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Eur J Cardiothorac Surg 1998;13:514-519
© 1998 Elsevier Science NL

Bi-directional cavopulmonary shunt associated with ventriculo and valvuloplasty in Ebstein's anomaly: benefits in high risk patients¹

Department of Cardiovascular Surgery, Hospital Broussais, 96, rue Didot, 75014 Paris, France

Received 8 December 1997; received in revised form 9 February 1998; accepted 16 February 1998.

Corresponding author. Hôpital Broussais, Service de Chirurgie Cardiovasculaire, 96, rue Didot, 75014 Paris, France. Tel.: +33 1 43959346; fax: +33 1 43959342.

	Abstract

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Objective: The prognosis for surgical repair of Ebstein's anomaly depends on the tricuspid valve repair and on the right ventricular function. In order to decrease the preload of the compromised right ventricle, a bi-directional cavopulmonary shunt was added to the intracardiac repair. Methods: Among 113 patients operated on for Ebstein' s anomaly at our institution from 1980–1997, a cohort of 60 patients was selected for high risk for postoperative complications. Patients presented with one or more several criteria: massive tricuspid valve dysfunction, extended atrialized right ventricle, poor right ventricular contractility, or long standing atrial fibrillation. After prior informed consent, this cohort was divided into two groups. Both groups had similar preoperative clinical patterns: Group I (45 patients), surgical technique included longitudinal right ventricular plication and tricuspid valve valvuloplasty; Group II (15 patients), where the surgical technique was similar to Group I except a bi-directional cavopulmonary shunt was added at the end of the procedure. Results: Operative mortality was 24% (11/45) in Group I and 0% (0/15) in Group II (P<0.05). The survival at 5 years was 66.1±14% in Group I and 80±16% in Group II (not significant). Reoperation rate was 11% (5/45) in Group I and 0% (0/15) in Group II. No deleterious effects of the bi-directional cavopulmonary shunts were observed clinically. Residual tricuspid valve insufficiency rate was 26% in both groups. However, patients with the bi-directional cavopulmonary shunt had a better tolerance and have not needed reoperations to date. Conclusions: In high risk patients with Ebstein's anomaly, an associated bi-directional cavopulmonary shunt seems to offer several distinct advantages including decreased operative mortality and better tolerance of the residual tricuspid valve dysfunction.

Key Words: Ebstein's anomaly • Congenital tricuspid valve insufficiency • Right ventricular failure • Bi-directional cavopulmonary shunt

	Introduction

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Classically, Ebstein's anomaly is considered a downward displacement of the septal and postero-inferior leaflets of the tricuspid valve (TV) within the inlet of the right ventricle (RV) [1] [2]. From an anatomic standpoint, four features are invariably present: dilatation of the tricuspid valve annulus, downward displacement of the hingepoint of the septal and postero-inferior leaflets toward the apex of the right ventricle, and restricted motion of the antero-superior leaflet. The spectrum of variation is wide from one patient to the other [3]. Techniques of conservative surgery deal with the anatomic and functional anomalies [4]. A key point in the surgical results appears to be the right ventricular contractility [5]. The wall of the functional ventricle is often thin and this fact is seen in the clinical evolution in neonates [6] [7]. Poor right ventricular contractility is not only of congenital origin, but also may be due to the long-term evolution of the tricuspid valve insufficiency [8] [9]. The aim of this study was to assess the effect of decreasing the preload of the right ventricle in association with an intracardiac repair in patients with a high risk of postoperative right ventricle failure.

	Methods

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From 1980 to 1997, 113 patients underwent intracardiac surgery for Ebstein's anomaly at our institution. The overall operative mortality was 10% (12/113). Among them, five died of early postoperative RV failure. The clinical characteristics seen in this fatal outcome were acute dilatation of an akinetic right ventricle, recurrent TV insufficiency (due to dilatation of the right ventricle), renal failure and normal contractility of an empty left ventricle. Death occurred within 24–48 h. Two additional patients had severe rhythm disturbances eventually leading to the previously described situation. This complication led us, in 1992, to add a bi-directional cavopulmonary shunt in order to decrease the preload in patients with a potentially compromised right ventricle. We compared two groups of patients with similar clinical features that were chosen as high risk patients.

Criteria for selection in this study was the presence of one or more several features: massive tricuspid valve insufficiency (with hepatomegaly and/or ascites), extended atrialized right ventricle, poor right ventricular contractility based on echocardiography and/or direct visual assessment during surgery, or long standing atrial fibrillation. Sixty patients were selected for the study after prior informed consent. Two groups were compared:

Group I: from 1980–1991, 45 patients were included. The mean age was 22±14 years (range, 4–55). The surgical technique was intracardiac only with tricuspid valvuloplasty and longitudinal plication of the RV. It will be detailed later.

Group II: from 1992–1997, 15 patients were included. The mean age was 25±15 years (range, 5–65). The surgical technique included intracardiac repair and bi-directional cavopulmonary shunt at the end of the cardiac repair.

Demographically, there were no differences between the two groups regarding age, the preoperative clinical condition and associated anomalies (Table 1). The most frequently associated anomaly was ostium secundum and patent foramen ovale (65%) which was present in both groups. Others anomalies were: in Group I, mitral valve insufficiency (1), ventricular septal defect (1), previous Blalock–Taussig shunts (2) and sub-aortic stenosis (1); and in Group II, patent ductus (1) and pulmonary artery hypoplasia (1). Functional Type according to Carpentier's Classification is indicated in Fig. 1 .

View larger version (25K): [in this window] [in a new window]   Fig. 1. Types of functional anomalies according to Carpentier classification. The main characteristics are as follows. Type A, minimal displacement of the attachment of the septal leaflet. The atrialized right ventricle is small. Type B, moderate displacement of the septal leaflet with a large atrialized right ventricle. Type C, important displacement of the septal and postero-inferior leaflet associated with a non- or dyskinetic atrialized right ventricle, and restricted anterior leaflet motion with short cords. Type D, tricuspid sack.

View larger version (270K): [in this window] [in a new window]   Fig. 2. Correction of tricuspid valve and right ventricular anomalies. (a) Mobilization of the anterior leaflet and longitudinal plication of the atrialized portion of the right ventricle. (b) Clockwise rotation of the anterior leaflet and suture on the atrioventricular junction.

The superior venous cannula was placed into the innominate vein and the azygos vein was divided in all patients. The superior vena cava was transected 4 mm superior to the junction of the right atrium. The distal extremity was sutured twice taking care not to injure the sinus node with the cross-clamp. The right pulmonary artery was freed of all surrounding structures: the roof of the left atrium, mediastum and the posterior wall of the ascending aorta. The total length of the pulmonary artery was mobilized from its origin to the bifurcation. The anastomosis of the superior vena cava was performed on the superior wall of the right pulmonary artery, in an end-to-side position with no special attention paid to the angle of the two vessels. Our main concern was to perform the anastomosis as wide as possible. The main pulmonary artery was left open.

The bi-directional cavopulmonary shunt was performed in all patients except one, at the end of the intracardiac repair prior to weaning the patient from bypass.

In one patient, attempts to wean the extra-corporeal circulation after intra-cardiac repair alone lead to dilatation of the right atrium and ventricle associated with low systemic pressure. A bi-directional cavopulmonary shunt was then added in order to save the situation. Four years after the initial operation, one patient received a bi-directional cavopulmonary shunt associated with a second repair of the TV valve.

	Results

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The operative mortality was 24.4% (11/45) (CL 95%: 13–40) in Group I and 0% (0/15) (CL 95%: 0.1–25) in Group II (P<0.05) ( Fig. 3 ).

View larger version (69K): [in this window] [in a new window]   Fig. 3. Operative mortality in group I (without bi-directional cavopulmonary shunt) compared to group II (with bi-directional cavopulmonary shunt). Grey column indicates the total number of surviving patients in each group, the dash lines indicates the operative mortality.

In Group I, three patients died during mid-term follow up. Two patients died suddenly and unexplained at home, 0.3 and 2.2 years after their operations, respectively. No autopsies were performed. A patient was transplanted 3.5 years after the initial operation for recurrence of his TV insufficiency and RV failure. He subsequently died shortly after cardiac transplantation. In Group II, a patient died suddenly and unexplained at home 13 months after surgery. She had been in sinus rhythm with no cardiac problems. Autopsy was not performed.

The actuarial survival at 5 years was 66.1±14% in Group I and 80±16% in Group II ( Fig. 4 ). The difference is not statistically significant.

View larger version (16K): [in this window] [in a new window]   Fig. 4. Actuarial survival. The difference between group I and II is not statistically significant.

The functional improvements are shown in Table 3. There is a striking difference between the two groups. The improvement in clinical status was present in both groups compared with the overall preoperative condition. However, 15% (five patients) of Group I were in NYHA functional class 3 or 4 mainly due to the recurrent tricuspid valve insufficiency. These five patients were all eventually reoperated on.

Reoperations were due to recurrent and/or residual tricuspid valve insufficiency. One patient had severe right ventricular failure and two patients had second repairs with improvement in their tricuspid valve function with one patient receiving a bi-directional cavopulmonary shunt. Two patients underwent valve replacement.

	Discussion

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Surgery for Ebstein's anomaly may be achieved with a reasonable risk [10] [11]. However, the prognostic factors for operative mortality are not well established.

Adding a bi-directional cavopulmonary shunt to the intracardiac repair of Ebstein's anomaly raises two questions: is it useful and how does one assess the preoperative right ventricular function for the selection of the patients?

Bi-directional cavopulmonary shunt is usually performed to supply or increase pulmonary artery flow [12] [13]. Decreasing the right ventricular preload [14] [15] is a very unusual procedure in a repair with two ventricles and no septal defect. Bi-directional cavopulmonary shunt was originally reported in a stenotic Ebstein's anomaly by Marcelletti [16], in a case similar to tricuspid valve atresia.

We assumed that in Ebstein's anomaly the pulmonary artery resistances were low before and after intracardiac repair. Operative measurement of mean pulmonary artery pressure was always below 15 mmHg, and confirmed our hypothesis. The surgical procedure is simple and safe and seems to offer no deleterious effects on early and mid-term follow-up. Pulsatile flow through the pulmonary valve seems to have distinct advantages [17] and tends to decrease the risk of blood stasis in the vena cavae. The situation is quite different when pulmonary stenosis is also present with additional pulmonary blood flow from the systemic circulation [2] [18] [19]. Pleural effusions may occur, as well as, chylothorax.

In our series, operative mortality was significantly reduced when using an associated bi-directional cavopulmonary shunt. The early benefits are obvious. The associated procedure may be added even when right ventricular cavities dilate after initial repair. In one of our patients, the result was clearly lifesaving probably due to the immediate decision to decrease the preload of the right ventricle.

The mechanism of improvement of the bi-directional cavopulmonary shunt may be due to an energy sparing effect on the right ventricle during systolic work. The techniques of ventricular and valvular repair were similar in both groups. The benefit may also be attributed to an improvement in our reparative surgery thus following the normal learning curve.

The key point in this series was the assessment of the right ventricular function. Unfortunately, there is no reliable method in Ebstein's anomaly to measure the right ventricular wall stress, ejection fraction, and shortening fraction in a deformed right ventricle [20]. We were missing quantitative measurements for predicting outcome when the contractility seemed to be decreased before repair [21] [22]. This is the reason why we defined a group of patients with high operative risks, using clinical and echocardiographic [22] parameters. It may however be a pitfall to have included a patient with an apparently good form of Ebstein's anomaly in the high risk group. However, patients with grade 1 or 2 TV insufficiency (4% of Group I) (Table 1) had severe rhythm disturbances or TV stenosis. Patients in functional class 1 or 2 also had rhythm disturbances and patients with Type A or B (Carpentier classification) had massive tricuspid valve insufficiency and/or atrial fibrillation. Our experience with the analysis of the operative mortality, lead us to define these criteria. The bi-directional cavopulmonary shunt diverts between 30 and 40% of the systemic venous return from the right ventricle. The wall stress and work index, even if not measured, is proportional to the decrease of the preload. The benefit of the bi-directional cavopulmonary shunt could be related to decreasing the work index of the compromised right ventricle and subsequently prevent postoperative ventricular dilatation.

Interesting results were obtained with bi-directional cavopulmonary shunts in single ventricles with pulmonary stenosis [23]. In young patients, end diastolic volume is reduced after the shunt. In spite of this situation which is hemodynamically different, the reduction of the preload improved the ventricular function.

Analysis of our results shows a discrepancy between the functional improvement and the residual tricuspid valve insufficiency in Group II. In Group I, it was clear that patients who were not improved after repair had to be reoperated on for residual or recurrent TV insufficiency (except for one case which was transplanted). In Group II, the incidence of residual tricuspid valve insufficiency is similar to Group I (Table 4). However, the clinical improvement is much more significant than what was seen in Group I and no patients had to be reoperated on. The number of patients in Group II is small, however, the bi-directional cavopulmonary shunt seems to prevent the deleterious effects of residual tricuspid valve insufficiency. At mid-term follow-up, the incidence of reoperation was dramatically reduced (11% vs. 0%).

Improvements in our results and techniques with the bi-directional cavopulmonary shunt have lead us to extend the indication of this associated procedure. However, the long-term consequences of the shunt are unknown.

In conclusion, high risk operative repair of Ebstein's anomaly needs to be assessed and to be defined for patient risk stratification. Also, bi-directional cavopulmonary shunt associated with tricuspid valve repair and right ventricular remodeling decreases operative mortality and the incidence of reoperation. Further study and long-term follow-up of patients will be needed to define the role of this new surgical tool in the repair of Ebstein's anomaly.

	Footnotes

 
Presented at the 11th Annual Meeting of the European Association for Cardio-thoracic Surgery, Copenhagen, Denmark, September 28 – October 1, 1997.

	Appendix A. Conference discussion

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Dr S. Conte (Leuven, Belgium): I would like to complimentDr Chauvaud for this nice presentation and veryinteresting study.

My question is what is the best timing thatyou can imagine for this procedure in anasymptomatic patient, and what could be yourapproach in small infants with this high risk?Dr Chauvaud: In a non-symptomatic patientI think there is no reason to do this procedure.All of our indications are very classical; cyanosis,right ventricular failure, and some cases ofarrhythmia. It is very interesting to look forindications now with echo Doppler on the rightventricular function, but it is not ready to beapplied.

The problem of Ebstein's anomaly, ininfants is it is very different than the patients Italked about because many infants had associatedlesions, as pulmonary artery atresia, and I thinkthat many are symptomatic due to poor pulmonaryartery blood flow, and in most of these cases it ismore important to increase pulmonary artery bloodflow with a shunt or something else.Dr J. Monro (Southampton, UK): Your results in the smallergroup were very good. Are you inclined to use thattechnique more often now?Dr Chauvaud: Yes. It is obviously whatcomes to our mind. I'm a little bit reluctant toadvise doing it more extensively. However, I'mdoing it extensively. But we don't know thelong-term consequences of partial Glenn. Probablythey will be minimal. But if it is not useful, andespecially when the right ventricle is quite normal,I think there is some question about the indicationof a partial Glenn.Dr Monro: As I understand it,you still have blood going through the rightventricle into the pulmonary artery as well as fromthe SVC, so hopefully you'll get hepatic factorgoing through and won't be worried about theAV history.Dr Chauvaud: Yes.Dr C. Brizard (Victoria, Australia): Sylvain, I left the unit2 years ago, but do you do now the cavopulmonaryconnection systematically for all patients?Dr Chauvaud: No. The results I presentedtoday are for high-risk patients, and these criteriaare quite subjective because we lack a reliableexamination of right ventricular contractility. Butin some cases the isolated intracardiac repair willbe very successful without any associated procedure.So it is for high-risk patients especially.Dr Brizard: Have you had to docavopulmonary connection in the early postoperativecourse, in the first night postoperatively when youhave a low cardiac output due to compression of theleft ventricle by the dilated and hypokinetic rightventricle?Dr Chauvaud: I think it could be wisewhen we get in trouble after the operation, but whatI know of these very dramatic, strong, earlydifficulties, it is too late. So when I feelfilling means that the right ventricle wall is verythin itself I use the partial Glenn, of course atthe end of the procedure during rewarming; I don'thesitate.Dr C. Knott-Craig (Oklahoma City, OK, USA): Have you had a look atthese patients postoperatively and functionally and beenable to demonstrate any difference in the functionalability of those with and without the bidirectionalGlenn?Dr Chauvaud: Yes. We look at them verycarefully, and functionally they are doing betterthan the patient in which we didn't perform apartial Glenn, and I think it is due to the factthat the right ventricle contractility is soimpaired in these patients that it is exactlysimilar to a Fontan procedure with a non-tonic,non-contracted right ventricle, and when a partialGlenn is associated the functional status is muchmore well tolerated than with the intracardiacrepair alone.Dr Knott-Craig: If that is the case, doyou think it's time to start liberalizing theindications for repair of Ebstein's anomaly?Dr Chauvaud: Yes, it is, definitely.

	References

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