's preoperative mean LV end-diastolic volume index was significantly above normal (143 ± 85 in infants; 158 ± 97 in children). LV ejection fraction was severely impaired (less than 30%) in six patients, mildly impaired (35% to 45%) in three, and normal in two. The preoperative ejection fraction was 28% ± 8% in infants and 46% ± 18% in children.
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The pulmonary sinuses were designated right, left, and nonfacing according to the view from the nonfacing sinus looking toward the aorta. 3 The LCA originated from the right pulmonary sinus in seven patients, from the nonfacing sinus in three patients, and from the pulmonary bifurcation in one.
After separation of both great arteries and transection of the PA above the level of commissures was accomplished, coronary transfer was performed in ten cases according to the technique described by Neirotti and associates 2 (Fig. 1, A to E). The ostium of the LCA was excised with a sizable cuff of the adjacent sinus of Valsalva and the LCA subsequently was mobilized up to the origin of the left circumflex artery. After the opening was made in the left lateral wall of the ascending aorta, the LCA button was anastomosed to the aorta with 7-0 absorbable continuous suture. Because extra length was required to ensure tension-free communication with the ascending aorta in an infant in whom the LCA originated from the nonfacing pulmonary sinus (patient 6, 
Table I), a pulmonary artery flap, constructed according to a technique described by Stiles, 4 was augmented with autologous pericardium (Fig. 1, F).
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Additionally, because of severe (grade III) mitral insufficiency, three patients (patients 5, 7, and 8; Table I) underwent a modified Kay mitral annuloplasty. The giant left atrium was plicated in two of these patients. Aortic crossclamping time ranged from 22 to 79 minutes (mean 54 minutes). After bypass the chest was left open in seven children: in two because of severe ventricular arrhythmia (patients 4 and 7; Table I); in three because of hemodynamic instability, which developed after temporary sternal closure (patients 1, 2, and 5; Table I); and in another two because of implantation of LV and right ventricular (RV) assist devices. One infant's chest (patient 3, Table I) was reopened 6 hours after the operation because of bleeding and the development of severe low cardiac output syndrome.
RESULTS
No early or late deaths occurred during the 4- to 18-month follow-up period. Six patients had low cardiac output after extracorporeal circulation; four of them required inotropic support for 24 to 36 hours after the operation. Two patients could not be weaned from extracorporeal circulation because of severe LV and RV dysfunction. The first patient was a 10-month-old infant (patient 6, Table I) with severely impaired LV function (ejection fraction 15%; LV end-diastolic pressure 20 mm Hg; cardiothoracic ratio 0.65) who had been referred as a candidate for heart transplantation. Coronary reimplantation was performed instead. After bypass, the sternum was left open while LV assist with a centrifugal pump (BP-50, Medtronic Bio-Medicus, Eden Prairie, Minn.) was provided for 9 days. When repeated transesophageal echocardiography indicated that LV function had improved, LV assist was discontinued and the chest was closed on the tenth postoperative day. The infant was extubated on the twelfth postoperative day. LV ejection fraction had increased from 15% before the operation to 46% after 2 weeks and 56% after 1 month.
The second patient, a preoperatively asymptomatic 9-year-old boy (patient 10, Table I), had severe RV failure after LCA reimplantation and also could not be weaned from bypass. The sternum was left open while extracorporeal membrane oxygenation (Avecor Cardiovase Inc., Plymouth, Minn.) was provided with a centrifugal pump (BP-50, Medtronic Biomedicus) for 5 days. After RV function had improved, extracorporeal membrane oxygenation was discontinued and the chest was closed on the seventh postoperative day. The child was extubated on the ninth postoperative day. Three months after the operation, LV and RV function was found to be normal.
For the remaining patients, the sternum was closed 1 to 5 days after coronary reimplantation. Polytetrafluoroethylene surgical membrane was used for pericardial substitution in five patients. No mediastinal infection was observed. The postoperative course was uneventful for the three older children and they were extubated 6 to 18 hours after the operation.
All patients showed significant improvement after coronary reimplantation. Color Doppler echocardiograms (eleven patients) and coronary angiograms (three patients) (Fig. 2) indicated that the reimplanted LCA was patent in all eleven patients during the follow-up period.
Mitral incompetence diminished in severity or disappeared in eight patients and remained unchanged in one. Three patients who underwent mitral annuloplasty later exhibited only insignificant (grade 0 to I) mitral insufficiency. None of the patients had signs of aortic valve insufficiency. Mild supravalvular pulmonary stenosis was found in three patients.
A postoperative improvement in LV function was observed in all of the patients. Three months after the operation the LV end-diastolic volume had decreased to 56 ± 12.7 ml/m 2 in the infants and to 71.1 ml/m 2 in the children. The previously impaired LV ejection fraction improved progressively and 3 months after operation had attained a near normal range in both the infants (52.6% ± 7%) and the older children (55% ± 6.1%) (Fig. 3).
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Anomalous origin of the LCA from the PA is a rare congenital anomaly that often causes myocardial ischemia and infarction within the first few weeks or months after birth, subsequently leading to LV dysfunction, mitral valve incompetence, and congestive heart failure. Even with medical treatment, mortality in infants with signs of LV ischemia and infarction approaches 85% during the first year of life. 5 Developing collateral circulation between RCAs and LCAs allows the patients to live somewhat longer; however, long-term follow-up studies show that many of these patients later die suddenly, usually of arrhythmia, and congestive heart failure and angina develop over time in the remaining patients. 5-7 Thus surgical repair appears to provide the only viable hope for extended survival.
Several surgical methods have been developed for treating anomalous origin of the LCA from the PA. Ligation of the LCA at its origin from the PA is rarely performed today because of high early and late mortality, especially in small infants. 8 A study of the LV function and myocardial perfusion of eight patients who preoperatively had had poor intercoronary collaterals was conducted by Kececioglu and colleagues 9 3 to 21 years after the patients had undergone ligation of the anomalous LCA. All of the patients showed LV ischemia during exercise scintigraphy.
Establishing a two-coronary system is the main goal of surgical therapy today. 1,2,10,11 The continuity of the LCA with the aorta may be established by several methods. In 1966 Cooley, Hallman, and Bloodwell 12 reported two cases of anomalous origin of the LCA from the PA in children in whom an LCA–aortic anastomosis was created by means of Dacron graft in one case and with autologous saphenous vein in another. Reports about similar procedures appeared later. 13,14 Long-term studies showed that graft occlusion and stenosis, especially in young children, were the major drawbacks of this method. 15-17
Some surgeons recommend anastomosis with the left 18 or right 19 subclavian artery to preclude the necessity of cardiopulmonary bypass or the occurrence of myocardial ischemia. A number of surgeons consider this the method of choice. 20-22 However, fatal myocardial fibrillation during the creation of an anastomosis, late anastomosis stenosis, subclavian artery kinking, and right arm ischemia present formidable obstacles when this procedure is used. 13,23 Moreover, in patients with associated severe mitral valve incompetence, extracorporeal circulation may be required during mitral valve repair or replacement.
Anastomosis between the internal thoracic artery and LCA has also been proposed as a surgical method for treating anomalous origin of the LCA from the PA, 24,25 but the procedure is difficult to perform in small infants and late patency of the anastomosis is uncertain.
A two-coronary system can be created by a Takeuchi operation. 26 The mortality associated with this procedure is low, but occurrences of supravalvular pulmonary stenosis, baffle obstruction, and aortic regurgitation in the late postoperative period have been reported. 10,11,27
Performing a Takeuchi operation is difficult in cases in which the anomalous LCA has a high origin or is located adjacent to pulmonary cusps. The Takeuchi operation, aorta-coronary bypass, and internal mammary artery anastomosis have been recommended for some adult patients with anomalous origin of the LCA from the PA because of the somewhat anterior location of the vessels on the pulmonary artery and the stretch that is necessary for coronary reimplantation. 27,28
Direct reimplantation of the anomalous LCA to the aorta is an attractive approach. First performed by Neches and colleagues, 29 this procedure has been successfully used ininfants 1,2 and adults. 7,30 The operation is coupled with low mortality and a high rate of anastomotic patency, and it brings about a dramatic improvement in LV function, 1,31 as substantiated by the results of this study. Surgical difficulties, particularly those encountered when the anomalous LCA originates from the left or nonfacing pulmonary sinuses or adjacent to pulmonary cusps, can be avoided during direct reimplantation by modifying the surgical techniques 4,32 to provide an extra length of anomalous LCA. A modified procedure using an extension of the LCA with autologous pericardium was successfully used in one of the patients in this study.
Myocardial preservation is of paramount importance during reimplantation of the anomalous LCA. Aortic crossclamp time can be reduced during the construction of a pulmonary anastomosis after the aortic clamp is released. To achieve uniform distribution in the myocardium of both ventricles, we now inject cardioplegic solution into both great arteries.
Delayed sternal closure was used in eight of the eleven patients in this study. Other researchers 33,34 have also found that delayed sternal closure provides better conditions for treating low cardiac output, particularly when hemodynamic instability initially occurs after temporary sternal closure or becomes aggravated. Subsequent sternal closure does not increase the risk of mediastinal infection. For patients with residual mild mitral incompetence, polytetrafluoroethylene surgical membrane can be used for pericardial replacement to reduce the danger of repeat sternotomy in the event of subsequent mitral valve surgery.
Assessing the recovery potential of myocardial function is difficult in patients with anomalous origin of the LCA from the PA because of the lack of precise methods to detect irreversible myocardial damage. For this reason, some authors have had to perform orthotopic cardiac transplantation in infants with severely damaged LV function. 34,35 Taking into consideration that implantation of the anomalous LCA to the aorta leads to the recovery of global LV function in patients in whom it is severely depressed, heart transplantation is difficult to recommend for these patients. One such patient in this study, a 10-month-old infant with severe LV dysfunction who was initially considered for cardiac transplantation, underwent reimplantation of the anomalous LCA. The patient required support with an LV assist device for 9 days, but afterward the child had an uneventful recovery period during which LV function improved dramatically.
The exact cause of RV failure in another patient, who was postoperatively supported with extracorporeal membrane oxygenation, was not clear. Incomplete myocardial protection, caused by extensive coronary collateral development leading to washout of cardioplegic solution, was assumed. During the operation, excessive blood flow from the transected LCA and spontaneous myocardial fibrillation occurred despite multiple doses of cardioplegic solution. Presumably, deep hypothermia or continuous cardioplegic infusion may be preferable in such a situation to prevent myocardial damage. Several reports have been published concerning the use of an LV assist device and extracorporeal membrane oxygenation after surgery for anomalous origin of the LCA from the PA. 36,37 Karl, Horton, and Mee 38 concluded that this syndrome presents a unique situation in which isolated LV dysfunction, which is sometimes aggravated by surgery and cardioplegia, can be successfully treated with an LV assist device because of the absence of multiorgan failure. It appears that such an approach should be contemplated when a patient with anomalous origin of the LCA from the PA is being considered for cardiac transplantation.
Mitral valve surgery in patients with anomalous origin of the LCA from the PA is controversial. It has been shown that establishing a two-coronary system in patients with mild or moderate mitral valve incompetence leads to improved mitral valve insufficiency, which gradually decreases, albeit without completely going away. Although patients show an improvement in global ventricular function, normally they are not free of symptoms because of residual mitral valve incompetence. 1,11,20,21 Simultaneous correction of severe mitral valve incompetence in patients with anomalous origin of the LCA from the PA appears to be a logical means of supporting cardiac output during the critical immediate postoperative period.
Several authors have reported conducting perioperative or postoperative mitral valve repair or replacement in patients with severe mitral valve incompetence. 10,11,27 Performing mitral valve reconstruction and establishing a two-coronary system concurrently in the three patients with severe mitral valve incompetence did not significantly prolong aortic crossclamp time and prevented aggravating hemodynamic instability after the operation.
Mean aortic crossclamp time in three patients with concomitant mitral valve reconstruction was 56 minutes, compared with 46 minutes in four patients with origin of the LCA from the right-facing pulmonary sinus who did not undergo mitral valve reconstruction. No patient with mitral valve reconstruction in this series exhibited severe hemodynamic instability after the operation. One patient was extubated on the day of the operation, another on the first postoperative day, and a third, who was intubated because of preoperative cardiopulmonary instability, was extubated on the fourth postoperative day.
Modified Kay annuloplasty results in an immediate improvement in mitral valve function. None of three patients in this study who underwent modified Kay annuloplasty had a recurrence of mitral incompetence during the follow-up period.
Surgery to create a two-coronary system in patients with anomalous origin of the LCA from the PA should not be delayed, because patchy myocardial fibrosis accompanied by secondary impairment of LV function occurs early in infancy. 3,6,31
The serial echocardiographic data recorded in this study indicated progressive postoperative improvement of LV dysfunction in patients with anomalous origin of the LCA from the PA (Fig. 3). Short-term follow-up results revealed an unexpectedly favorable potential for myocardial recovery within 1 year after direct aortic reimplantation of the anomalous LCA. Some authors reported similar improvements in various LV function parameters after a two-coronary system was established. 10,13,15,20,22,31,39-43
The prognosis for patients with severely reduced LV contractility and endocardial fibroelastosis after LCA reimplantation is not clear. Because of the possible presence of scars and fibrosis, an improvement in LV echocardiographic parameters (end-diastolic volume, ejection fraction) does not necessarily indicate that myocardial perfusion abnormalities and regional wall motion, especially during exercise, are also normal. 31 More long-term studies are necessary, but thus far the immediate and short-term results are promising. On the basis of our experience at the German Heart Institute Berlin, we recommend that LCA reimplantation be performed on children with anomalous origin of the LCA from the PA as soon as this anomaly is diagnosed.
Addendum
Since the submission of this manuscript, a 6-year-old boy with anomalous origin of the LCA from the PA has successfully undergone LCA reimplantation into the aorta. The postoperative course was uneventful and the LV ejection fraction increased from 32% before the operation to 45% 2 weeks after the operation.
Acknowledgments
We thank Jonathan Davis for editorial assistance in the preparation of the manuscript.
Footnotes
From the Department of Thoracic and Cardiovascular Surgery a and the Department of Pediatric Cardiology, b German Heart Institute Berlin, Germany. 
J THORAC CARDIOVASC SURG 1994;108:354-62
*Gore-Tex surgical membrane, registered trademark of W. L. Gore & Associates, Inc., Elkton, Md.
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