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Eur J Cardiothorac Surg 1999;16:104-110
© 1999 Elsevier Science NL

Bi-directional cavopulmonary shunt: is accessory pulsatile flow, good or bad?

Department of Thoracic and Cardiovascular surgery, Laennec Hospital, Paris, France

Corresponding author. Nieuwe Hescheweg 104, NL 5342 EE Oss, The Netherlands. Tel.: +31-412-623213
e-mail: vandewal{at}wxs.nl,

	Abstract

Top Abstract 1. Introduction 2. Patients and methods 3. Statistical analysis 4. Results 5. Discussion 6. Conclusions Appendix A References

 
Objective: Evaluation of the effect and long-term outcome of accessory pulsatile blood flow versus classical bi-directional cavopulmonary connection (BCPC). Methods: Retrospective review of the medical and surgical records. Results: Two-hundred and five patients (119 boys, 86 girls) underwent BCPC from 1990 to 1996. Accessory pulsatile flow was present in 68%, flow being maintained through the pulmonary trunc in 46%, systemic-to-pulmonary artery shunt in 13% and mixed in 7%, or patent ductus arteriosus in 2%. Patients with accessory pulsatile flow had lower hospital mortality (3% versus 5%), while mean pulmonary artery pressure (14.1 versus 12.6 mmHg P=0.050) and increase of oxygen saturation (12.4 versus 8.7, P=0.034) were significantly higher. The period of artificial ventilation (1.9 day) and ICU stay (6 days) did not differ for both groups. Late mortality was higher following accessory pulsatile flow (6% versus 1%). At late follow-up patients with accessory pulsatile flow had significantly higher oxygen saturation (mean 85±4%, versus 79±4%; P0.005). If subsequent completion of Fontan is considered the optimal palliation and subsequent systemic to pulmonary artery shunt, arteriovenous fistula and transplantation is considered a failure, patients with accessory pulsatile flow had significantly more and earlier completion of the Fontan procedure (mean 1.7±2.4 years, versus 2.7±4.4 years; P=0.008). Survival is not influenced by age at bi-directional cavopulmonary shunt surgery, left or right functional ventricular anatomy or previous palliative surgery. One patient with accessory pulsatile flow developed systemic-to-pulmonary collateral's eventually requiring lobectomy. Conclusion: Despite two different initial palliative techniques the outcome was not significantly different. Accessory pulsatile blood flow appeared not to be a contra-indication for a completion Fontan procedure. Moreover, the data suggest that after accessory pulsatile flow can safely be performed, at late follow-up oxygen saturation is higher, while, significantly more and earlier completion of Fontan occurred. Age at bi-directional cavopulmonary shunt, basic left or right ventricular anatomy or previous palliative surgery did not influence survival.

Key Words: Functionally single ventricle • Glenn procedure • Pulsatile blood flow • Follow-up

	1. Introduction

Top Abstract 1. Introduction 2. Patients and methods 3. Statistical analysis 4. Results 5. Discussion 6. Conclusions Appendix A References

 
Bi-directional superior cavopulmonary connection (BCPC) is a widely used method of providing pulmonary blood flow in patients with complex congenital cardiac malformations characterised by univentricular atrio-venous connections [1,2,3]. Besides being used as an intermediate stage between systemic-to-pulmonary artery shunt or pulmonary artery band and Fontan procedure it has also been advocated as initial palliation in patients who have progressed beyond the neonatal period without the need for previous intervention [4,5].

Accessory pulsatile blood flow is defined as the presence of either a systemic-to-pulmonary artery shunt, a patent ductus arteriosus or a patent right ventricular outflow tract. Information on the use of accessory pulsatile blood flow in young infants is limited, few reports have addressed the issue of whether or not to leave an additional source of pulmonary blood flow at the time of BCPC [3,6,7]. Concerns remain about peri-operative morbidity and mortality and about post-operative systemic arterial oxygenation in very young patients [5,8].

We have therefore reviewed our experience and long-term outcome of BCPC with and without accessory pulsatile flow.

	2. Patients and methods

Top Abstract 1. Introduction 2. Patients and methods 3. Statistical analysis 4. Results 5. Discussion 6. Conclusions Appendix A References

 
2.1. Patients
This study is a retrospective review of our institutional experience with the BCPC procedure. From January 1990 to December 1996, 205 consecutive children underwent a BCPC procedure at Laennec Hospital. There were 119 boys and 86 girls, mean age 5.6 years (range, 26 days to 34.2 years). Five patients (2%) were <3 months, seven patients (3%) between 3 and 6 months and 19 patients (9%) were between 6 and 12 months of age at the time of BCPC (Fig. 1). Mean weight was 18.17 kg (range 3–88 kg). Basic morphology consisted of; univentricular left heart (n=128, 63%), quasi univentricular left heart (n=13, 6%), univentricular right heart (n=46, 23%), quasi univentricular right heart (n=9, 4%), double discordance (n=5, 2%) and criss-cross heart (n=4, 2%). In 49 children (25%) BCPC was the primary palliative procedure. The remaining 156 (75%) children had undergone a total of 209 palliative procedures prior to the BCPC. One-hundred and ten children underwent one previous palliative procedure, 40 children two, five children three and one child even four. The final palliative procedure before BCPC is presented in Table 1. Prior to the BCPC procedure pulmonary blood flow was from the following sources; antegrade from a systemic-to-pulmonary artery shunt (n=101, 50%), the ventricle (n=44, 21%), patent ductus arteriosus (n=4, 2%) or mixed sources (n=56, 27%).

View larger version (18K): [in this window] [in a new window]   Fig. 1. Age breakdown of patients undergoing classical BCPC (n=65, open bars) or BCPC with accessory pulsatile blood flow (n=140, filled bars).

Right ventricular morphology was defined as univentricular right heart and quasiuniventricular right heart. Left ventricular morphology was defined as anatomically univentricular left heart; quasiuniventricular left heart, Criss-cross heart and double discordance. Preoperative palliative procedures were classified as those which diminished pulmonary flow; i.e. pulmonary artery banding and those which improved pulmonary flow; i.e. systemic-to-pulmonary shunt, opening right ventricular outflow tract, Fontan procedure, Norwood 1 procedure, creation of an ASD. Accessory pulsatile blood flow was defined as the presence of flow being maintained through the pulmonary trunc, a systemic-to-pulmonary artery shunt, patent ductus arteriosus or a combination of those. BCPC was classified according to the presence or absence of accessory pulsatile blood flow. Operative death was defined as death that occurred within 30 days of operation or during the hospital stay. Follow-up was arbitrarily classified as optimal palliation if a completion Fontan procedure was done and failure if subsequent systemic-to-pulmonary artery shunt, arterio-venous fistula, lobectomy or transplantation was performed.

2.3. Surgical technique
All BCPC procedures were performed through a median sternotomy. In 12 children (6%) no cardiopulmonary bypass was used. In the remaining 193 children (92%) the cavopulmonary shunt was constructed with extra corporeal circulation. The mean bypass time was 68±52 min (range 25–390 min). Aortic cross clamping and cardioplegic arrest was used in 75 children (39%). A unilateral right cavopulmonary shunt was constructed in 160 children, a unilateral left cavopulmonary shunt in 20 children, and bilateral cavopulmonary shunts in all 25 children with bilateral superior caval veins. An extra source of pulmonary blood flow was present in 140 children (68%), either through the native pulmonary artery (n=62), banded pulmonary artery (n=28), systemic-to-pulmonary artery shunt (n=32), pulmonary artery together with a systemic-to-pulmonary shunt (n=14) or patent ductus arteriosus (n=4) (Table 3).

	3. Statistical analysis

Top Abstract 1. Introduction 2. Patients and methods 3. Statistical analysis 4. Results 5. Discussion 6. Conclusions Appendix A References

	4. Results

Top Abstract 1. Introduction 2. Patients and methods 3. Statistical analysis 4. Results 5. Discussion 6. Conclusions Appendix A References

 
4.1. Early results
There were eight operative deaths (4%), five following BCPC with accessory pulsatile blood flow compared to three without accessory pulsatile flow (not significant). Causes of operative death are presented in Table 5.

The period of post-operative ventilation (1.9 days) and length of ICU stay (6 days) did not differ in both groups.

4.2. Late results
One-hundred and fifty-three patients were available for late follow-up, mean follow-up was 2.3 years (range 3 days to 7.2 years). The remaining patients came from abroad and were lost for follow-up. Mean follow up was 1.9±1.6 years (range 35 days to 7.2 years).

During the follow-up period of those children who underwent BCPC with accessory pulsatile blood flow (n=95), 10 underwent completion of the Fontan procedure at a median of 1.7±2.4 years. Eight patients underwent reoperation to provide either an extra source of pulmonary blood supply (n=4), arterio-venous fistula (n=2), heart transplantation (n=1) or lobectomy (n=1). There were four late deaths, due to myocardial failure (n=2), cardiopulmonary arrest (n=1) and respiratory failure (n=1) (Table 5).

During the period of follow-up of those children who underwent a classical BCPC operation, 17 children underwent completion of Fontan procedure at a median of 2.7±4.4 years after shunting. Fifteen children underwent reoperation either for addition of an extra source of pulmonary blood flow (n=9), arterio-venous fistula (n=4), or heart transplantation (n=2). There was one late death due to myocardial failure. Late mortality between both groups was not significantly different.

The incidence of reoperation following BCPC surgery is not significantly different for both groups. However, if subsequent completion of Fontan is considered the optimal palliation and reoperation to accomplish systemic to pulmonary artery shunt, arteriovenous fistula and transplantation is considered a failure, patients with accessory pulsatile flow had significantly more and earlier completion of the Fontan procedure (1.7±2.4 versus 2.7±4.4 years; P=0.008).

Oxygen saturation at last follow-up was significantly higher in children with accessory pulsatile flow (mean 85±4%, versus 79±4%; P0.005). Whereas pre-operative saturation were not different (10.64±9.3 for children with accessory pulsatile flow versus 6.27±10.0 for children without pulsatile flow P=0.016).

Although long term survival is not influenced by age at BCPC surgery, left or right ventricular morphology or previous systemic to pulmonary artery shunt procedure the survival following BCPC with accessory pulsatile blood flow appeared to be significantly better if previous palliative surgery consisted of banding of the main pulmonary artery (P=0.0018).

One child following BCPC with accessory pulsatile blood flow developed systemic-to-pulmonary artery collateral's eventually requiring lobectomy.

Actuarial analysis for patients undergoing BCPC with and without accessory pulsatile blood flow was not significantly different, 1-, 2- and 3-year survival rates following BCPC with accessory pulsatile flow 91%, 88% and 88%, versus 95%, 92% and 92% without accessory pulsatile blood flow. During the follow-up interval no further deaths were recorded.

	5. Discussion

Top Abstract 1. Introduction 2. Patients and methods 3. Statistical analysis 4. Results 5. Discussion 6. Conclusions Appendix A References

 
BCPC was initially considered free of the problems associated with the classical Glenn anastomosis. However, systemic-to-pulmonary arterial collateral's [9], pulmonary arteriovenous fistulas [10] and systemic venous collateral's [11] have been found to develop more frequently than in other settings of congenital heart disease. The addition of a source of pulsatile flow to the pulmonary blood flow has been proposed as a potential means of mitigating some of these developments.

The concept of pulsatile cavopulmonary anastomosis was introduced in 1971 by Furose [12] in an animal experiment. In 1972 Glenn and Fenn [13] recommended creation of an artriovenous fistula between the axillary artery and vein to augment pulmonary flow after a failing cavopulmonary anastomosis. Although the cyanosis was much improved, the patient developed pulmonary hypertension. In 1989 Billingsley [14] reported BCPC and accessory pulsatile blood flow in the definitive biventricular repair of pulmonary atresia and intact ventricular septum who had augmentation of the right ventricular outflow tract and pulmonary artery with a transannular patch. In 1991 Kobayashi [15] reported the use of BCPC and accessory pulsatile blood flow in children who were not suitable for the Fontan procedure.

Well-controlled accessory flow to the pulmonary arteries has proven to be an option when a BCPC procedure is performed. Physiologically, there are differences between patients with and without accessory pulsatile blood flow, with the quantity of the pulmonary flow and the volume load on the ventricle varying in accordance with the amount of additional blood supplied to the pulmonary arteries. The surgeon must be aware of the potential beneficial influence on pulmonary vascular development and deleterious impact by imposing a volume load on the ventricles [9]. If the additional source of pulmonary blood flow is not carefully controlled, continued pulmonary recirculation and ventricular dysfunction secondary to a chronic volume overload may result, which has been shown to increase morbidity and mortality after the Fontan operation [16,17].

This study potentially represents one of the largest single institutional studies of patients undergoing BCPC. With rare exceptions [1,5,7,18,19], most reports have comprised fewer than 30 patients. We have paid particular attention to the influence of accessory pulsatile blood flow on the early and medium term results of BCPC.

5.1. Influence of accessory pulsatile blood flow on early outcome of BCPC
The main goal of BCPC is to provide perfusion to both lungs while avoiding an excessive increase in systemic venous pressure. One of the factors, which can affect the clinical outcome of the surgically reconstructed circulation, is the amount of pulsatile blood flow coming from the main pulmonary artery [9]. The hemodynamics in the pulmonary arteries following BCPC with accessory pulsatile blood flow can vary from case to case depending on associated problems. Under ideal circumstances, the flow in the entire pulmonary artery system will be pulsatile. The site of translocation from venous to arterial pressure in the superior vena cava will depend on the extent of systolic reflux from the pulmonary artery. The results, however, show that hemodynamics in the pulmonary arteries are greatly influenced by the amount of flow through the native main pulmonary artery and that the flow from the superior vena cava allows an equal distribution of the blood to both lungs, with a small predilection for the left side [20]. Non-pulsatile flow from BCPC is mainly directed to the ipsilateral lung, whereas pulsatile flow is directed to the contralateral lung [6]. Total perfusion of the ipsilateral lung is less than the perfusion of the contralateral lung [21]. In tight pulmonary artery stenosis (>75%), pulsatile forward flow is primarily directed to the left pulmonary artery, with little influence on superior vena caval pressure and the right pulmonary artery [22]. De Leval [22] also found that pulsatile forward flows corresponding to 15, 30, 45 and 60% of the systemic artery output increased the mean pulmonary artery and superior vena caval pressures by 1, 1.7, 2.4 and 3.6 mmHg, respectively.

Despite the two different policies we have demonstrated that accessory pulsatile blood flow:

is well tolerated after BCPC and in fact appears to have a small advantage in terms of early post-operative mortality,

mean pulmonary artery pressure and oxygen saturation were significantly higher,

no pulmonary hypertension developed,

appeared not to be a contra-indication for completion of the Fontan procedure.

It is even suggested that if a patient following BCPC has difficulty in weaning from bypass accessory pulsatile flow might be beneficial.

5.2. Chylothorax
Effusions are one of the principal causes of extended hospital stay after BCPC, occurring in approximately 10% [23]. Frommelt [3] concludes that patients with an additional source of pulmonary blood flow after BCPC have a higher postoperative central venous pressure, higher oxygen saturations and are at risk for the late development of a chylothorax. We have been unable to verify this observation in our own cohort.

5.3. Mortality
The reported mortality for BCPC rages from 0 to 33% [1,4,7,15,18,19]. Interpretation is complicated by the different eras of surgery, patient populations and surgical techniques. The mortality in the present series (3 and 5%).

5.4. Influence of accessory pulsatile blood flow on late outcome of BCPC
Children with accessory pulsatile flow showed higher late mortality. Late systemic O₂ saturation was significantly higher when compared to classical BCPC anastomosis. If subsequent Fontan procedure is the optimal palliation and a systemic-to-pulmonary artery shunt, arterio-venous fistula or transplantation is considered as failure, patients with accessory pulsatile flow have a higher rate at an earlier age of completion Fontan procedure. The Fontan procedure was not performed electively, despite the fact that in the group of BCPC with accessory pulsatile blood flow, pulmonary vascular resistance might have become too high. The criteria to proceed with a Fontan procedure were: decreasing exercise capacity or increasing cyanosis due to insufficient pulmonary flow, with low pulmonary vascular resistance.

Previous reports [6,7,15] have focused on the hemodynamic and clinical effects of BCPC associated with accessory pulsatile flow. Muster [6] demonstrated that BCPC may be successfully used to accomplish the so-called ‘one and one-half repair’. Kobayashi [15] described 10 high risk Fontan candidates who underwent BCPC with accessory pulsatile flow. Webber [7] in a multi-institutional study described 108 patients and concluded that accessory pulsatile flow is well tolerated in the short and medium term and that early post-operative saturation improved.

Our data show that long term survival following BCPC with accessory pulsatile flow is significantly better if previous palliative surgery had included banding of the main pulmonary artery. Although Reddy [5] reported age less than 2 months at time of BCPC as a significant predictor of poorer survival. We have been unable to verify the other observations in other reports.

5.5. Consequences for the pulmonary circulation
The haemodynamic consequences of BCPC on the pulmonary circulation are less well understood. Several authors have drawn attention to the potential benefits of leaving pulsatile flow in the pulmonary arteries after BCPC [6,8,15]. Kobayashi [15] emphasised the possible beneficial effects of accessory pulsatile flow in preventing late development of pulmonary arterio-venous malformations.

Although there are many reports of arteriovenous malformations after classical Glenn shunt [24,25], there are only two reported cases of pulmonary arteriovenous malformations after the Fontan operation [26]. Possible causes of pulmonary arteriovenous malformations after Glenn procedures;

time: the Glenn shunt has been used for more than 30 years, whereas the Fontan operation has been widely applied for only the past 10 years.

‘hepatic factor’: perhaps the absence of some important interaction between a hepatic factor and lung blood vessels induces formation of arteriovenous malformations after the Fontan operation.

If a ‘hepatic factor’ [8] is important for the prevention of pulmonary arteriovenous malformations this might represent an advantage of BCPC with accessory pulsatile flow. However in one of our patients with BCPC with accessory pulsatile flow pulmonary arteriovenous malformations developed, necessitating lobectomy. To our knowledge we are the first to report this complication following BCPC with accessory pulsatile flow. In addition pulmonary blood flow has been advocated as a means of promoting pulmonary arterial growth. There are few published data to support or disapprove these assumptions [10].

	6. Conclusions

Top Abstract 1. Introduction 2. Patients and methods 3. Statistical analysis 4. Results 5. Discussion 6. Conclusions Appendix A References

 
Although, there now seems to be consensus that pulsatility is not useful to improve the operative mortality of the Fontan operation and there is widespread speculation that absence of pulsatility may contribute to the late deleterious effects of this procedure. Our data suggest that BCPC with accessory pulsatile flow compared to classical BCPC can:

be safely performed;

gives at late follow-up higher oxygen saturation's;

does not develop pulmonary hypertension cq is no contra-indication for Fontan

have more and earlier completion of Fontan procedure.

	Acknowledgments

 
The authors would like to thank Steven Chamuleau for his assistance with the statistical analysis and Stephen R. Woolley FRCS for his help in preparing the manuscript.

	Footnotes

 
Presented at the 12th Annual Meeting of the European Association of Cardio-thoracic Surgery, Brussels, Belgium, September 20–23, 1998.

	Appendix A

Top Abstract 1. Introduction 2. Patients and methods 3. Statistical analysis 4. Results 5. Discussion 6. Conclusions Appendix A References

 
Conference discussion
Dr J.A. van Son (Leipzig, Germany): In our experience and that of others bi-directional cavopulmonary anastomosis with an additional source of pulmonary blood flow has a higher post-operative morbidity rate than bi-directional cavopulmonary anastomosis without accessory blood flow. In the majority of your patients the cavopulmonary anastomosis was performed beyond 1 year of age, which I think is rather late in terms of avoidance of volume overloading of the single ventricle and its related detrimental sequelae. Please comment on the concept of performing the bi-directional cavopulmonary anastomosis before 6 months of age.

Dr van de Wal: We have not been able to show in our retrospective analysis that age is a significant factor, indeed although theoretically the presence of an accessory pulmonary blood flow might increase the potential for volume overload, in practice our experience does not confirm this. As regards the possibility of performing the bidirectional cavopulmonary anastomosis before 6 months of age I think we should be guided by the experience in other congenital defects where clearly as our own expertise increases so the age of definitive operation tends to decrease.

Dr S. Conte (Leuven, Belgium): You used bi-directional Glenn and accessory flow both as intermediate stage to Fontan and as end-stage alternative to Fontan. Assuming from the data that you presented that this procedure is better than bi-directional Glenn alone as an end-stage, do you have comparative hemodynamic or clinical data in patients after Fontan completion to support bi-directional Glenn and accessory flow also as intermediate stage? And did you never have to renounce to Fontan completion because of the accessory flow?

Dr van de Wal: No. The Fontan operation was the end-stage of measurement. So we followed up those patients who had a bi-directional Glenn, with or without accessory flow, and measured the period until last clinic follow-up or death or completion of Fontan operation, as the end point for this study, therefore, I can't answer the question of what happened after their Fontan operation.

Dr S. Conte: And did you have to renounce Fontan completion because of the accessory flow in any patient?

Dr van de Wal: No.

Dr H. Jalali (Queensland, Australia): I studied similar patients like yours, and we had 140 patients with bi-directional Glenn and maintenance of some forward flow. The results were presented last year in Sydney, with very similar conclusions to yours in regard to better oxygenation and not compromising the long term. But there were 12 patients out of 140, which is nearly 10%, which failed at the time of the primary operation. And what I call failure was things such as highly elevated and pulsatile central venous pressure beyond 20–25. Did you have any of these early failures in your experience?

Dr van de Wal: We have not been able to show in our retrospective analysis that age is a significant factor, indeed although theoretically the presence of an accessory pulmonary blood flow might increase the potential for volume overload, in practice our experience does not confirm this. As regards the possibility of performing the bidirectional cavopulmonary anastomosis before 6 months of age I think we should be guided by the experience in other congenital defects where clearly as our own expertise increases so the age of definitive operation tends to decrease.

Dr Jalali: But you didn't take anyone back to suppress the accessory flow?

Dr van de Wal: Not that I am aware of, no.

	References

Top Abstract 1. Introduction 2. Patients and methods 3. Statistical analysis 4. Results 5. Discussion 6. Conclusions Appendix A References

 

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