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Ann Thorac Surg 1999;68:1692-1697
© 1999 The Society of Thoracic Surgeons

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Original Articles

Long-term survival and functional follow-up in patients after the arterial switch operation

^a Department of Cardiovascular Surgery, German Heart Center of Munich, Munich, Germany

Address reprint requests to Dr Haas, Department of Cardiovascular Surgery, German Heart Center of Munich, Lazarettstrasse 36, 80636 Munich, Germany
e-mail: haas{at}dhm.mhn.de

Presented at the Thirty-fifth Annual Meeting of The Society of Thoracic Surgeons, San Antonio, TX, Jan 25–27, 1999.

	Abstract

Top Abstract Introduction Material and methods Results Comment References

 
Background. For many years, the arterial switch operation (ASO) has been the therapy of choice for patients with transposition of the great arteries (TGA). Although excellent short- and mid-term results were reported, long-term results are rare.

Methods. Between May 1983 and September 1997, ASO was performed on 285 patients with simple TGA (n = 171), TGA with ventricular septal defect (VSD) (n = 85), and Taussig-Bing (TB) anomaly (n = 29). This retrospective study describes long-term morbidity and mortality over a 15-year period.

Results. Hospital mortality was 3.5% for simple TGA, 9.4% for TGA with VSD, and 13.8% for TB anomaly. Late death occured in 2 patients, 1 with simple TGA and 1 with TGA and VSD. The cumulative survival for all patients at 5 and 10 years is 93%, and at 15 years is 86%. Reoperations were required in 31 patients and were most common for stenosis of the right ventricular outflow tract (RVOT). However, no correlation was found between technical variations on pulmonary artery reconstruction and this type of complication. Forty-six patients underwent follow-up angiography, which revealed five cases with coronary occlusion or stenosis. Follow-up is complete in 96% of the patients from 1 to 15.2 years. Sinus rhythm is present in 97%; 88% of the patients show no limitations on exertion.

Conclusions. The ASO can be performed with low early mortality, almost absent late mortality, and infrequent need for reoperation. The favorable long-term results demonstrate that the ASO can be considered as the optimal approach for patients with TGA and special forms of double-outlet right ventricle.

	Introduction

Top Abstract Introduction Material and methods Results Comment References

 
After the first successful arterial switch operation (ASO) in 1975 by Jatene [1] and the ingenious development by Lecompte [2], anatomic correction has been accepted as the procedure of choice for treatment of children with simple transposition of the great arteries (TGA), TGA and ventricular septal defect (VSD), and for special forms of double-outlet right ventricle (DORV). Although several authors have demonstrated excellent short- and mid-term follow-up of the ASO [3–6], long-term results are rare. Before the ASO can be considered as the optimal approach for these congenital malformations, the assessments of late mortality and morbidity with special regards to the function of the anatomic pulmonary valve, the reconstructed pulmonary artery, as well as the fate of the reimplanted coronary arteries are required. This retrospective study describes the experience with the ASO over a 15-year period at our institution.

	Material and methods

Top Abstract Introduction Material and methods Results Comment References

 
Between May 1983 and September 1997, 285 ASOs were performed by three surgeons at the German Heart Center of Munich. Of these 285 patients, 171 had simple TGA, 85 had TGA with VSD, and 29 patients had transposition-like forms of DORV with subpulmonary (25), noncommitted (2), perimembranous (1), and inlet VSD (1). Patients with Taussig-Bing (TB) anomaly and DORV represent one group in this study. Age at operation for ASO, associated cardiac anomalies, and coronary anatomy are shown in Table 1 for the three different groups. Previous operations prior to ASO were performed in 48 patients. Two of 3 patients with simple TGA underwent resection of aortic coarctation, and in 1, additional pulmonary artery banding was performed. One patient with an angiographically determined small left ventricle also had prior pulmonary artery banding, who underwent ASO 6 days later. Patients with TGA and VSD as well as Taussig-Bing anomaly/DORV displayed significantly more frequent (p < 0.01) and more complex associated anomalies (p < 0.01), which required previous operations in 21 and 24 patients, respectively. PAB (38), resection of aortic coarctation (17), and atrioseptectomy (ASE; 10) were the main previous procedures in those groups. One patient with interrupted aortic arch (type B) previously underwent repair, and 2 patients required a "two-stage" operation, consisting of temporary PAB and aortopulmonary shunt.

Three different surgical techniques were employed for pulmonary artery reconstruction. From 1983 to 1989, direct pulmonary artery anastomosis without any patch material was used in 40 patients according to the original description of Pacifico and associates [8]. Reconstruction with two free pericardial patches was employed between 1984 to 1994 in 101 patients. The "pantaloon patch" technique, introduced by Paillole and associates [9], has been applied since 1990. For postoperative follow-up, data from 256 patients (96% of the survivers) were collected from the outpatient records. Besides clinical follow-up, electrocardiogram (ECG), roentgenogram and two-dimensional echocardiography with Doppler flow studies were performed at 6-month intervals during the first postoperative year and annually thereafter. Doppler peak gradients for the right ventricular outflow tract are demonstrated after discharge, at 1 year, and at the very last examination during follow-up after a mean of 5.8 ± 2.7 years after ASO. For this late examination, only patients with a follow-up of more than 24 months were included.

Statistical analysis was performed by standard techniques; time-related events were examined by actuarial methods. Continuous variables are expressed as mean values ± SD. Associations between categorical data were evaluated using the ² test; the nonparametric Mann-Whitney U test was employed to assess differences in aortic insufficiency and pulmonary gradients between groups. A p value less than 0.05 was considered statistically significant.

	Results

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Mortality
Early mortality occurred in 6 patients (3.5%) with simple TGA. Acute myocardial failure within 12 hours perioperatively, was the primary cause. One patient developed fatal myocardial infarction 10 days after switch operation. In one case, intraoperative death was associated with an intramural course of the coronary artery. Eight patients (9.4%) with TGA and VSD died early postoperatively. Six died due to acute myocardial failure within 24 hours, 1 patient due to renal failure in the presence of overlooked coarctation 14 days after successful switch operation, and 1 due to progressive myocardial failure after 24 days. Acute myocardial failure was associated with an abnormal coronary pattern in 3 patients, and an ectopic left coronary artery far above the sinus of Valsalva was found in 1 patient. Risk analysis for coronary pattern and weight <3,000 g could not show any correlation with early mortality. This was also the case for patients with Taussig Bing anomaly, in which 4 patients (13.8%) died within 24 hours after operation.

Late mortality
Only one cardiac-related deathoccurred in the group of simple TGA. This patient showed progressive mitral valve regurgitation and severe biventricular dysfunction due to idiopathic dilated cardiomyopathy. Any association to myocardial ischemia was excluded by coronary angiography and endomyocardial biopsy. Recent valvuloplasty and mitral valve replacement did not improve myocardial function, and death occurred almost 13 years after ASO before cardiac transplantation could be accomplished. In the group of TGA and VSD, only 1 patient died after 226 days, due to progressive pulmonary hypertension. This patient was 305 days old at the time of ASO with prior ASE and PAB. In the group of Taussig Bing anomaly, there was no late mortality. Actuarial survival rates including early mortality for all patients undergoing ASO were 93% at 5 and 10 years, and 86% at 15 years. Separate actuarial survival for the three groups is demonstrated in Figure 1.

View larger version (11K): [in this window] [in a new window]   Fig 1. Actuarial survival curve with the hospital mortality included. (Dotted line = simple TGA, solid black line = TGA+VSD, black stripes = TB anomaly/DORV.)

Figure 2 shows the mean gradients of the RVOT in relation to the three different surgical techniques for pulmonary reconstruction followed for approximately 6 years. Direct pulmonary artery anastomosis was associated with the lowest gradients after discharge (2.2 ± 4.8 mm Hg) and after 1 year (3.3 ± 7.8 mm Hg), whereas pulmonary reconstruction using pericardial patch material had significantly higher RVOT gradients (p < 0.01). However, this significant difference could not be detected after a mean time of 5.8 ± 2.7 years. At this time, direct pulmonary anastomosis had a mean gradient of 11.2 ± 15.7 mm Hg, whereas two pericardial patches and the pantaloon patch technique showed a mean gradient of 11.3 ± 9.7 and 14.1 ± 15.2 mm Hg, respectively. In contrast to the technique of direct pulmonary anastomosis, in which a progressive and significant increase in pressure gradients occurred after 1 year (p < 0.01), pulmonary reconstruction using pericardial patch material did not show any change in pressure gradients over time. In the early years of our experience, glutaraldehyde was not used for fixation of the pericardium. However, we could not demonstrate any significant difference concerning a higher rate of reoperation.

View larger version (10K): [in this window] [in a new window]   Fig 2. Pulmonary gradients for the three different surgical techniques for pulmonary reconstruction over time, expressed as mean ± SEM. Squares = direct pulmonary anastomosis; circles = two free pericardial patches; triangles = a "pantaloon-shaped" pericardial patch. Numbers indicate the frequency of reoperation according to the three different techniques.

View larger version (20K): [in this window] [in a new window]   Fig 3. Development and severity of neoaortic insufficiency over time for the three different groups. White areas = mild aortic insufficiency; hatched areas = moderate aortic insufficiency; black areas = severe aortic insufficiency.

Left ventricular function was normal in all except 3 children. Severe neoaortic insufficiency was the cause of global functional impairment in 1 patient, whereas coronary occlusion in the forementioned 2 patients resulted in regional myocardial dysfunction. Two patients had severe biventricular dysfunction, due to dilated cardiomyopathy in 1 and severe neoaortic stenosis accompanied by severe RVOT stenosis in the other. In both patients, coronary angiography did not reveal any stenosis. Separate global right ventricular dysfunction was observed in 4 patients. In 1 child, it was associated with severe pulmonary hypertension; the remaining 3 children had severe RVOT obstruction, which was relieved after reoperation.

Follow-up ECG in all but 7 patients showed sinus rhythm. Six patients with complete heart block after VSD closure and 1 patient with sick sinus syndrome required permanent pacemaker implantation. Only 4 patients were on antiarrhythmic medication due to intermittent av-junctional disturbances. Five other patients required digoxin, diuretics, or ACE inhibitors to relieve heart failure symptoms. Eighty-eight percent of all patients have no limitations on exertion; 34% of those patients are either attending public school or working; 54% are in preschool age but normally developing. Reasons for limitation are cardiac related in only 6 patients.

	Comment

Top Abstract Introduction Material and methods Results Comment References

 
This retrospective analysis of our experience with the ASO confirms that the ASO can be accomplished with low early and late mortality in patients with TGA. The reported early mortality of 3.4% and the negligible late mortality for simple TGA agrees well with previous reports [3, 10]. However, it should be emphasized that these results were not influenced by a "learning curve," because in the first 2 years of this series, only patients with TGA and VSD or TB anomaly/DORV were chosen for anatomic correction. The reasons for deaths were in general acute myocardial failure due to imperfect transfer of the coronary arteries. Although 3 out of 6 patients who died early postoperatively had an unusual coronary pattern or an intramural course of the coronary artery, no association was found between these variations in coronary anatomy and early mortality in the present study. However, we have to accept that intramural course is a risk factor, as previously reported [7, 11]. Early mortality for the group of TGA with VSD was significantly higher compared with simple TGA (p < 0.02). However, 3 of these patients were operated on more than 3 months after birth, before the policy was adopted that patients with TGA and VSD should undergo repair within the first 2 months of life to prevent progression of pulmonary vascular disease [12]. Employing our recent policy instituted after 1990 to treat a patient with VSD in the same manner as a patient with intact ventricular septum, early mortality would decrease to 5.8%, which would not be significantly different from that of simple TGA (3.5%). Late mortality was negligible in both groups and was absent in TB anomaly/DORV, which agrees well with the reports of Lupinetti and Hazekamp and associates [5, 13].

In our series, 10.5% of all patients underwent reoperation due to several causes, as reported in other studies [13, 14]. RVOT obstruction represents the most common reason for reoperation. Although several authors have postulated that pulmonary reconstruction using a single "pantaloon-shaped" pericardial patch results in normal pulmonary artery growth and lower RVOT pressure gradients with a decreased incidence for reoperation [5, 9, 14], this study failed to show any superiority of the three different surgical techniques employed. Moreover, pulmonary reconstruction using pericardial patch material had significantly higher RVOT gradients (p < 0.01) after discharge and 1 year after operation compared with the direct pulmonary artery anastomosis. Low RVOT gradients have also been observed by Carrell and associates using the direct pulmonary anastomosis in an equal number of patients [15]. In agreement with our results, it is suggested that pulmonary reconstruction without the use of any foreign patch material has a considerable potential for unlimited tissue growth. Even though, in the present study, this technique showed a progressive and significant increase in pressure gradients after almost 6 years, resulting in similar values for the three different surgical techniques. Why this significant increase did occur so late after ASO is hard to explain. However, due to the fact that each suture line is associated with the development of fibrous tissue, even in the presence of reabsorbable sutures, one may speculate that, in the occurrence of unimpaired growth of the main pulmonary artery, discrete circumferential narrowing will result from the former suture line, which will be detected by sensitive Doppler echocardiography. Mild transpulmonary gradients ( 20 mm Hg) were observed in most of the patients, independent of the surgical technique used. It is an accepted fact that any change or occurrence within these limits does not represent clinical relevance. In contrast to the technique of direct pulmonary anastomosis, pulmonary reconstruction using pericardial patch material did not show any change in pressure gradients over time, indicating sufficient enlargement of the coronary explantation sites either with one "pantaloon shaped" pericardial patch or two free pericardial patches. Due to the fact that we could not find any clinically relevant superiority of one of the three different techniques, our policy at the moment is to continue to utilize one "pantaloon-shaped" pericardial patch.

During follow-up, neoaortic insufficiency showed a progressive and significant increase in all groups. While only 5% of all patients showed mild aortic insufficiency after discharge, after a mean follow-up of almost 6 years, aortic regurgitation is described in almost 30%. However, significant insufficiency occured infrequently, as already reported by others [16, 17]. In contrast to previous studies in which prior PAB has been demonstrated to a be a risk factor for neoaortic regurgitation, we could not find any correlation between these two determinants [17, 18]. As expected, patients with TGA and VSD, as well as with DORV/TB anomaly, displayed a significantly higher frequency of aortic regurgitation than patients with simple TGA. This is related to the consistent preoperative anatomy and increased flow across the anatomic pulmonary valve, resulting in preoperative significant annular differences. In addition, patients with substantial size mismatch between neoaortic root and distal aorta, as is common in patients with VSD, the often difficult reconstructive procedures may result in progressive regurgitation over time.

Although in the majority of patients neoaortic insufficiency does not represent a matter of concern, careful echocardiographic follow-up is mandatory. It is possible that the observation time of up to 15 years is too short and the number of aortic valve replacement will increase during the decades to follow.

Compression of the left main brochus by the posterior displaced aorta was repaired by aortopexy in 3 patients. All other procedures, such as permanent pacemaker insertion and closure of a residual VSD, that have been performed in this series, are not specifically related to the ASO.

Left ventricular function was impaired in only 3 patients up to 15 years after the AOS. In 2 patients, the impairment was due to coronary occlusion, and in 1 patient to severe aortic insufficiency. The low prevalence of left ventricular dysfunction may serve as an indication that coronary transfer can be accomplished successfully, resulting in unimpaired coronary blood flow. In a limited number of coronary angiographic controls, 2 patients showed complete occlusion at the level of the ostium, and 3 patients had coronary stenosis. Although in 1 patient revascularization was required, it has been shown that patients can be asymptomatic, even though coronary occlusion or stenosis but sufficient collateralization is present.

In summary, first, the AOS can be performed with low early mortality, almost absent late mortality, and infrequent need for reoperation. Second, the prevalence of neopulmonary outflow tract obstruction is not associated with the surgical technique used. Third, in the majority of patients, neoaortic insufficiency at the present time does not represent a matter of concern; however, careful follow-up should be performed to detect the minority of patients with clinically relevant regurgitation. Finally, the arterial switch operation can be considered as the optimal approach for the treatment of patients with transposition and special forms of DORV, independent of the coronary pattern present.

	References

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