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Ann Thorac Surg 1996;61:1330-1338
© 1996 The Society of Thoracic Surgeons

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Original Articles: Cardiovascular

Taussig-Bing Anomaly: Arterial Switch Versus Kawashima Intraventricular Repair

Divisions of Cardiovascular-Thoracic Surgery and Cardiology, The Children's Memorial Hospital, and Departments of Surgery and Pediatrics, Northwestern University Medical School, Chicago, Illinois; and Division of Cardiology, Kosair Children's Hospital, and Department of Pediatrics, University of Louisville, Louisville, Kentucky

	Abstract

Top Footnotes Abstract Introduction Material and Methods Results Comment References

 
Background. Current corrective surgical approaches for the Taussig-Bing heart include arterial switch with ventricular septal defect (VSD) closure and intraventricular repair as described by Kawashima.

Methods. Between 1983 and 1994, 20 children underwent intracardiac repair of Taussig-Bing anomaly. Mean age at operation was 17 months (range, 1 week to 9 years). Prior palliation included pulmonary artery band (15) with coarctation repair (8) and atrial septectomy (1). Arterial switch with VSD closure was performed in 16 patients, 10 with anteroposterior great arteries. Kawashima repair was performed in 4 patients, all with side-by-side great arteries.

Results. After arterial switch, there was one operative death (6.2%) due to myocardial ischemia and three late deaths (18.7%) due to pulmonary hypertension, gastrointestinal bleeding, and acute lymphocytic leukemia. In the Kawashima repair group there have been no deaths. After arterial switch, 9 patients underwent 11 reoperations for residual coarctation (3), residual pulmonary artery stenosis (2), aortic valve replacement, aortic valvuloplasty, unrecognized VSD, mitral valvuloplasty, mediastinitis, and pacemaker insertion. After Kawashima repair, 1 patient underwent reoperation for baffle stenosis and 1 for an unrecognized VSD.

Conclusions. For children with Taussig-Bing anomaly, the Kawashima intraventricular repair (for side-by-side great arteries) preserves the native aortic valve and avoids coronary dissection. The arterial switch operation with VSD closure can be applied without ventriculotomy to all great artery relationships and allows neonatal repair with or without concomitant coarctation repair. Both techniques yield excellent early and intermediate-term results despite the high rates of prerepair palliation and postrepair reoperation for both groups.

	Introduction

Top Footnotes Abstract Introduction Material and Methods Results Comment References

 
See also page 1338.

Defining the Taussig-Bing heart has been the subject of some controversy, mostly centered around whether this malformation is indeed a double-outlet right ventricle [1] or whether the pulmonary artery trunk ``overrides'' the left ventricle [2] (Fig 1). In 1949, Taussig and Bing [3] reported on a morphologic syndrome consisting of transposed aorta, subpulmonic ventricular septal defect, and levo-position of the pulmonary artery. They emphasized that overriding of the pulmonary artery was an integral part of the malformation. Lev and associates [4] coined the term ``Taussig-Bing heart'' in 1950 and subsequently introduced the concept of a spectrum of Taussig-Bing hearts depending on the degree of pulmonary artery overriding (right-sided, intermediate, and left-sided) [5]. Despite this controversy, most authors of surgical reviews describe the Taussig-Bing heart as double-outlet right ventricle, subpulmonic ventricular septal defect, and subarterial conus or coni, perhaps more because of established usage rather than an endorsement of one interpretation over the other. Whatever the definition, the goal of therapy is anatomic correction whereby the resultant repair connects the morphologic left ventricle to the aorta (or neoaorta) and the morphologic right ventricle to the pulmonary artery (or neopulmonary artery). The options for anatomic repair are determined by great artery orientation, internal cardiac morphology [6], and associated lesions, which include aortic coarctation, small ascending aorta relative to the pulmonary artery, multiple ventricular septal defects, straddling mitral valve, and tricuspid valve attachments to the septum.

View larger version (66K): [in this window] [in a new window]   Fig 1. . Taussig-Bing heart: artist's cut-away view showing the malalignment subpulmonary ventricular septal defect (VSD), subaortic conus, and partial overriding of the pulmonary artery trunk.

Present-day controversy between arterial switch and the Kawashima intraventricular repair centers around (1) anatomic factors favoring one or the other procedure, such as the distance between the tricuspid valve and pulmonary valve for an internal baffle, the relationship of the transposed great arteries (anteroposterior versus side-by-side), and the intraventricular anatomy, specifically mitral and tricuspid valve attachments; (2) neonatal one-stage application that includes concomitant coarctation repair [23]; and (3) the relative long-term benefits and drawbacks of each operation. The purpose of this article is to compare and contrast our experience with arterial switch versus the Kawashima intraventricular repair.

	Material and Methods

Top Footnotes Abstract Introduction Material and Methods Results Comment References

 
Between 1983 and 1994, 20 infants and children (15 boys, 5 girls) underwent intracardiac repair of Taussig-Bing anomaly at Children's Memorial Hospital of Chicago (n = 18) and Kosair Children's Hospital of Louisville (n = 2). Sixteen patients had arterial switch with ventricular septal defect closure (switch group) and 4 had the Kawashima intraventricular repair (Kawashima group). Mean ages at reparative operation were 1.47 ± 2.68 years and 1.37 ± 1.8 years for the switch and Kawashima groups, respectively. Prior palliative operations were common in both groups. Thirteen of 16 patients (81%) underwent pulmonary artery band placement (7 with concomitant coarctation repair, 1 with concomitant atrial septectomy) before arterial switch operation. Four of the 13 patients had band placement and arterial switch operation within the same hospitalization; interval between palliation and repair ranged from 7 to 38 days (mean, 25.5 ± 14.5 days). Interval between band placement and repair for the remaining patients in this group ranged from 28 days to 6.65 years (mean, 1.40 ± 2.17 years). One patient had bilateral modified Blalock-Taussig shunts before arterial switch repair. That patient had accessory mitral valve tissue and a tricuspid valve pouch obstructing the left ventricular outflow tract [24]. Two patients had no palliation before arterial switch intracardiac repair, both at 1 week of age.

Two of 4 patients (50%) had pulmonary artery band placement (1 with concomitant coarctation repair) before Kawashima operation. The intervals between palliation and repair were 0.43 and 3.76 years. The low number of patients in this group precludes statistical comparison of the banding–intracardiac repair intervals. Two patients required no palliation before Kawashima intracardiac repair at 0.38 and 0.60 years of age.

Arterial switch with ventricular septal defect closure was performed in 16 patients. One of these patients had two ventricular septal defects closed. Ten had anteroposterior and 6 had side-by-side related great arteries. All patients had favorably facing aortic and pulmonary artery sinuses of Valsalva for coronary artery transfer regardless of great artery relationship. The coronary artery anatomy (Leiden classification [25]) was sinus I (left anterior descending, circumflex)-sinus II (right coronary artery) in 12, sinus I (left anterior descending)-sinus II (circumflex, right coronary artery) in 3, and sinus I (left anterior descending, right coronary artery)-sinus II (circumflex) in 1. The ventricular septal defect was closed through the right atrium in 10, through the pulmonary artery (neoaorta) in 5, and through both the pulmonary artery and right atrium in 1. The Lecompte maneuver was accomplished in 14. One patient had aortic relocation for coarctation (ascending aorta-to-descending aorta end-to-end anastomosis) and right ventricle-to-pulmonary artery valved homograft conduit placement [23] (Fig 2) in combination with arterial switch.

View larger version (46K): [in this window] [in a new window]   Fig 2. . Arterial switch, ventricular septal defect closure, and aortic relocation: one-stage neonatal repair of (A) the Taussig-Bing heart with subaortic obstruction, small ascending aorta, and coarctation of the aorta. (B) After great artery transection and coronary artery transfer are accomplished, the coarctation is repaired under circulatory arrest by coarctectomy with end-to-end anastomosis between the ``proximal'' ascending and descending aorta. A longitudinal incision is made in the lateral ascending aorta, which is now oriented in a transverse position for anastomosis to the neoaorta. (C) Completed repair showing the neopulmonary artery reconstruction with a valved homograft after subvalvular resection.

View larger version (49K): [in this window] [in a new window]   Fig 3. . Kawashima intraventricular repair: Artist's cut-away view of the Taussig-Bing heart with side-by-side great arteries. (A) The subpulmonic ventricular septal defect (VSD) and subaortic conus are represented in relationship to the great arteries and semilunar valves. (B) The excised subaortic conus is shown in preparation for (C) the left ventricle-to-aorta intraventricular tunnel, shown here constructed with a Dacron patch and interrupted pledgeted sutures. (Ao = aorta; PA = pulmonary artery; SVC = superior vena cava.)

In general, operative management of these patients included the following: (1) coarctation repair and pulmonary artery banding in those patients with concomitant coarctation (40% in our series) before repair; (2) pulmonary artery banding before arterial switch or intraventricular repair early in the series, with tendency toward early repair (arterial switch or intraventricular repair) later in the series; and (3) intraventricular repair for side-by-side great artery orientation and arterial switch for more or less anteroposterior great artery orientation (although 6 patients had arterial switch for side-by-side great artery orientation).

	Results

Top Footnotes Abstract Introduction Material and Methods Results Comment References

 
Perioperative survival was 100% in the Kawashima group (4/4) and 93.8% in the arterial switch group (15/16); the death occurring early in the series was due to myocardial ischemia probably caused by coronary flow problems. There were three late deaths after arterial switch. One patient who had neonatal pulmonary artery band placement and coarctation repair had arterial switch and ventricular septal defect closure at 3.3 years of age and closure of a second ventricular septal defect at 4.5 years of age. He died of progressive pulmonary hypertension 4 years postoperatively (pulmonary vascular resistance = 9.6 U/m² and unresponsive to O₂ at catheterization 1 year before death). The second death occurred 4 years after arterial switch and 3.5 years after prosthetic (21-mm Medtronic-Hall) neoaortic valve replacement due to exsanguinating gastric ulcer with a therapeutic prothrombin time. This child had had two modified Blalock-Taussig shunts and underwent arterial switch at the age of 9 years. Acute lymphocytic leukemia developed in the third child, who died 3 years postoperatively (arterial switch at age 3 months).

Significant perioperative complications occurred in both the arterial switch and Kawashima groups. Mean postoperative length of stay in survivors after arterial switch (n = 15) was 16.47 ± 18.38 days. Without two outliers who required prolonged intravenous antibiotic therapy, mean postoperative length of stay was 9.92 ± 5.09 days. Mean postoperative length of stay after Kawashima operation was 16.5 ± 7.14 days, not significantly different from that of the arterial switch group. Mean follow-up in all patients is 4 years.

The outcome for each patient with the pertinent demographic data with regard to age and type of palliation and age at intracardiac repair are shown in Table 1. Patients 3, 4, and 5 were lost to follow-up 4, 5, and 0.17 years after intracardiac repair. The other patients have been followed up for 1 to 8 years (mean, 4 years). Six patients have had postoperative echocardiography only, and 13 patients have had postoperative echocardiography and cardiac catheterization. Significant findings in the postoperative period after arterial switch and ventricular septal defect closure included mild aortic insufficiency in 8/15 patients (53%), moderate in 1/15 (7%), and severe aortic insufficiency in 2/15 (13%). One of these patients underwent aortic valve replacement, and another underwent aortic valvuloplasty. Five patients (33%) had mild pulmonary stenosis, 1 (7%) had moderate pulmonary stenosis, and 2 (13%) had severe pulmonary stenosis after arterial switch. One patient underwent reoperation for supravalvular pulmonary stenosis and 1 had right ventricle-to-pulmonary artery conduit change. Three patients required reoperation for recoarctation 3 months and 2 years after neonatal subclavian flap aortoplasty and 6 months after aortic relocation. One patient required pacemaker insertion after arterial switch. One patient underwent mitral valvuloplasty 5 years after arterial switch. One patient in the arterial switch group (patient 1) and another in the Kawashima group (patient 18) required reoperation for an unrecognized second ventricular septal defect. One patient underwent reoperation for baffle stenosis after the Kawashima procedure. Two patients have moderate subaortic stenosis (25 and 36 mm Hg) after the Kawashima procedure. One of 4 patients in the Kawashima group has mild aortic insufficiency.

	Comment

Top Footnotes Abstract Introduction Material and Methods Results Comment References

Mean age in the intraventricular repair group was 2.25 years; that in the arterial switch group was 1.2 years. In our series the ages of the two groups were similar (1.4 and 1.5 years, respectively). The relatively ``older'' age of the patients in both groups is partly a result of prior pulmonary artery band placement. Interestingly, in light of early correction for other anomalies, few cases of neonatal Taussig-Bing repair have been reported. Besides the 2 in this series, Serraf and associates [16] reported 4 and Liddicoat and colleagues [23] documented 1. As surgeons progress more to neonatal repair of this lesion, arterial switch may have an advantage over the Kawashima intraventricular repair because of its applicability to a wider group of patients.

Pulmonary Artery Band Placement
Pulmonary artery banding has been extensively used as prior palliation in both the arterial switch and Kawashima groups (77% and 51%, respectively, in the combined series in Tables 2 and 3; 80% and 50% in our group). Under these circumstances, the fate of the pulmonary valve must be considered before choosing between arterial switch and intraventricular repair because neoaortic insufficiency has been associated with arterial switch after pulmonary artery banding. Two patients in our arterial switch group required reoperation for neoaortic insufficiency, whereas 9 others are being followed up with mild (8) and moderate (1) neoaortic insufficiency. For patients with side-by-side great arteries who have had pulmonary artery banding, we prefer the Kawashima intraventricular repair, which has the advantage of preserving the native aortic valve. For patients with anteroposterior great arteries, we still prefer arterial switch despite the possibility of neoaortic insufficiency which, if progressive, can be addressed in the future.

Associated Coarctation of the Aorta
Parr and coauthors [27] reported that 53% of 105 patients with Taussig-Bing had either a coarctation of the aorta or significant left ventricular outflow tract obstruction. Combining the two surgical series in Tables 2 and 3, 40% of the patients had associated coarctation repair. In the intraventricular repair group, only 1 patient had simultaneous intraventricular repair and coarctation of the aorta repair [21]. In the arterial switch group, Serraf and associates [16] reported 2 patients who had simultaneous arterial switch and coarctation of the aorta repair. Liddicoat and colleagues [23] recently reported aortic relocation, arterial switch, and ventricular septal defect closure in a 9-week-old infant with Taussig-Bing and interrupted aortic arch. In our series, 1 patient had simultaneous arterial switch and coarctation of the aorta repair with relocation of the aorta. If a child has the Taussig-Bing anomaly with coarctation of the aorta and primary neonatal repair is preferred, arterial switch will probably be the procedure of choice. If the child has coarctation repair and pulmonary artery band placement, later Kawashima or arterial switch operation will be feasible depending on factors already discussed.

Technical Factors
The Kawashima intraventricular repair has the advantage of preserving the native aortic valve and avoids coronary artery dissection [16]. However, we have found that a right ventriculotomy and subvalvular outflow tract patch are essential for adequate conal resection, intraventricular tunneling, and unobstructed right ventricle-to-pulmonary artery flow. Whether this approach can be equally successful in the neonate for the short and long term remains to be seen.

For arterial switch, the ventricular septal defect is directed from the left ventricle to the pulmonary artery (neoaorta). This can be done from the right atrium, pulmonary artery, or right ventricle. In the collected series, the right atrial approach was used 56% of the time, the pulmonary artery 25%, and the right ventricle 19%. We prefer in all cases to avoid a ventriculotomy and have used either the pulmonary artery (6/16) or right atrium (10/16) approach.

In the arterial switch group, a decision must be made whether or not to perform the Lecompte maneuver. In the collected series, 91% of the patients had a Lecompte performed. We used the Lecompte maneuver in 14/16 (87%) of our patients. Earlier it was thought that there might be some danger in performing the Lecompte maneuver for patients with side-by-side great arteries [28] because the position of the neoaorta might stretch and narrow the left pulmonary artery. Clearly, as experience has been gained this has not been borne out. It is necessary in some cases to take measures to compensate for the very large pulmonary artery in comparison with the smaller ascending aorta at the time of arterial switch.

Great Artery Anatomy
Since the issue was first addressed by Yacoub and Radley-Smith [11], many surgeons have thought that side-by-side great arteries are most suitable for intraventricular repair and anteroposterior great arteries are most suitable for arterial switch. In the combined series, 77% of patients who had intraventricular repair had side-by-side great arteries; 59% of patients who had arterial switch had anteroposterior great arteries. It has been noted that it is not necessarily the anteroposterior versus side-by-side anatomy that is important but rather the distance available between the tricuspid and pulmonary valves for an intraventricular tunnel [6]. Van Praagh [29] commented that the surgical significance is that when the great arteries are anteroposterior, the aortic valve is very anterior and the ventricular septal defect-to-aorta conduit (for 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.

Reoperation
One of the stark revelations that emerged from our review was the high postrepair reoperation rates in both groups. In the combined literature review, reoperation was required in 22% of patients after intraventricular repair and 30% of patients after arterial switch. The incidence of reoperation does not seem to be a factor in favor of either procedure. It does reflect the complexity of the repairs, with multiple anomalies requiring correction (ventricular septal defect, transposed great arteries, coarctation of the aorta, subaortic stenosis) and multiple structures at risk of injury (atrioventricular node, aortic valve, coronary arteries).

Mortality
The operative mortality in the combined series for intraventricular repair was 3% versus 13% for arterial switch. This is quite similar to our series (0% versus 6%). The late mortality in both series is 5% to 6%. In our series, all the late mortality occurred after arterial switch. Of the three late deaths in our series, one was clearly unrelated to the procedure-acute lymphocytic leukemia-and the other two could probably have been prevented by earlier repair rather than at 3.3 years of age (death due to pulmonary hypertension) and 9 years of age (required aortic valve replacement with warfarin therapy; gastrointestinal bleeding).

We conclude that for patients with Taussig-Bing anomaly the Kawashima intraventricular repair is attractive for patients with side-by-side great arteries, especially for those with unusual coronary artery anatomy or a pulmonary valve that is not considered adequate to function as a systemic valve. Intraventricular repair preserves the native aortic valve and avoids coronary dissection. These patients are at risk of the development of subaortic stenosis from the intracardiac left ventricle-to-aorta baffle. The arterial switch operation can be applied without ventriculotomy to all great artery relationships (anteroposterior as well as side-by-side arteries) and seems more suited to neonatal repair with or without concomitant coarctation repair. These patients are at risk of aortic insufficiency from prior pulmonary artery banding and use of the pulmonary valve as the neoaortic valve.

	Footnotes

Top Footnotes Abstract Introduction Material and Methods Results Comment References

 
Presented at the Forty-second Annual Meeting of the Southern Thoracic Surgical Association, San Antonio, TX, Nov 9-11, 1995.

Address reprint requests to Dr Mavroudis, Division of Cardiovascular-Thoracic Surgery, Children's Memorial Hospital, 2300 Children's Plaza, M/C #22, Chicago, IL 60614.

	References

Top Footnotes Abstract Introduction Material and Methods Results Comment References

 

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