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Eur J Cardiothorac Surg 2007;31:229-235. doi:10.1016/j.ejcts.2006.11.034
Copyright © 2007, European Association for Cardio-Thoracic Surgery. Published by Elsevier B.V. All rights reserved
Department of Paediatric Cardiac Surgery, Diana, Princess of Wales Children's Hospital, Steelhouse Lane, Birmingham B4 6NH, United Kingdom
Received 21 August 2006; received in revised form 13 November 2006; accepted 20 November 2006.
* Corresponding author. Tel.: +44 121 333 9435; fax: +44 121 333 9441. (Email: william.brawn{at}bch.nhs.uk).
| Abstract |
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Key Words: TaussigBing Arterial switch Surgical strategy
| 1. Introduction |
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The TaussigBing anomaly is the second most common form of DORV after the Fallot-type, and in some series an incidence of more than 20% amongst DORV population has been reported [2]. Malalignment of the infudibular septum causes preferential flow from left ventricle to the pulmonary artery. Therefore aortic arch abnormalities are often associated and coarctation and/or hypoplasia of the aortic arch are found in more than 50% of cases [3,4]. Furthermore, different relationship of the great arteries and anatomical variations of the coronary arteries have also been described. Presentation of these infants is normally characterized by congestive cardiac failure and pulmonary hypertension. Some patients require ventilation and treatment with prostaglandins pre-operatively.
Many types of repairs have been proposed. Intra-ventricular re-routing with resection of the infundibular septum described by Kawashima and colleagues [5,6] was recommended for TaussigBing anomaly with side-by-side great arteries [7]. Anatomic correction with arterial switch operation and closure of ventricular septal defect (ASO + VSDc) has been applied in many of reported series with fairly consistent results [8,9]. For more complex form of TaussigBing anomaly, staged establishment of Fontan circulation has also been proposed. Controversy remains as to whether intra-ventricular repair or arterial switch is the preferred technique and also whether complex anatomies should be treated with a staged approach or single-stage complete repair.
This institution has historically adopted the arterial switch technique (ASO + VSDc) as procedure of choice since 1988 with the use of a staged approach in complex cases. This paper reviews our experience with this protocol and compares outcomes with other techniques.
| 2. Patients and methods |
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There were 25 males (76%) and eight female. The median age at ASO + VSDc was 39 days (range 1753 days) and median weight was 3.5 kg (range 2.611.8 kg).
The pre-operative status of these children was characteristically heterogeneic. Fifteen patients required intensive care resuscitation involving prostaglandin infusion (PGE1, n = 10), intubation and ventilation (n = 9) and/or inotropic support (n = 3). In addition, one child required prolonged cardiopulmonary bypass for 24 h following palliative surgery (see below). Fourteen patients were in congestive heart failure controlled with diuretics and four patients were moderately cyanosed (SatnO2 <85%). Amongst all patients, 14 had emergency balloon atrial septostomy (BAS) at time of admission.
The relationship of the great arteries was antero-posterior in 19 patients (Group I) and side-by-side in 14 patients (Group II). Coronary anatomy was recorded based on Yacoub's classification [10]. Types A and D were almost exclusive in group I (95%) whereas types D and E were predominant in group II (64%). According to the Leiden convention, the arrangement 1LCx 2R was the most common in group I, whereas in Group II the arrangements 1LR 2Cx and 1L 2RCx were equally encountered (Table 1 ). The VSD was perimembranous in 19 patients (58%), muscular in 11 (33%) and three children (9.1%) had multiple ventricular septal defects.
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| 3. Operation |
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In ASO + VSDc, the heart was exposed via a midline sternotomy incision and an autologous pericardial patch was harvested and kept moist in a cold saline swab. The aorta, main pulmonary artery, and left and right pulmonary arteries were dissected carefully and encircled with silastic tapes. In cases requiring repair of aortic arch or coarctation, the aortic arch, proximal descending aorta and head vessels were dissected as well.
A single right atrial cannula was used in small infants (<3 kg) and bicaval venous cannulation for all other cases. When a single right atrial cannula was employed, the nasopharyngeal temperature was lowered to 18 °C, and periods of circulatory arrest were used for closure of the VSD and atrial septal defect (ASD) or atrial septostomy. When bicaval cannulation was used, the nasopharyngeal temperature was normally lowered to between 22 and 25 °C. Alphastat pH strategy was employed during CPB.
The VSD was routinely exposed through the right atrium. In difficult cases, the VSD was also approached through the right ventricle or pulmonary artery. The VSD was patched with interrupted pledgeted prolene sutures, and a double velour Dacron patch. At the same time, the subaortic region was carefully inspected and, when necessary, muscle resection performed to ensure unobstructed passage of blood from the left ventricle through the VSD into the neo-aorta. The associated repair of hypoplastic aortic arch or coarctation was performed under deep hypothermic circulatory arrest (18 °C) or half flow bypass with the arterial cannula pushed into the innominate artery.
After the repair of hypoplastic arch, the vascular clamp and arterial cannula were then repositioned so that the aortic arch and the descending aorta were re-perfused. The aorta was transected 35 mm above the commisures, and the main pulmonary artery was then transected at the same level. The coronary arteries were excised with a generous cuff of aortic sinuses and the defects in the old aorta were repaired with autologous pericardium as a single patch. The coronary artery buttons were mobilized and sutured to medially hinged trap-door incisions in the pulmonary artery. The Lecompte manoeuvre was then performed to move the distal aorta posterior to the pulmonary artery and it was accomplished in all patients of this series regardless of the arrangement of the great vessels and we did not experienced any coronary compression.
The neo-aorta was then reconstructed, the heart was reperfused and the right ventricular outflow tract was completed during re-warming with the heart beating. Position of the neo-pulmonary artery was decided according to the best geometry achievable for the neo RVOT, if necessary this meant shifting the opening in the pulmonary artery bifurcation across to the right side if this was the most natural position. The principle was to allow the neo-pulmonary artery to lie in its most natural position, without tension and in our experience that has always required the Lecompte manoeuvre.
One patient required the placement of a right ventricle-to-pulmonary artery (RV-PA) conduit (Hancock® Bioprosthetic Valved Conduits) due to abnormal course of right coronary artery. Cardio-pulmonary bypass was discontinued with inotropic support, commonly in the form of dobutamine and adrenaline with sodium nitroprusside as vasodilatator. Left atrial line was routinely placed.
The chest was closed formally in presence of stable hemodynamics and if the heart was not distended. In our experience, the chest was left open in 15 patients (45.5%). In these children, the skin only was closed or a GoreTex patch (W.L. Gore & Associates Inc., AZ, US) was used for temporary closure. Subsequently, the chest was closed on the intensive care unit within 96 h (median time, 48 h; range, 1896).
Associated aortic coarctation and hypoplastic arch were repaired with extended end-to-end anastomosis technique in 10 cases, one with end-to-side anastomosis repair, and one with subclavian flap technique. Pulmonary homograft patch was used to repair the arch and neo-aorta in five cases and bovine pericardial patch in one patient. One child required a complex reconstruction repair involving an extended end-to-side anastomosis augmented by a pulmonary homograft patch. Isolated coarctation was treated by resection and extended end-to-end anastomosis. However, most cases had associated arch hypoplasia in which case repair was effected with resection of the coarctation ridge and homograft patch augmentation of the underside of the arch. The median duration of cardiopulmonary bypass, aortic cross-clamp and deep hypothermic circulatory arrest was 147 min (range 95388 min), 112 min (range 84164 min) and 20 min (range 0121 min), respectively. The patients with previous palliative procedure had shorter cumulative period of DHCA (14 min (range, 035 min) vs 32 min (range, 0121 min), p = 0.04), although the cardiopulmonary bypass and aortic cross clamp times were similar (145 min (range, 95388 min) vs 154 min (range, 98195 min), p = 0.67; 112 min (range, 84164 min) vs 115 min (range, 84135 min, p = 0.45). Details of ASO + VSDc are reported in Table 3. Follow-up was complete by our institution and local hospitals with echocardiography and angiogram as indicated.
| 4. Data analysis |
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Data have been examined by analysis of variance using a commercial statistical software package (SPSS for Windows, version 12; SPSS Inc., Chicago, IL). Continuous variables are expressed as mean ± standard deviations (SD) or medians (range). Comparative univariable analyses were carried out using the T-test, the MannWhitney U-test or the Wilcoxon signed-rank test. Binomial or ordinal data are expressed as percentages, and comparative univariable analyses were conducted with the
2 test, the two-sided Fisher exact test, or binomial logistic regression, as appropriate.
Actuarial survival, freedom from re-operation and freedom from catheter-based re-intervention were estimated by using the KaplanMeier product limit method. These results have been expressed as a probability estimate ±1SEM. The influence of surgical strategies on these actuarial outcome measures have been made with the log-rank test.
| 5. Results |
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The median ventilation time was 48 h (range, 12360 h), intensive care unit stay 3 days (range, 156 days) and hospital stay 10 days (range, 656 days) (Table 3).
Fourteen patients (42.4%) suffered one or more post-operative complications including low cardiac output (n = 3); renal impairment requiring peritoneal dialysis (n = 3); bleeding necessitating re-exploration (n = 1); phrenic nerve palsy requiring hemidiaphragm plication (n = 2); seizures (n = 2); pleural effusion or chylothorax (n = 4); and sepsis (n = 2). In addition two patients developed supraventricular tachyarrhythmia, and one patient had complete heart block requiring a permanent pacemaker (PPM) insertion. All of the patients, except the patient with a PPM, are currently in sinus rhythm (Table 4 ).
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Four patients needed late catheter based re-interventions. They were balloon angioplasty of recurrent aortic coarctation (n = 1), of stenotic pulmonary valve (n = 1), of RPA stenosis (n = 1), and multiple balloon angioplasties with stenting of LPA stenosis (n = 1). Freedom from re-intervention was 79 ± 9% at 5 and 10 years (Fig. 1).
The freedom from recurrent RVOTO and branch pulmonary artery stenosis was 88% ± 7 at 5 years and 81 ± 9% at 10 years. The freedom from recurrent subaortic stenosis and coarctation was 97% ± 3 at 1 year and 90 ± 7 at 5 and 10 years (Fig. 2 ).
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By contrast, side by side great vessels, subaortic obstruction and aortic arch abnormalities increase the risk of surgical re-operation or catheter-based re-intervention in our series (p = 0.003, 0.05 and 0.002, respectively).
The influence of the coronary artery anatomy on the risk of surgical re-operation or catheter-based re-intervention cannot be statistically determined, primarily because of the rarity of certain variants and the mortality attributed to this anatomy. However, type B had three patients of three requiring re-operation and re-intervention and type D had two of five and E had three of 10. By contrast, no patient of type A group (total = 14) and type C group (total = 1) had re-operation or re-intervention.
| 6. Discussion |
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Initial palliation with pulmonary artery banding, correction of aortic obstruction and subaortic stenosis with delayed definitive repair has emerged as a possible option. This was performed in our center when there were VSD anatomy and position or associated lesions that made repair unfavourable. Furthermore, initial palliation has also allowed patients to be referred to specialist centers experienced with the management and anatomical correction of TaussigBing anomaly. Two of the patients in our series were palliated elsewhere before being referred for definitive surgery. All patients who underwent initial banding successfully underwent subsequent anatomical repair and this group had excellent long-term outcomes with no patient requiring subsequent re-intervention. An explanation for this may be that the delay between the two stages allows for a period of growth of the great vessels and also allows for reassessment of the outflow tracts such that further muscle resection or patch augmentation can be performed at time of definitive repair. However, the band was not always well tolerated and one case needed to be resuscitated with cardiopulmonary bypass and proceeded to emergency anatomical correction.
Nevertheless, with increasing experience and improved results in neonatal arterial switch, one-stage complete repair is being advocated for all TaussigBing anomalies [12]. Disadvantages of two-stage repair include a second operation, which may be difficult and hazardous because of the adhesions and distortion of already abnormal anatomy, increased risk of pulmonary vascular disease, a pressure and volume loaded right ventricle, adverse effect of persistent cyanosis and congestive cardiac failure. The long-term outcome of the neo-aortic valve after PA banding may be less favourable with increased incidence of aortic regurgitation or subaortic stenosis (although we have not shown this in this series). However, one-stage complete repair is a complex undertaking in these neonates and the reported mortality varies between different series [1618]. The presence of aortic arch obstruction had been shown not to influence the outcome in term of survival or risk of re-operation [3,12].
In our series, all four deaths were amongst children who had single stage repair, but this finding is not statistically significant and the results suggest that the single stage approach can be performed with acceptable morbidity and mortality. Long-term survival after initial post-operative recovery is excellent although there is a significant risk of re-intervention. The risk of recurrent RVOT obstruction and pulmonary artery stenosis has been reported to be as high as 57% after 5 years [12]. Our series showed a lower rate of 13.8% at a mean follow-up of 6.2 years. There is also a lower but significant risk of recurrent subaortic stenosis and coarctation of 6.9%.
In summary, the arterial switch operation can be successfully applied to all patients with TaussigBing anomaly. Coronary artery patterns, position of the great vessels and aortic arch problems are not a contraindication to this approach and all anatomical variants are amenable to repair. There is no clear advantage or disadvantage from a staged approach, which would suggest that a policy of single stage repair is preferable. However, the decision on whether to opt for single or two-stage repair can be a difficult one. We would recommend as a general concept that we would aim for single stage repair. We would not regard weight as a specific contraindication to single stage repair and this is borne out by the results. Neither would we consider coronary pattern a reason not to proceed with one stage repair. However, we would consider a two-stage repair in cases of more complex anatomy, i.e. (1) concern over adequate size of the right ventricle or possible straddling of the AV valve (this will allow a period of time to reassess the RV and its growth), or (2) multiple VSDs or combination of arch hypoplasia with a VSD that cannot be accessed easily and might require ventriculotomy to repair. Long-term data will be necessary to establish this.
| Appendix A |
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Dr E. Belli (Marseilles, France): When do you plan biventricular repair in case of preoperatively stable and without significant aortic arch obstruction patients? I mean, first week or you wait second or third week of life?
Dr Griselli: Well, in a routine transposition after balloon atrial septostomy, the normal time is within 2 weeks the switch is performed. This is an ideal situation in a simple transposition.
In these other cases, it depends on the clinical situation, the anatomy. That's why the two-stage and one-stage repair has been designed, particularly looking at the coronary anatomy and the position of the VSD, more than the arch abnormalities.
Dr Belli: Yes, but without arch anomaly, with a complex intracardiac anomaly and side-by-side vessel, what do you plan? Do you wait that the patient becomes more mature?
Dr Griselli: Well, ideally, if the patient is stable, yes, of course, there is a tendency to wait. Basically the preoperative wait is very important, as you know, so we wait a bit longer. But, of course, it depends on the clinical situation. Yes, up to 6 weeks we have been waiting for the switch.
Dr B. Maruszewski (Warsaw, Poland): I just would like to share our experience. I think the results are very good, but I personally cant see any reason why we should postpone TaussigBing repair and to treat it differently to the regular transposition of the great arteries.
In our experience, the mortality is not significantly higher. We treat them exactly as we do for a simple transposition. And I think that the discrepancy between the great arteries, when they are a couple of days old, is smaller, and I think that the right ventricular outflow tract increases with the time. So actually I think this surgery is simpler if you treat them exactly as you do for the simple transposition of great arteries.
Dr Griselli: And I think you are absolutely right. In looking at the literature, it's been reported always a constant discrepancy between the size of the pulmonary artery and the aorta.
I dont think this was a problem in our series. We havent looked at this, to be honest, at the discrepancy. It has been looked at in previous literature. And it hasnt been a factor, which has basically made us to operate early or late, or later than normally we do in our practice.
Dr J. Comas (Madrid, Spain): I think the major difference between the classical switch and the normal transposition and switch in TaussigBing is the incidence of re-operations. You could describe what youre planning to do to reduce this incidence that is quite high?
Dr Griselli: I think, to be honest, the incidence hasnt been so high as in other series. I think we had basically, in total, six children who required re-operation and re-intervention in total. Particularly what we discovered is a common complication is right ventricular outflow tract.
And consider that one of these children also had right ventricle artery conduit, as necessarily to be changes on the stage. I dont think I have had five children of the series was very high. But it is a recognized complication we reported.
And then we have treated, particularly in side-by-side great arteries, the position of the anatomy sometimes is difficult to establish, particularly because there is not enough space to reconstruct the right ventricular outflow tract. So it's an expected complication. I think in this series it is not very high as in other series, to be honest.
| Footnotes |
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| References |
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This article has been cited by other articles:
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K. Agematsu, M. Aoki, Y. Naito, and T. Fujiwara Surgical Palliation for Taussig-Bing Anomaly with Multiple Lesions Asian Cardiovasc Thorac Ann, October 1, 2008; 16(5): 412 - 413. [Abstract] [Full Text] [PDF] |
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B. Alsoufi, S. Cai, W. G. Williams, J. G. Coles, C. A. Caldarone, A. M. Redington, and G. S. Van Arsdell Improved results with single-stage total correction of Taussig-Bing anomaly Eur. J. Cardiothorac. Surg., February 1, 2008; 33(2): 244 - 250. [Abstract] [Full Text] [PDF] |
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