Asian Cardiovasc Thorac Ann 2007;15:240-242
© 2007 Asia Publishing EXchange Ltd
Repair of Anomalous Origin of Left Coronary Artery from the Pulmonary Artery
Hong-Wei Guo, MD,
Jiang-Ping Xu, MD,
Yun-Hu Song, MD,
Xiang-Dong Shen, MD,
Shou-Jun Li, MD,
Sheng-Shou Hu, MD
Department of Cardiovascular Surgery, Fu Wai Hospital, Chinese Academy of Medical Sciences, Peking Union Medical College, Beijing, China
For reprint information contact: Hong-Wei Guo, MD Tel: 86 10 8839 8063 Fax: 86 10 6831 3012 Email: ghwdr{at}hotmail.com, Department of Cardiovascular Surgery, Fu Wai Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, 167 Beilishi Road, Beijing 100037, China.
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ABSTRACT
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Patients with anomalous origin of the left coronary artery from the pulmonary artery often have mitral valve regurgitation. Although establishing dual coronary circulation is the procedure of choice, there remains controversy as to how the mitral valve is handled. Between April 1999 and August 2005, 8 patients underwent surgical correction at our institution. There were 4 males and 4 females, aged from 9 months to 13 years (mean, 6.4 years). Six patients underwent direct aortic reimplantation and 2 had a Takeuchi procedure. Simultaneous mitral annuloplasty was performed in 7 patients with moderate or severe mitral regurgitation. There were no deaths or postoperative complications. Follow-up ranged from 4 to 80 months (mean, 34 ± 26 months). Left ventricular function improved significantly from a preoperative fractional shortening of 0.21 ± 0.09 to 0.35 ± 0.06. Mitral regurgitation decreased on follow-up in the 7 patients who had mitral annuloplasty. We recommend performing mitral annuloplasty at the time of operation in patients with moderate or severe mitral regurgitation and anomalous origin of the left coronary artery from the pulmonary artery.
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INTRODUCTION
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Anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA) is a rare lesion. To establish a double-ostium coronary system, correction of the aberrant vessel is recommended.1,2 There are 2 major surgical options for 2-coronary repair: aortic reimplantation (direct coronary transfer), and the Takeuchi operation (intrapulmonary tunnel repair).3 However, the approach to the mitral valve remains controversial. We reviewed our experience of 2-coronary repair of ALCAPA and mitral annuloplasty at Fu Wai Hospital.
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PATIENTS AND METHODS
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Between April 1999 and August 2005, 8 patients with ALCAPA underwent surgical intervention at our institution. There were 4 males and 4 females, aged from 9 months to 13 years (mean, 6.4 years). Electrocardiograms indicated abnormalities of the ST segment and T wave in 5 patients, left ventricular hypertrophy in 4, left axis deviation in 3, and sinus arrhythmia in 2. The preoperative cardiothoracic ratio ranged from 0.53 to 0.63 (mean, 0.59). Diagnosis was made on the basis of echocardiography in 7 patients by demonstrating a dilated right coronary artery with retrograde flow through the left coronary artery into the main pulmonary artery (PA). One patient was diagnosed by echocardiography with a fistula of the left main coronary artery to the main PA. Severe mitral regurgitation (MR) was seen in 2 patients, moderate MR in 5, and mild MR in 1. Coronary angiography confirmed the diagnosis of ALCAPA in all patients, demonstrating collateral circulation between the right and left coronary arteries and a coronary steal phenomenon into the PA (Figure 1
).
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Figure 1. Selective coronary angiography showing an anomalous left coronary artery originating from the pulmonary artery (ALCAPA). Ao = aorta, PA = pulmonary artery, RCA = right coronary artery.
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Surgery was performed through a median sternotomy on cardiopulmonary bypass using bicaval and single aortic cannulation and moderate hypothermia. Myocardial protection was achieved by antegrade cardioplegia through the aortic root after cross clamping the aorta. As a result of abundant intercoronary collaterals, delivery of cardioplegia could be simplified by placing gentle digital pressure over the left coronary artery as the cardioplegic solution was infused into the ascending aorta. Six patients underwent direct aortic reimplantation. The main PA was opened transversely, and the location of the anomalous orifice was identified. Venting was achieved via the main PA. The main PA was divided, the anomalous origin of the left coronary artery was excised with a button of the PA, and subsequently mobilized. The left coronary button was anastomosed to the aorta. The coronary artery transfers respected the course and origin, and the repairs were not limited only to the anomalous origin. The coronary defect in the PA sinus was closed with an autologous pericardial patch.
The other 2 patients had a Takeuchi procedure. An aortopulmonary window was created in the left wall of the aorta and the right wall of the PA using a 4-mm puncher.3 An autologous pericardial patch was used to construct an intrapulmonary tunnel between the aortopulmonary window and the anomalous origin of the left coronary artery. The anterior wall of the PA was widened with an autologous patch. Simultaneous mitral annuloplasty was performed in 7 of the 8 patients. The clinical details of all 8 patients are given in Table 1. We recognized infarction and fibrosis of the papillary muscle in 2 patients at the time of repair, one in the anterior and the other in the posterior papillary muscle. The commissure plication annuloplasty used here was similar to that described by the groups of Kay4 and Reed5 with 4/0 or 5/0 Prolene suture used for the annuloplasty.
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RESULTS
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There were no deaths or postoperative complications. All patients recovered uneventfully. Follow-up ranged from 4 to 80 months (mean, 34 ± 26 months), and no late deaths occurred during the follow-up period. All patients showed a progressive decrease in left ventricular size. The left ventricular end-diastolic dimension decreased significantly from a preoperative value of 44.71 ± 7.18 mm to a postoperative value of 39.29 ± 6.10 mm ( p = 0.011). The fractional shortening improved significantly from 0.21 ± 0.09 to 0.35 ± 0.06 ( p = 0.006).
We did not find flow abnormalities in the main PA on echocardiography in those who had undergone the Takeuchi procedure. Preoperative mitral incompetence had decreased both at the time of discharge and at follow-up. Clinical details of the mitral annuloplasty are given in Table 1
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DISCUSSION
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Anomalous origin of the left coronary artery from the pulmonary artery is a rare but serious congenital anomaly, also called Bland-White-Garland syndrome. Anomalous origin of the left coronary artery from the pulmonary artery does not present prenatally because of favorable fetal physiology that includes equivalent pressure in the main PA and aorta secondary to a nonrestrictive patent ductus arteriosus, and equivalent oxygen concentrations due to parallel circulations. However, symptoms occur as PA pressure diminishes after birth, and flow in the left coronary artery reverses. Insufficient collateral flow from the right coronary artery and coronary steal from the left coronary artery into the pulmonary trunk result in myocardial ischemia. Approximately 80% to 90% patients with ALCAPA die during the first year of life from heart failure or sudden cardiac death if surgical correction is not performed. Current surgical procedures are directed at creating a 2-coronary artery system via either direct reimplantation or the Takeuchi procedure.
There is still controversy as to the best management of MR associated with ALCAPA. Mitral regurgitation is related both to ischemic left ventricular dilatation with mitral annulus enlargement and to ischemic dysfunction of the papillary muscles. Some reports advocate not interfering with the mitral valve when the initial operation is performed.6–9 Others recommend routine mitral annuloplasty in patients who have varying degrees of incompetence at the time of coronary artery revascularization.10 On the other hand, some groups advise mitral annuloplasty only in the presence of severe MR.11,12 It is difficult to conclude which approach is best because the duration of follow-up and the numbers of patients differ. As experiences accumulate and follow-up periods lengthen, the criteria for mitral valve repair in ALCAPA may be determined in the future. In our series, mitral annuloplasty was performed for moderate or severe MR in 7 patients, and the case of mild MR was left untreated. During follow-up of 34 ± 26 months, all 7 patients showed mild MR. We believe that mitral annuloplasty for moderate or severe MR is beneficial in the acute phase after 2-coronary repair; pre and postoperative mild MR is acceptable.
It was concluded from our experience in these 8 patients that good mid-term results can be achieved after surgical intervention for ALCAPA, although more cases and a longer follow-up are required. We recommend that simultaneous mitral annuloplasty be performed in patients with ALCAPA who have moderate or severe mitral incompetence.
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REFERENCES
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- Kay JH, Egerton WS, Zubiate P. The surgical treatment of mitral insufficiency and combined mitral stenosis and insufficiency with use of the heart-lung machine. Surgery 1961;50:67–74.[Medline]
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ABSTRACT
INTRODUCTION
