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Ann Thorac Surg 1997;64:495-502
© 1997 The Society of Thoracic Surgeons
Cardiac Surgical Unit, Royal Children's Hospital, Melbourne, Australia; and Department of Pediatric and Congenital Heart Surgery, The Cleveland Clinic, Cleveland, Ohio
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Abstract |
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 TopFootnotes Abstract IntroductionPatients and MethodsResultsCommentReferences |
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Methods. Fourteen patients have had the operation performed since July 1989. Age and weight medians were 12 months (range, 0.5 to 120 months) and 8.2 kg (range, 3.2 to 34 kg). All but 1 patient had a left ventricular to right ventricular pressure ratio greater than 0.7, due to a large ventricular septal defect (with or without a previous pulmonary artery band), severe congestive heart failure caused by right ventricular dysfunction and tricuspid insufficiency, or a pulmonary artery band for left ventricular retraining. At least 10 patients had strong contraindications to "classic" repair, including right ventricular hypoplasia (n = 2), moderate to severe right ventricular dysfunction (n = 5), or moderate to severe tricuspid insufficiency (n = 9).
Results. There was one hospital death, occurring in a neonate (7%; 95% confidence interval = 0% to 34%). Actuarial survival beyond 10 months is 81% (95% confidence interval = 42% to 95%), currently with 389 patient-months of total follow-up time. The median grade of tricuspid insufficiency fell from 3/4 preoperatively to 1/4 postoperatively (p = 0.003). Right ventricular function is normal in 11/12 current survivors, all but 1 of whom are in New York Heart Association class I or II.
Conclusions. Senning plus arterial switch operation is a good option for selected patients with congenitally corrected transposition of the great arteries with a similar or lower early risk (as compared with classic repairs). Some of the long-term problems associated with congenitally corrected transposition of the great arteries may be avoided with this strategy.
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Introduction |
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TopFootnotesAbstractIntroductionPatients and MethodsResultsCommentReferences |
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Congenitally corrected transposition of the great arteries* (ccTGA), or atrioventricular (AV) and ventriculoarterial discordance, is very rare in isolation and is commonly complicated by a large ventricular septal defect (VSD) and a degree of obstruction of the left ventricular (LV) outlet. The anomaly is found in both situs solitus and situs inversus, usually but not always corresponding with levocardia or dextrocardia, respectively. There is a high incidence of heart block and structural abnormalities of the tricuspid left AV valve, leading to eventual insufficiency in most cases. Although the systemic right ventricle (RV) in ccTGA may function well into adult life, there is a tendency toward deterioration after the second decade. Children with additional lesions, especially tricuspid insufficiency (TI), typically encounter problems much earlier in life. The potentially unfavorable natural history of ccTGA (with additional cardiac lesions) for patients with a systemic RV has been documented in numerous reports [1–4]. The fact that some patients with ccTGA reach adulthood without symptoms usually implies an absence of associated defects, but our past surgical experience has included patients with TI as the sole associated anatomic defect, suggesting that VSD and LV outflow tract obstruction (LVOTO) are not necessary to generate an unfavorable natural history.
Repair of ccTGA has been possible since the late 1950s. The "classic" repair was typically performed for ccTGA with VSD and LVOTO, involving VSD closure and reconstruction of the LV outflow tract, usually with an extracardiac LV to pulmonary artery (PA) valved conduit. Even in the more recent era, operative risk for classic repairs has varied from 4% to 15% with actuarial survival as low as 55% to 83% at 10 years [3–8].
Although survivors of classic repairs at the Royal Children's Hospital have been primarily in New York Heart Association class I, 16/24 had at least moderate TI, and 12/22 had significant RV dysfunction at rest, worsening with exercise [3]. These changes were noted within 3 years of operation in more than half the affected patients, and those with elevated pulmonary-to-systemic flow ratio preoperatively were at highest risk. Heart block was acquired in 27% of children during the course of the procedure. Similar findings have been noted by others, and in nearly all reported series, there has been an increasing hazard function for late death after surgical treatment, primarily due to congestive heart failure [9, 10]. This situation is reminiscent of that of patients with late failure of the systemic RV after Mustard and Senning operations for concordant transposition of the great arteries (TGA), but even less favorable due to the added burden of a structurally abnormal tricuspid valve [11].
Our dissatisfaction with results of classic repairs encouraged exploration of an alternate strategy that would place the LV and mitral valve in the systemic circuit. The arterial switch operation (ASO) and Senning techniques were combined at the Royal Children's Hospital by Mee and associates in July 1989 (Fig 1
). The "double-switch" operation had already been performed in Japan by Yamagishi and associates earlier the same year [12]. The combined Senning plus ASO has now evolved to become the treatment of choice for patients with ccTGA who meet the following criteria:
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Patients and Methods |
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).
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). The preoperative LV/RV pressure was at least 0.7 in all patients but 1. The anatomic and physiologic basis for this pressure distribution is also noted in Table 2. Many patients in the series had features that presented a strong contraindication to classic repair, including mild to moderate RV hypoplasia (n = 2), moderate to severe TI with leaflet dysplasia (n = 9), and moderate to severe RV dysfunction (n = 5). Details of preoperative and postoperative tricuspid valve and RV function are presented in Tables 2 and 3. Two patients were ventilator- and inotrope-dependent just before the operation, and 4 already had complete heart block.
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-blockade with phenoxybenzamine. Patient 2 also had repair of a hypoplastic aortic arch (see Table 2
) using a period of circulatory arrest. The ASO was generally performed first, using a technique similar to that employed for concordant TGA [15]. Coronary translocation was accomplished using medially based rectangular flaps cut into the facing sinuses. Although the great vessel position was side by side (aorta to left) in all patients, coronary translocation could be performed after mobilization of the proximal vessels in all but 1 patient. The Lecompte maneuver was used in 13/14 cases. Three patients required the addition of homograft neopulmonary arterial wall to obtain a pulmonary anastomosis with minimal tension. The Senning procedure was performed according to previously described techniques [16]. A flap of limbic tissue, or polytetrafluoroethylene if necessary, was used to separate the pulmonary veins from the tricuspid valve compartment. Autologous atrial flaps were used to construct the intercaval pathway, leaving the coronary sinus and mitral valve in the new pulmonary venous atrium. The pulmonary venous atrium was closed using atrial tissue, or augmented with autologous pericardium if necessary.
Ventricular septal defect closure was performed via the right atrium and mitral valve in all but 1 patient. The latter patient, with situs solitus/dextrocardia, had a trans-left atrial–trans-tricuspid valve approach. The technique of de Leval and associates [5] was employed to avoid injury to the anterosuperior conduction system.
Postoperative management proceeded according to our institutional protocols for open heart operations in children, but special attention was paid to LV function in patients who had subsystemic preoperative LV pressure, emphasizing low-dose inotropes and afterload reduction.
Special Problems Encountered
The first patient in our series had undergone VSD closure and tricuspid valve replacement 12 weeks before the Senning plus ASO. In the interim, severe RV dysfunction developed. At the original operation, a planned ASO had been abandoned because of the presence of a single coronary artery from the nonfacing sinus, making direct translocation impossible. Due to lack of options, he was returned to operation and had a Senning plus ASO, with extension of the single proximal coronary artery using a 6-mm bovine carotid arterial graft. Just before the second operation, LV/RV pressure ratio was 0.5. He required rapid LV retraining using a ventricular assist device, but eventually made a good recovery.
A second child (patient 6) had severe tricuspid insufficiency with congestive heart failure and suprasystemic PA pressure (without VSD or PA band). Because of extreme dysplasia of the tricuspid valve and presumed pulmonary vascular disease, he had Senning, ASO, and concurrent tricuspid replacement with a 16-mm Carbomedics (Austin, TX) prosthesis, using a transatrial septal approach.
A third child (patient 8), with ccTGA and VSD, aged 15 days, had undergone coarctation repair on day 8 of life. He already had severe TI, and was not a good candidate for PA banding. After Senning plus ASO, hemodynamic instability developed in the intensive care unit and he required an LV assist device and then extracorporeal membrane oxygenation for resuscitation and support after a cardiac arrest. He died after 5 days, with minimal signs of recovery of ventricular function. Permission for autopsy was not granted.
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Results |
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All survivors are in New York Heart Association class I or II with the exception of patient 4. Postoperative, structural, and hemodynamic data (echocardiography, catheterization, or both) are available for all patients, and are summarized in Tables 2 and 3. Of the 13 early survivors, 12 had normal resting RV function as assessed by two-dimensional echocardiography (n = 13) and gated radionuclide study (n = 4). Tricuspid and mitral valve function (before and after Senning plus ASO), assessed with two-dimensional echocardiography, is summarized in Table 1. Most patients with TI had an improvement after the operation. The median grade of TI fell from 3/4 preoperatively to 1/4 postoperatively (p = 0.003).
There have been no problems related to obstruction in the Senning pathway, and all patients but 1 have widely patent ventriculoarterial connections by echocardiographic assessment. Mild neoaortic insufficiency was present in 2, and mild pulmonary insufficiency in 2. Postoperative angiographic studies have been performed in 4 patients. Except for the patient who required revision of a stenotic left coronary graft, all coronary arteries were normally patent. No other reoperations have been performed, except for pacemaker placement or revision in children with congenital or acquired heart block.
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Comment |
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TopFootnotesAbstractIntroductionPatients and MethodsResultsCommentReferences |
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As with many new procedures, however, the earliest candidates are often the worst. They are often selected by virtue of being unsuitable for the existing operations due to the presence of various risk factors and contraindications. A number of patients in our series would probably not have survived classic repair, and had no surgical option other than Senning plus ASO. A direct comparison of results of classic repairs and Senning plus ASO is likely to be biased toward classic repair, as the patient cohort is more favorable. Given this fact, one might conclude that Senning plus ASO is the better overall option, but only after the operation can be offered confidently to the lower risk subset can the question of improved long-term outlook be resolved. Increasing expertise and experience with the ASO will ameliorate some concerns over possible technical problems and the complexity of operation. Due to the paucity of candidates for the procedure, however, it may be many years before detailed analysis in large numbers of patients becomes possible. However, based on this series, results with other operations for the same anatomic condition, and our experience with patients with complex concordant TGA, some inferences are possible regarding indications for and outcome of this operation.
Left Ventricular Outflow Tract
In ccTGA, at least in the {S,L,L} form, a direct resection of LVOTO (without causing heart block) is unlikely to result in an unobstructed connection. Furthermore, the pulmonary valve itself may be mildly to severely abnormal, raising further concerns about ASO. Other associated forms of LVOTO encountered in ccTGA include subvalvar membranes, membranous septal aneurysm, valve excrescences, fibrous tissue tags, or malalignment of the ventricular septum. Many patients with ccTGA, LVOTO, and VSD who are unsuitable for Senning plus ASO may be better candidates for an Ilbawi operation, if otherwise suitable. This strategy will also allow placement of the mitral valve, LV, and aortic valve in the systemic circuit [13, 18].
Right Ventricular Size
In ccTGA, hypoplasia of one ventricle may occur, and is usually associated with an overriding or straddling AV valve, or other positional anomalies, making septation problematic. Many such patients will eventually be better Fontan candidates, if otherwise suitable. Even without a straddling AV valve, the prospects for classic septation are poor in the presence of a small RV. In our series, 2 patients had abnormally small RV cavities, (estimated volume less than 80% of LV) and both survived ASO plus Senning operation. It is possible that the ASO plus Senning operation is the most suitable option for hearts with only mild to moderate degrees of hypoplasia, although the limits of this strategy are currently untested.
Atrioventricular Valves
Tricuspid insufficiency has emerged as the most important risk factor for death after classic repairs [20]. Even without operation, there is a high risk of TI development by the third decade of life. Because the tricuspid annulus is poorly supported, modest degrees of RV dysfunction may induce important degrees of TI, resulting in a vicious circle. Tricuspid valve repair may be technically unsatisfactory, and replacement carries a significant risk [6–9, 20, 21]. Long-term outlook after isolated procedures to improve tricuspid competence is poor [21]. In our own patients, Senning plus ASO appears to improve or at least stabilize TI in most patients. This outcome is quite different to that of classic septation strategies. Tricuspid insufficiency may resolve without any specific plastic procedures on the valve, by reducing RV pressure or improving RV function.
Although mitral valve abnormalities are also seen in a subset of hearts with ccTGA, clinically important mitral insufficiency is not a common feature of the natural history [22]. An important degree of preoperative mitral insufficiency might preclude Senning plus ASO, unless concurrent valve repair is feasible. Closure of the VSD through the mitral valve potentially places the latter at risk for injury and subsequent incompetence. However, mitral insufficiency has been rare in our series, despite the fact that all VSD closures but one were performed with a transmitral approach (see Table 2). Only 1 survivor had an increased grade of mitral insufficiency postoperatively.
Ventricular Function
A similar effect has been noted in regard to RV function after Senning plus ASO for ccTGA. Although Senning plus ASO is not associated with complete resolution of preoperative RV dysfunction in all cases of ccTGA, the majority of our patients have normal function of both RV and LV postoperatively (see Table 2). Although measurable deterioration developed within 3 years of operation in many patients in the Royal Children's Hospital series undergoing classic repair, follow-up time for Senning plus ASO is short, and further comparisons over the long term will be required. The available data and results of Senning plus ASO are encouraging, and favorable effects on RV function after conversion of ccTGA hearts to an LV-dependent systemic circulation have been demonstrated in other series as well [12, 13].
Heart Block and Arrhythmias
The propensity of patients with ccTGA to acquire heart block with or without operation is well known to pediatric cardiac surgeons [23–25]. In hearts with a discordant AV connection, the atrial septal attachment to the cardiac fibrous skeleton is rightward of the ventricular septal attachment [24]. The AV node, at least in {S,L,L} hearts, consequently may be physically unattached to the ventricular septum [25]. It is estimated that 5% to 10% of patients with ccTGA have complete heart block at birth with an increase of 2% per year. Other lower grade AV conduction delays are even more common. Closure of the VSD is therefore an important, but not sole factor in generation of heart block. Use of the de Leval technique, with suture placement on the RV side of the septum (ie, working through the VSD), has not eliminated the risk of postoperative heart block in our own patients (30%; 95% confidence interval = 7% to 65%). It is possible that a transaortic (transneopulmonary arterial) approach to VSD closure in suitable patients might further reduce this risk. Also, in patients with discordance of atrial situs and position of the apex, a trans-left atrial/tricuspid valve approach to the VSD could further reduce the possibility of trauma to the conduction axis.
Wolf-Parkinson-White syndromes and other accessory pathway syndromes are strongly associated with ccTGA [26]. Two patients in our series had clinically important supraventricular tachycardia, without specific signs of preexcitation on electrocardiography. In 1 case the arrhythmia was noted preoperatively (patient 2, who died 10 months after operation). In the second (patient 4), chronic ß-blockade has been required for control.
Coronary Arteries
Typically, patients with discordant TGA have coronary arterial branching that is mirror image to that seen in concordant TGA (sinus 1 usually gives rise to the artery supplying the RV, and sinus 2 to the arteries supplying the LV). However, there may be malalignment of aortic and pulmonary sinuses, so that true "facing" sinuses (a nearly constant feature of concordant TGA) do not exist. The main coronary trunks are also known to be shorter than in concordant TGA. These features may complicate but are unlikely to preclude safe coronary translocation using standard ASO technique with medially based rectangular flaps [15]. Two patients in our series had a single coronary artery arising from a nonfacing sinus: in 1 of them it could not be translocated without the use of a tube graft. The second child had a direct translocation, but did not survive operation. We have not seen this type of coronary anatomy in any of our 400 concordant TGA cases. The relatively high incidence of problematic coronary anatomy in this series may be due to chance alone, but as there are only 14 patients under study we cannot reach any firm conclusions regarding the general question of ASO suitability.
Atrial Switch
We prefer the Senning technique for atrial reconstruction in combination with ASO. Our familiarity with this approach and its general superiority to the Mustard procedure in the long term in most published TGA series are also considered. The presence of dextrocardia in a situs solitus heart (25% of cases) does not preclude a Senning operation, as exposure under cardioplegic conditions should be adequate. The Senning operation should also be technically feasible from the left side for cases of situs inversus. Senning neoseptation can be difficult in a very small chamber, especially in younger children and infants. Although we try to avoid the use of prosthetic material, augmentation of the left atrium with autologous pericardium (preferably employing an in situ technique) is advisable in some of these cases, especially in those with discordance of atrial situs and position of the apex. Long-term problems associated with the Senning procedure in concordant TGA (pathway obstruction, RV dysfunction, arrhythmia) have been rare in this series, although follow-up time is short in comparison. One might still expect to encounter pathway-related problems, and perhaps atrial arrhythmias, at an incidence similar to that seen for post-Senning concordant TGA patients. Post-Senning problems such as RV dysfunction and progressive TI theoretically should be less likely to occur with Senning plus ASO, due to the presence of a systemic LV and mitral valve.
Reoperations
Excluding pacemaker implantation and revision, the only patient in our series requiring reoperation was patient 1, who had a coronary graft used at the initial operation. The probability of reoperation in ccTGA after classical repair is primarily related to the presence of LV-to-PA conduits, TI, and the need for pacemaker implantation and revision. Therefore if we consider only patients without LVOTO, ie, those also potentially suitable for Senning plus ASO, the likelihood of reoperation over the first decade will probably be similar. Results of the Senning operation and the ASO for concordant TGA would suggest that the reoperation probability is very low over the first decade of follow-up, which should also apply to the same operations applied in the double switch.
Potential Technical Problems for Arterial Switch Operation
Great vessel position may complicate coronary translocation and great artery anastomosis. In patients with solitus artrial arrangement (14/14) in this series) the aorta is most commonly leftward (lateral) to the PA, and the outflow tracts are parallel, not in a spiral relationship. The opposite arrangement is seen in ccTGA patients with situs inversus. As in the Taussig-Bing anomaly, coronary translocation may be more difficult with side-by-side vessels, requiring more extensive coronary artery mobilization than usual. The use of medially based rectangular flaps is especially helpful in this situation. Size mismatch between the great vessels (as in concordant TGA with VSD or the Taussig-Bing anomaly) can usually be handled by simple gathering of the proximal neoaorta into the distal native aorta.
Timing of Operation
Timing will vary from patient to patient, depending on associated anomalies and clinical condition. Key issues include maintenance of an adequate LV pressure before Senning plus ASO, and avoidance of pulmonary vascular disease. Neonates with symptoms due to excessive pulmonary blood flow should probably undergo PA banding in preparation for a definitive operation at about 12 months of age. Such a situation is unusual in ccTGA, as even with a large VSD pulmonary blood flow tends to be lower than in infants with concordant AV connections. Babies with severe TI and RV dysfunction might not tolerate placement of a PA band, and primary correction may be a better option in this subset. Only 2 children in our series (1 of whom was ventilator dependent) required neonatal correction (patients 2 and 8). In children without excessive pulmonary blood flow, operation could be considered at 3 to 12 months of age. The atrial reconstruction is somewhat easier at this time in comparison with neonates.
Role of Left Ventricular Retraining
Although 80% of ccTGA patients have a large VSD [2], not all will have high pulmonary blood flow, due to the effect of LVOTO. Some of the features that cause LVOTO may also reduce the size of the VSD, creating a pressure restriction and allowing ventricular "deconditioning" to occur. Such patients, as well as children who have undergone classic septation and who subsequently deteriorate, might still be considered candidates for Senning plus ASO after a period of LV reconditioning. This can be accomplished using a PA band to induce LV hypertrophy, with Senning plus ASO 6 to 12 months afterward [11]. Although this strategy has been extensively tested in concordant TGA after atrial repair, the application in ccTGA has, to date, been quite limited. Our experience includes 2 patients (patients 3 and 7), both of whom survived operation. The banding to Senning plus ASO intervals were 6 months and 10 months, respectively. Finally, in patients who had a systemic LV in the relatively recent past, rapid ventricular reconditioning might be accomplished postoperatively using an LV assist device, as in patient 1 [27]. We have employed a similar strategy for some patients undergoing ASO (for concordant TGA with intact ventricular septum) after 3 weeks of age [28].
Conclusion
The Senning plus ASO is a suitable option for selected children with ccTGA. Initial risk of operation is similar to that of classic repairs, and long-term outlook is likely to be better. We can currently recommend this operation for otherwise suitable children with ccTGA, a systemic LV pressure, and nonobstructed outflow tracts. The presence of TI, mild to moderate RV hypoplasia, or RV dysfunction are features that would strongly favor Senning plus ASO over classic repair, but the operation is also eminently suitable for those without these features.
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TopFootnotesAbstractIntroductionPatients and MethodsResultsCommentReferences |
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Address reprint requests to Dr Karl, Victorian Paediatric Cardiac Surgical Unit, Royal Children's Hospital, Flemington Road, Parkville, 3052, Melbourne, Australia (e-mail: cardiac{at}cryptic.rch.unimelb.edu.au).
* In this article the terms "congenitally corrected transposition of the great arteries" and "discordant transposition of the great arteries" are considered equivalent and used interchangeably. "Left" and "right" ventricle refer to morphology rather than spatial orientation. "Tricuspid" and "mitral" refer to valve leaflet morphology, and do not imply a specific type of atrioventricular connection. 
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