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Ann Thorac Surg 2007;83:1451-1457
© 2007 The Society of Thoracic Surgeons
a Section of Cardiothoracic Surgery, James W. Riley Hospital for Children and Indiana University School of Medicine, Indianapolis, Indiana
b Section of Cardiothoracic Surgery, St. Louis University School of Medicine, St. Louis, Missouri
Accepted for publication October 27, 2006.
* Address correspondence to Dr Rodefeld, Section of Cardiothoracic Surgery, Indiana University School of Medicine, 545 Barnhill Dr, EH 215, Indianapolis, IN 46202-5123 (Email: rodefeld{at}iupui.edu).
Presented at the Poster Session of the Fifty-second Annual Meeting of the Southern Thoracic Surgical Association, Orlando, FL, Nov 10–12, 2005.
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Abstract |
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Methods: From 1986 through July 2005, 34 patients with Taussig-Bing anomaly underwent the arterial switch operation. The median age at operation was 21 days. Based on position of the great arteries, patients were divided into group I (side by side; n = 16) and group II (anteroposterior; n = 18). Aortic arch obstruction was present in 18 patients (53%), of whom 16 had prior repair with aortic arch reconstruction. Abnormal coronary artery patterns were present in 9 patients (27%).
Results: There were 4 early deaths and 1 late death (3 from group I and 2 from group II). The actuarial survival rate was 85% at 15 years (81% in group I and 89% in group II). Right ventricular outflow tract obstruction (mean gradient, 46.0 ± 5.5 mm Hg) developed in 5 cases (2 from group I and 3 from group II). One patient underwent reoperation for residual aortic arch obstruction. Freedom from reoperation was 80% at 15 years, and thereafter 85% in group I and 75% in group II. Statistical analysis of potential risk factors revealed no significant identifiers for death or need for reoperation between groups.
Conclusions: The arterial switch operation remains our preferred choice of treatment for children with Taussig-Bing anomaly. The position of the great arteries has no effect on postoperative morbidity and mortality. In the presence of aortic arch obstruction, staged arch reconstruction followed soon thereafter by early intracardiac repair has yielded excellent outcomes in our experience.
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Introduction |
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The evolution of surgical repair for the Taussig-Bing DORV has progressed from atrial baffle operations with intraventricular partition [4], Damus-Kaye-Stansel procedure [5, 6], and Rastelli-type intracardiac baffle and right ventricle-pulmonary artery conduit repair [4, 7, 8] to the arterial switch operation (ASO) with VSD closure [9–12] and intraventricular repair [10, 13, 14]. In addition, Fontan-type procedures may be proposed as a solution for even more complex forms.
Of the intraventricular repairs, one (Patrick-McGoon operation) has been used for anteroposterior great artery anatomy by tunneling left ventricular flow anterior to the pulmonary valve, whereas the other (Kawashima operation) is used for side-by-side great artery anatomy by tunneling left ventricular flow posterior to the pulmonary valve [13, 15]. Because of intraventricular geometric factors that can lead to tunnel stenosis, the Patrick-McGoon operation has been largely replaced by the ASO. The Kawashima intraventricular repair is still favored by some surgeons for patients with side-by-side orientation of the great arteries [13, 15].
Since 1986, we have performed ASO to manage all patients presenting with Taussig-Bing anomaly (side-by-side or anteroposterior great artery relationships). In the presence of aortic arch obstruction, we have utilized an early staged approach to repair these patients. The purpose of this retrospective study is to identify the impact of anatomic relationship of the great arteries and use of an early staged approach on outcomes after ASO in children with Taussig-Bing anomaly.
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Material and Methods |
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Taussig-Bing anomaly is defined as an anomalous ventriculoarterial connection with VSD and DORV. The VSD is subpulmonary in location and the outlet septum is malaligned. In addition, tricuspid-aorta discontinuities are present. The aorta may be positioned rightward and anterior (D-transposition) or alongside the pulmonary trunk (side-by-side orientation). The assigned anatomic diagnosis was based on a combination of two-dimensional echocardiographic and angiographic evidence (by cardiologists) and surgical inspection (by surgeons). Based on position of the great arteries, patients were divided into group I (side-by-side; n = 16) and group II (anteroposterior; n = 18).
There were 20 boys (59%) and 14 girls (41%). The mean age at the time of surgery was 10.5 ± 34.5 months, with a range of 3 days to 16 years (median age, 21 days). Twenty-six patients (77%) were neonates (n = 21) or infants younger than 3 months (n = 5). Associated anatomic lesions are listed in Table 1. Aortic arch obstruction, manifested as aortic coarctation (n = 14) or interrupted aortic arch (n = 4), was common (18 of 34 patients; 53%). Two patients had an additional muscular VSD. Prior palliative and concomitant cardiovascular operations are shown in Table 2. Twenty-two patients (65%) underwent previous palliative procedures. All patients (except 1 with coarctation and 1 with interruption of the aorta) with a coarctation of the aorta and with an interrupted aortic arch underwent early two-stage repair.
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Operative Technique
In the presence of associated aortic arch obstruction, we have approached the majority of these patients with a closely staged combined operative approach. If aortic arch obstruction is present, we have performed early arch repair through thoracotomy, followed closely (1 to 3 weeks) by intracardiac repair through median sternotomy. This has the advantage of simplifying the overall repair at each procedure, yet does not preclude ASO within the neonatal time period. The surgical technique for ASO is well outlined in several previous reports. We perform the ASO using continuous full-flow bypass at 20°C. Myocardial protection is accomplished with intermittent dose of cold 4:1 blood: crystalloid cardioplegia administered every 20 to 30 minutes at 10 to 15 cc/kg. Cardioplegia is given into the aortic root before the aorta is opened and directly into the coronaries while the neoaorta is reconstructed and coronary transfer is being accomplished.
The neoaorta and coronary anastomoses are carried out with 7-0 absorbable monofilament sutures in running fashion. Once the neoaortic anastomosis has been completed, the aortic cross-clamp is released, allowing the aortic root to distend. The coronary artery buttons are excised from their respective sinuses taking most of sinus tissue. The coronary buttons were mobilized for a distance of 4 to 6 mm and allowed to rotate to the location on the distended neoaortic root where they will reside without torsion or tension. The ideal location on the neoaortic root is marked with a fine suture. A stab wound is made at the mark, taking care not to injure the previously marked anterior neoaortic commissure. The aortic cross-clamp is reapplied while a 2.5-mm aortic punch is used to create a site for coronary implantation. The location of the neoaortic commissure is confirmed, and the opening is enlarged if necessary to accommodate the coronary buttons.
Selecting the ideal location for the coronary buttons on the closed and distended neoaortic root has greatly reduced the incidence of left ventricular dysfunction that was observed early (1985 to 1989) in our experience, and subsequently dramatically improved our mortality risk. During this phase, the coronary buttons were reimplanted as the neoaortic anastomosis was completed. The change in coronary implantation technique has eliminated our need to reposition the coronary buttons once implanted. Once coronary artery button transfer is complete, the aortic cross-clamp is removed, and the patient is rewarmed while the neopulmonary valve is reconstructed with a generous pantaloon-shaped patch of fresh autologous pericardium 2.5 to 3.5 times larger than the combined area of the transferred coronary buttons. Using a large pericardial patch to reconstruct the neopulmonary root has greatly reduced our incidence of late supravalvar pulmonary artery stenosis.
In 18 patients (14 coarctation and 4 interrupted aortic arch), the surgical procedures included prior (n = 16) or concomitant (n = 2) aortic arch repair: in 8, subclavian flap type of coarctation repair; in 7, direct anastomosis between the ascending and descending aortic segments; in 2 patients (with interrupted aortic arch), the left common carotid artery was divided, spatulated appropriately and anastomosed as a reverse flap to the descending thoracic aorta; and in 1 patient, synthetic patch aortoplasty of a primary anastomosis. After aortic reconstruction, the pulmonary artery was banded (n = 13) to reduce the distal pulmonary artery pressure to 50% or less of systemic pressure.
The VSD was closed through the right atrium in 7 patients and through the pulmonary artery (neoaorta) in 27 patients. Our approach the Taussig-Bing VSD is from the left side of patient. After dividing the aorta and pulmonary artery for the switch approach, the VSD is closed by starting the patch with a running 6-0 polypropylene sutute on the right inferior aspect of the VSD and running the suture line from right to left, sewing away from the crest of the septum and away from the conduction tissue. The surgeon then carries the suture line anterior and superior to the base of the neoaortic valve. The Lecompte maneuver was performed in all but 1 patient. No tension was noted on the left pulmonary artery in this subset of patients. One had restrictive VSD that was enlarged anteriorly. One patient with subaortic stenosis underwent resection of a hypertrophic infundibular septum during repair.
Statistical Analysis
Variables including demographics, previous palliative procedures, morphology, coronary artery pattern, and operative procedure related variables were assessed by means of univariate analysis utilizing 
2 test and multivariate logistic regression analysis. In the analysis of risk factors for early death, variables with significance levels of 0.1 in univariate analysis were admitted to a multivariate logistic regression model. Factors with p values of less than 0.05 were considered significantly related to early death. Kaplan-Meier analysis was used for the actuarial survival rates and freedom from reoperation rate. Differences in survival curves were assessed by log-rank test. All analyses were performed with standard commercially available statistical software (SPSS, Chicago, Illinois). Early mortality was defined as death during initial hospitalization or within 30 days of operation. Any deaths later than that were defined as late mortality. In this series, all early deaths occurred during the initial hospitalization and all late deaths occurred after discharge from the initial hospitalization.
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Results |
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There was 1 late death in group II at a mean follow-up of 5.1 ± 3.8 years (range, 6 months to 16 years). This patient had undergone ASO repair at 21 days of age. This death occurred 1 year after initial repair and was due to viral pneumonia.
Overall survival including operative mortality according to Kaplan-Meier analysis was 85% at 1, 5, and 15 years. Figure 1 shows the actuarial survival, including operative mortality, which was 81% and 89% at 15 years in patients in group I and group II, respectively. Statistical analysis did not reveal any significant risk factors for early, late, or overall mortality between both groups.
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Overall freedom from reoperation was 93% at 1 year, 80% at 5 and 75% at 15 years (Fig 2) and thereafter 85% in group I and 75% in group II. Statistical analysis of contributing factors revealed no significant risk factors for need for reoperation between groups. The incidence of significant RVOT obstruction was not influenced by the presence of aortic arch obstruction for side-by-side (p = 0.68) or anteroposterior great arteries (p = 1.00), the side-by-side relationship of the great arteries (p = 0.92), the anteroposterior relationship of the great arteries (p = 0.71), or by the right coronary artery crossing the infundibulum for side-by-side (p = 0.84) or anteroposterior great arteries (p = 0.86). The difference between neonatal repair versus older repair was significant in patients with aortic arch obstruction (p < 0.001), but was not significant between single-stage versus staged repair (p = 0.88).
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All 29 survivors are in New York Heart Association functional class I, without medication and are in normal sinus rhythm. At last follow-up, Holter examination performed in 2 patients did not reveal major abnormalities, and Doppler studies showed no subaortic obstruction in 28 and a mild subaortic obstruction (gradient at rest less than 30 mm Hg) in 1 patient. Neoaortic incompetence was moderate in 3 (10%; 3 of 29), and trivial or mild in 26 patients (90%; 26 of 29). Right ventricular outflow tract obstruction (echocardiographic gradient more than 30 mm Hg) was present only in 2 patients (7%). There is no significant difference in neoaortic or neopulmonary obstruction and incompetence between groups.
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Comment |
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TopAbstractIntroductionMaterial and MethodsResultsCommentReferences |
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Intraventricular diversion of left ventricular blood flow to the aorta may provide a physiologic repair; however, this operation is technically difficult to perform. It involves the insertion of a patch through the VSD to the aortic conus, with hypertrophied muscle in the RV possibly necessitating resection of the aortic conal septum and a significant suture load to stably affix the patch in the RV. Snoddy and associates [18] have reported that tunnel repair of the VSD in the small RV may result in RVOT obstruction. In this situation, a valved external conduit from the RV to the pulmonary artery may provide good hemodynamic results. Recently, Binet and colleagues [19] reported treating physiologically complete transposition of the great arteries by closing the VSD and repairing the transposition by means of the Damus-Kaye-Stansel technique. The long-term outcome of this approach is limited, however, by the suboptimal durability of valved external conduits. Reoperation will eventually be necessary due to structural deterioration of biological valved conduits in children [20].
The ASO, which was introduced as a corrective technique for simple transposition of the great arteries, has become the procedure of choice for patients with all forms of D-transposition including those with Taussig-Bing DORV. At our institution, ASO is currently the most commonly used technique for two-ventricle repair of Taussig-Bing DORV.
The ASO is always feasible; however, intraventricular repair seems more attractive as it preserves the native aortic valve and avoids coronary dissection. That has led Yacoub and Radley-Smith [21] to propose a classification according to the extracardiac anatomy: when great arteries are side by side, an intraventricular repair may be performed; when the great arteries relation is more or less anteroposterior, arterial switch may be performed. These conclusions were then discussed by Sakata and Lecompte [3], who could not define any strict relation between the intervalvular distances and the relation of the great arteries. Van Praagh [22] commented that the surgical significance is that when the great arteries are anteroposterior, the aortic valve is very anterior and the VSD-to-aorta conduit (for Rastelli-type conduit repair or intraventricular repair) would have to run anteriorly and would cause iatrogenic pulmonary stenosis. With side-by-side great arteries, the conduit can pass posteriorly and allow unimpeded flow to the pulmonary valve. Based on this previous work, we have developed our guidelines for surgical decision making. Our approach is to first consider the relationship of the great arteries: if anteroposterior, an arterial switch is performed with closure of the VSD. If side-by-side orientation of the great vessels is present, then the tricuspid-pulmonary valve distance is evaluated; if less than aortic valve diameter or if there are abnormal tricuspid valve chordae present, ASO and VSD closure are performed; if this distance is greater, pulmonary artery banding is performed before 1 year of age to permit growth, and then a Rastelli-type intraventricular conduit repair is subsequently performed beyond 1 year of age.
The coronary distribution in Taussig-Bing DORV is often quite different than that in transposition of the great arteries and may be a source of technical difficulties in ASO. In patients in the ASO group, coronary patterns were normal (Yacoub type A) when the great vessels were in an anteroposterior relationship. But when the great vessel orientation is side by side, almost exclusively, other types of coronary artery distribution are encountered (Yacoub type B, D or E). In a detailed pathologic study, Uemura and associates [23] found that a single coronary artery (type B) was present in 27% of hearts with a side-by-side great artery relationship.
Associated aortic arch anomalies are frequently observed in Taussig-Bing DORV [7, 10, 23, 24]. Repair of the aortic arch obstruction in a one- or two-stage procedure remains controversial, although the one-stage procedure has recently become increasingly popular [9, 10]. However, analysis of the overall mortality of patients with associated aortic arch anomalies in this series in which a closely sequenced two-stage repair was utilized, the existence of aortic arch anomalies was not an incremental risk factor, provided that an adequate aortic arch repair was achieved. If aortic arch reconstruction is complicated by an inadequate luminal diameter and pulmonary artery banding is performed, biventricular outflow tract obstruction will result. Finally, as we have performed in several patients in the latter part of our series, a one-stage repair, including aortic arch reconstruction, ASO, and closure of the VSD can certainly be successfully performed in selected patients. A one-stage approach has the advantage of avoiding multiple operations, the development of pulmonary vascular disease, and may be performed in a neonate with suitable ventricular function. However, the optimal strategy utilized depends upon an overall assessment of the patients’ intracardiac anatomy and severity of associated extracardiac anomalies.
Patients with Taussig-Bing DORV face a higher risk of neoaortic incompetence from a mismatch of the large pulmonary artery and a small aorta. Minor neoaortic regurgitation is common after ASO, with reported rates ranging from 5% to 55% [24]. It has been speculated that resection of tissue of the neononcoronary sinus may lead to dilatation of the sinus of Valsalva and adjacent leaflet prolapse, which in turn leads to neoaortic insufficiency [7]. Moreover, the increased preoperative flow across the anatomic pulmonary valve may result in dilatation of the pulmonary valve annulus and postoperative neoaortic insufficiency, or reimplantation of the abnormal coronary arteries into the neoaorta may distort the architecture of the valve. Because the degree of neoaortic regurgitation present in most of our patients was hemodynamically insignificant, further follow-up will be required to determine whether trivial or mild regurgitation will eventually become clinically relevant.
We conclude that Taussig-Bing DORV can be repaired by ASO and VSD closure in neonates and young infants with an excellent outcome. Staged correction of associated cardiac anomalies (especially aortic arch obstruction) closely followed by early intracardiac repair (1 to 3 weeks) had no detrimental influence on survival and later outcome. Therefore, we prefer to perform a two-stage complete repair with each stage closely timed in the neonatal period. The position of the great arteries has no effect on postoperative morbidity and mortality. Right ventricular outflow tract obstruction often is the most commonly observed late complication and is a leading cause for reintervention. To determine secondary effects of neoaortic regurgitation and their clinical relevance, continued follow-up is mandatory after ASO repair of Taussig-Bing DORV.
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