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Ann Thorac Surg 2001;71:1985-1988
© 2001 The Society of Thoracic Surgeons
a Division of Cardiothoracic Surgery, Washington University School of Medicine, St. Louis Childrens Hospital, St. Louis, Missouri, USA
b Division of Pediatric Cardiology, Washington University School of Medicine, St. Louis Childrens Hospital, St. Louis, Missouri, USA
Address reprint requests to Dr Huddleston, #1 Childrens Hospital, Suite 5S 50, Childrens Hospital, St. Louis, MO 63110
e-mail: huddlestonc{at}msnotes.wustl.edu
Presented at the Forty-seventh Annual Meeting of the Southern Thoracic Surgical Association, Marco Island, FL, Nov 911, 2000.
| Abstract |
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Methods. We reviewed our experience with this lesion at St. Louis Childrens Hospital. Over the past 15 years, 17 infants under 18 months of age have undergone repair, with all but one being treated with reimplantation of the left coronary artery into the aorta; the other underwent the Takeuchi procedure (intrapulmonary artery baffle) and was excluded from this evaluation. The average age and weight at operation were 0.5 ± 0.3 years and 6.1 ± 1.9 kg, respectively. All presented with varying degrees of heart failure and 9 patients also had either moderate or severe MR.
Results. There was one early and no late deaths after reimplantation of the left coronary artery. The left ventricular function postrepair improved from a preoperative shortening fraction of 0.19 ± 0.09 to 0.34 ± 0.08 (p < 0.01). Moderate or severe MR was present in 2 patients postoperatively, and both developed significant obstruction in the left coronary artery postoperatively as well. Both underwent mitral valve repair and revascularization of the left coronary artery.
Conclusions. Excellent results can be obtained in the treatment of this very high-risk group of patients. Mitral valve repair is not generally necessary at the time of the initial operation. However, should MR recur or persist late, it may herald the presence of a coexistent, significant coronary stenosis. Cardiac catheterization should be performed to assess the patency of the left coronary artery before performing mitral valve surgery.
| Introduction |
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| Methods and patients |
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Echocardiographic and cardiac catheterization findings
The initial diagnosis by echocardiography and clinical impression was inaccurate in 9 of the 16 patients. Most were diagnosed with an idiopathic dilated cardiomyopathy. Other purported diagnoses were coarctation of the aorta and ventricular septal defect alone. The diagnosis of ALCAPA was confirmed in 16 of the cases by cardiac catheterization. The average left ventricular end-diastolic pressure was 11 ± 5 mm Hg. Severe global left ventricular dysfunction was present in all patients. M-mode echocardiographic measurements revealed an average shortening fraction of 0.19 ± 0.09. The average left ventricular end-diastolic dimension was 42.0 ± 9.2 mm. MR was graded according to a scale proposed by Helmeke and associates [3]. The ratio of maximum regurgitant jet area to the area of the left atrium was calculated in whatever two-dimensional echocardiographic plane demonstrated the largest area of regurgitation. A ratio of less than 0.2 was graded as mild, 0.2 to 0.4 was considered moderate, and greater than 0.4 severe. MR was present in 12 patients (75%) and was graded as mild in 3, moderate in 5, and severe in 4. Echocardiographic data were obtained on all surviving patients at regular intervals after operation. The shortening fraction, left ventricular end-diastolic dimension, and degree of MR were obtained at 1 week, 6 weeks, 6 months, 1 year, and annually thereafter. Cardiac catheterization was obtained at 1 year after repair to assess patency of the reimplanted coronary artery until 1995; at that time, there was sufficient confidence in the echocardiographic evaluation of the coronary patency to use that as a substitute.
Surgical technique
The preferred method of handling the anomalous coronary artery was removal from the pulmonary artery and reimplantation into the aorta. The aorta was incised anteriorly to assist in identifying an appropriate area in the left sinus of Valsalva and to avoid damage or distortion of the aortic valve. This area was generally slightly higher than the usual origin of the left main coronary artery. The defect in the pulmonary artery was patched and the pulmonary artery closed. Only 1 patient with severe MR preoperatively underwent mitral valve repair using an annuloplasty technique. This patient had the highest left ventricular end-diastolic pressure (24 mm Hg) along with severe MR. Cardiopulmonary bypass time was 83.3 ± 32.4 minutes, with an average aortic cross-clamp time of 39.6 ± 16.8 minutes.
| Results |
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Mitral valve status
As mentioned above, 75% (12/16) of patients had MR preoperatively, with 9 of those having moderate or severe degrees noted by echocardiography. Only 1 patient underwent mitral valve repair at the same time as the reimplantation procedure. As mentioned above, this patient had the highest left ventricular end diastolic pressure (LVEDP) preoperatively. This patient has done well with no residual MR in 3 years of follow-up. As of the latest postoperative evaluation for the entire group, 9 of the 15 hospital survivors had no MR, while 5 had mild and 1 had moderate MR. The degree of MR gradually improved by serial echocardiography, reaching the least amount by 6 months postrepair. Two patients underwent a secondary operation for MR, 1 at 6 years and 1 at 2 years postoperatively. One patient had mild and the other severe MR before reimplantation of the left coronary artery. Both patients had an initial reduction of the MR after left coronary artery reimplantation, but it gradually increased to significant levels. Cardiac catheterization before reoperation demonstrated significant obstruction in the reimplanted left coronary artery in both patients: 100% occlusion in 1 and 90% stenosis in the other. At the time of mitral valve repair, both patients underwent revascularization of the left coronary artery, 1 with left internal thoracic artery bypass graft and the other with patch angioplasty of the left main coronary artery. The mitral valve repair entailed both a chordal shortening procedure with an annuloplasty technique, as there was prolapse of the anterior and posterior leaflets due to fibrosis and elongation of the papillary muscles in both cases. The patient who underwent mitral valve repair and left internal thoracic artery bypass of the left coronary artery has no residual MR and normal exercise stress test at latest follow-up; the other patient has persistent moderate mitral regurgitation and good flow through the patched left main coronary artery by echocardiography. Of the other 13 long-term survivors, 4 have had patency of the reimplanted left coronary artery shown by angiography and 4 others have satisfactory definition of the left coronary patency by echocardiography. The other 5 patients have not had technically satisfactory echocardiography to definitively demonstrate patency.
Left ventricular changes
The left ventricular shortening fraction was derived from the end-systolic and -diastolic dimensions measured in all patients pre- and postoperatively (Table 1). It is interesting to note that the ventricular dimensions decreased slightly in the roughly 4 years of follow-up while the infants grew, as evidenced by the increase in weight and body surface area. When the left ventricular end-diastolic dimension was indexed to body surface area, the difference between pre- and postrepair values was statistically significant. The shortening fraction also improved significantly from an average of 0.19 prerepair to 0.34 postrepair. Serial echocardiograms at the intervals mentioned above revealed that the improvement in left ventricular function occurred by 6 months postrepair with little change in the shortening fraction beyond that time.
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| Comment |
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A rapid reduction in the left ventricular size is the rule for this group of patients. Within 1 week of operation, the left ventricular end-diastolic dimension is generally reduced by 20% from the preoperative value [9]. One would expect the mitral valve annulus dimension also to be reduced by a similar amount, thus producing an effect analogous to two pledgeted stitches in the mitral valve annulus. Why is it that some patients have persistent or recurrent MR? Previously published series dealing with this cardiac anomaly have between 8% and 40% incidence of late MR of at least moderate severity [47]. The reason for the persistence of the MR is occasionally discussed, but not commonly in the previously reported series. From our own experience, we feel that persistence of MR or recurrence after some improvement should lead one to suspect a problem with the reimplanted left coronary artery. In both instances where reoperation was necessary for MR, there was significant obstruction in antegrade flow into the left coronary. What we presume is playing a role is insufficient collateral flow to the papillary muscle resulting in ischemia/infarction with subsequent fibrosis of the tip of the papillary muscle and elongation of the chordal mechanism of the valve. In both cases, a somewhat complex mitral valve repair was performed in conjunction with revascularization of the left coronary artery. It is unlikely that revascularization alone would have had any impact on the degree of MR given what appeared to be fixed changes in the mitral valve structure.
Occlusion or stenosis of the left coronary artery after reimplantation of the anomalous left coronary artery arising from the pulmonary artery is occasionally seen. This is likely related to the traction placed on this vessel to reach the aorta from its position on the posterolateral aspect of the main pulmonary artery. Disruption of the vasa vasorum may also play a role. Although experience with the arterial switch operation for transposition of the great arteries has emboldened congenital heart surgeons to approach the reimplantation procedure in a similar fashion, there are times when alterations in the usual technique may be beneficial [1012]. Nonetheless, when late occlusion of the left coronary occurs after reimplantation, revascularization of the left coronary artery is appropriate as there is a clear risk of ischemia and sudden death late in a single coronary system, whether after the arterial switch operation [13] or repair of ALCAPA [14]. Revascularization may be via either patch angioplasty of the left main coronary artery [15] or coronary bypass using an internal thoracic artery, which can be successfully performed even in very young children [16].
In summary, we found that excellent results can be achieved in infants with ALCAPA using a technique of operation in which the anomalous coronary artery is reimplanted into the aorta. Although some infants may require postoperative mechanical support, the long-term outcome is very good, with substantial improvement of left ventricular function and dimensions. Simultaneous repair of the mitral valve at the time of initial operation is probably not necessary as a general rule. Although severe mitral valve regurgitation is a risk factor for death in some series, the added ischemic time for this procedure adds to the risk in an infant with very compromised ventricular function. Should MR persist or recur after the initial operation, we would recommend cardiac catheterization to evaluate the patency of the reimplanted coronary artery before proceeding with mitral valve repair.
| Footnotes |
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| Discussion |
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I looked at your paper and abstract a few weeks ago, and it was remarkable just how similar your experience has been with ours at Childrens Memorial Hospital.
Did you use transesophageal echocardiography for your patients? We have had very good results using this modality. In fact, one of our patients did not show flow in the left main coronary artery when we came off bypass, and we went back and revised the anastomosis, which resulted in better flow. The other issue involves abnormal coronary artery anatomy. Did you have any unusual coronary artery distribution, that is to say, the anomalous coronary artery coming off the right pulmonary artery or some other place, which might compromise your problem?
(Slide) This shows just how similar your experience is with ours. We had 17 patients: 16 aortic implantations and 1 Takeuchi procedure. We had a 0% mortality and we had no late deaths. Interestingly, and perhaps fortuitously, we did not use ECMO or LVAD for our postoperative management. It may be patient selection. It may be the way we give the cardioplegia. I wonder if you could also make a comment about how you deliver the cardioplegia.
I enjoyed this paper very much. Thank you.
DR HUDDLESTON: Thank you, Dr Mavroudis. I think that the experience from Childrens Memorial Hospital in Chicago has been excellent. In fact, out of my 18 references in the manuscript, two come from your institution about this entity.
Yes, we use transesophageal echocardiography, at least we have since the early 1990s, as a routine, and with that have had an excellent experience through our cardiologists of visualizing left coronary flow and have been very pleased with that. The 2 patients who had secondary operations for their mitral regurgitation and stenosis or occlusion in the left coronary artery had documented flow in the left pulmonary artery that seemed adequate at the time of operation. So it did not help in that regard.
We had only 1 patient in whom the coronary artery arose from a spot other than sort of the usual location, and that was just up further onto the left main pulmonary artery, and by its position made it a little bit easier to do the reimplantation technique, because it was a little bit closer to the aorta in that position.
The ECMO experience or the mechanical support experience in our series is actually a little more typical of all the other series published in the literature in that, generally speaking, somewhere between 5% and 20% of other patients reported with this entity have required either an LVAD or an ECMO postoperatively. I do not know what else to say about that. Two of the patients out of the 3 could not be separated from cardiopulmonary bypass. The third in our experience was a patient who had recurrent ventricular tachycardia, had it before operation, and because of recurrent episodes immediately after operation, I put that patient on ECMO just to control that particular problem better. He only stayed on ECMO for about 24 hours and then was removed.
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