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Ann Thorac Surg 1998;66:1546-1550
© 1998 The Society of Thoracic Surgeons

Alternative approach to the repair of Ebstein’s malformation: intracardiac repair with ventricular unloading

^a Cardiothoracic Surgery, University of California, San Francisco, San Francisco, California, USA
^b Pediatric Cardiology, University of California, San Francisco, San Francisco, California, USA

Accepted for publication May 20, 1998.

Address reprint requests to Dr Reddy, 505 Parnassus Ave, M589, San Francisco, CA 94143-0118

	Abstract

Top Abstract Introduction Patients and methods Results Comment References

 
Background. Moderate to severe Ebstein’s malformation remains a surgical challenge. Although the various approaches that have been used are appropriate and successful in many patients, there are many for which these approaches are suboptimal. To improve the prognosis for patients across the full spectrum of Ebstein’s malformation, alternative surgical approaches are necessary.

Methods. From December 1995 to October 1997, 10 patients (median age, 9 years) with moderate or severe Ebstein’s malformation and mild to severe tricuspid regurgitation had partial biventricular repair with reduction of right ventricular volume load. All patients were symptomatic in New York Heart Association functional class II (n = 9) or III (n = 1). In addition to bidirectional cavopulmonary anastomosis and closure of intracardiac defects in all patients, 6 underwent tricuspid valve repair using a variety of procedures, most often simple horizontal annuloplasty.

Results. There were no deaths. Early reoperation was required in 1 patient (atrial septostomy on the day after operation for right ventricular failure) and another required revision of the tricuspid valve repair 10 months postoperatively for recurrent regurgitation. At follow-up ranging from 2 to 24 months, all patients are in New York Heart Association class I and have trivial tricuspid regurgitation, including the 4 who had no tricuspid valvuloplasty performed.

Conclusions. We have presented an alternative approach to the management of severe Ebstein’s malformation that focuses on both the tricuspid valve and the right ventricle. Just as tricuspid valve repair and reduction of regurgitation will likely improve right ventricular performance, reducing the volume load on the ventricle may improve both ventricular (right and left) and tricuspid valve function. All patients have demonstrated improved exercise tolerance and right heart function at follow-up ranging to 24 months. Additional experience will be necessary to evaluate this strategy more completely.

	Introduction

Top Abstract Introduction Patients and methods Results Comment References

 
Ebstein’s malformation can present with a wide spectrum of morphologic and clinical severity. Although often referred to as Ebstein’s malformation of the tricuspid valve, this lesion involves not only the valve, but also the right ventricle. The distinctive pathologic features of the tricuspid valve are downward displacement of the proximal attachments of the septal and posterior leaflets and marked dilatation of the annulus [1, 2]. The right ventricular myocardium is generally abnormal as well, with impaired contractility and histologic dysplasia in severe cases, and atrialization of the segment between the attachments of the displaced leaflets and the true atrioventricular junction [3]. Symptomatic children tend to present with cyanosis (attributable to right to left shunting through the typical secundum atrial septal defect) and tricuspid regurgitation, whereas rhythm abnormalities are the most common cause of symptoms among patients presenting in adulthood [4]. Right ventricular volume overload and paradoxic septal motion may compromise left ventricular function as well [5, 6]. Patients with severe forms of this lesion may demonstrate profound congestive heart failure and die early in infancy or even during fetal life [7, 8]. In contrast, those with a mildly deformed tricuspid valve may remain asymptomatic well into adulthood [4].

Management for moderate to severe Ebstein’s malformation continues to pose a challenge to the surgeon. Various approaches have been used, including tricuspid valve replacement [9, 10], valve repair [11, 12], and right heart reconstructive techniques [10, 13]. Other investigators have opted for functional univentricular palliation using the Fontan operation [14, 15]. Although these approaches are appropriate and successful in many patients, there are many more for which they are suboptimal. To improve the prognosis for patients across the entire spectrum of Ebstein’s malformation, alternative surgical approaches are necessary. In this report, we review our experience with 10 recent patients in whom such an alternative approach was used; the right ventricle was unloaded with a bidirectional cavopulmonary shunt and complete intracardiac repair was performed, with tricuspid valvuloplasty when indicated.

	Patients and methods

Top Abstract Introduction Patients and methods Results Comment References

 
Between December 1995 and October 1997, 10 patients with moderate to severe Ebstein’s malformation underwent partial biventricular repair with correction of all intracardiac defects and supplementary bidirectional cavopulmonary anastomosis. Five patients with Ebstein’s malformation and severe right ventricular hypoplasia, pulmonary atresia, and intact ventricular septum underwent functional single ventricle repair during this time period and were excluded from the present study. Patient records were reviewed retrospectively and cross-sectional follow-up was performed in December 1997. Patients ranged in age from 2.4 to 31 years (median, 9 years). There were 7 female and 3 male patients. Four patients had undergone previous cardiac interventions: tricuspid ring annuloplasty in 1 patient, atrial septal defect closure in 1, pulmonary artery banding and subsequent ventricular septal defect closure in 1, and radiofrequency ablation of accessory conduction pathways in 1. All patients had moderate to severe downward displacement of the septal and posterior leaflets of the tricuspid valve, along with mild to severe tricuspid regurgitation, a hypoplastic right ventricle, and a secundum atrial septal defect (previously closed in 1 patient). According to the Carpentier classification, 5 patients were in group B and 5 were in group C [1, 13]. In addition, 2 patients had a ventricular septal defect (1 muscular, 1 perimembranous) and another had partially anomalous pulmonary venous drainage, with the left upper pulmonary vein draining to a left superior vena cava that entered the left atrium. One patient had a history of reentrant supraventricular tachycardia through accessory right-sided conduction pathways. All but 1 patient (who had undergone previous closure of the atrial septal defect) was shunting right to left at the atrial level, and preoperative arterial oxygen saturation in the 9 patients with an atrial septal defect ranged from 84% to 96% (median, 92%). Patients were in New York Heart Association functional class II (n = 9) or III (n = 1). All patients were evaluated preoperatively with cross-sectional and Doppler echocardiography [16], and none had cardiac catheterization performed as part of the diagnostic work-up. Patients were referred for operation on the basis of exercise intolerance, cyanosis, progressive tricuspid regurgitation, or intractable reentrant supraventricular tachycardia.

Surgical procedures
The heart and great vessels were exposed through a standard median sternotomy. The pericardium was harvested and fixed in glutaraldehyde. Pursestring sutures were placed in the aorta, superior vena cava, and inferior vena cava and cardiopulmonary bypass was instituted with aortic and bicaval cannulation. The superior caval cannula was placed at the level of the innominate vein to facilitate bidirectional cavopulmonary anastomosis. The venae cavae were snared and the patient was cooled to 28°C, after which the aorta was cross-clamped and cardioplegia was administered. A right atriotomy was performed and the atrial septum and tricuspid valve were inspected. In the 9 patients with a previously unrepaired secundum atrial septal defect, the defect was closed either with a patch of autologous pericardium or directly with pledgeted sutures. In the patient with a previously unrepaired ventricular septal defect, adjacent nonfunctional tricuspid valve tissue was used to close the defect. In 4 patients (patients 1, 2, 4, 7), a segment of dilated right atrial free wall was resected to reduce right atrial size. In one of these patients (patient 1), a modified Cox-Maze procedure [17] was incorporated into the right atrial reduction procedure. In the patient with intractable reentrant supraventricular tachycardia through accessory right-sided pathways (patient 8), multiple muscle bridges spanning the right atrioventricular function were found. To divide these muscle bridges and obliterate all accessory pathways, the right atrium was completely detached from the right ventricle around 80% of the circumference of the atrioventricular junction (sparing the septal attachment) and then reattached with continuous 4-0 monofilament nonabsorbable suture.

The right ventricle was saline-loaded with a bulb syringe to evaluate the competence of the tricuspid valve. In 4 patients with mild tricuspid regurgitation preoperatively, no specific defects were identified and it was estimated that reduction of right ventricular volume with a bidirectional cavopulmonary anastomosis would be sufficient to reduce the level of tricuspid regurgitation. In the remaining 6 patients, tricuspid valve repair was performed using a variety of techniques. Minor commissural annuloplasty was performed with 1 or 2 pledgeted horizontal mattress sutures in 6 patients. Anteroseptal commissuroplasty was performed in 2 patients by partially closing the base of the commissure between the large anterior and vestigial septal leaflets with interrupted pledgeted sutures. Three patients underwent suture closure of multiple or large fenestrations in the anterior (n = 2) or posterior (n = 1) tricuspid valve leaflets. One patient, who had previously undergone placement of a tricuspid annuloplasty ring at the true atrioventricular junction rather than the level of attachment of the leaflets, required removal of the previous ring and pericardial augmentation of the septal leaflet. A piece of fresh autologous pericardium was sutured to the septal surface of the tricuspid annulus until it approached the severely displaced posterior leaflet and then sewn to the free edge of the posterior leaflet. This patient also underwent commissuroplasty and annuloplasty and is included in the above mention of these procedures. After tricuspid valve repair, the valve was again tested by loading the right ventricle with saline from a bulb syringe. When a good result was achieved, the right atriotomy was closed.

All patients underwent bidirectional superior cavopulmonary anastomosis using standard techniques [18]. In 1 patient with bilateral superior venae cavae, a bilateral bidirectional cavopulmonary anastomosis was performed. In this patient, the left upper pulmonary vein was draining to the left superior cava just proximal to the left cavoatrial junction, and the cava was transected superior to this point, allowing the pulmonary vein to drain to the left atrium. The decision to proceed with a bidirectional cavopulmonary anastomosis was made preoperatively in 8 patients, in whom cardiopulmonary bypass was maintained and the procedure performed during rewarming. In the other 2 patients, the decision was made after separation from cardiopulmonary bypass when transesophageal echocardiography showed persistent moderate tricuspid regurgitation (n = 1) or when hemodynamic and transesophageal echocardiographic monitoring showed evidence of right ventricular overload, with a right atrial pressure of 12 mm Hg (simultaneous left atrial pressure 5 mm Hg) and a dilated right ventricle with significant septal bowing into the left ventricle. These patients were returned to cardiopulmonary bypass and the procedure was performed on a warm beating heart.

	Results

Top Abstract Introduction Patients and methods Results Comment References

 
There were no early deaths. Early reoperation was performed in 1 patient (patient 10), who required atrial septostomy 8 hours after repair for poor right ventricular function, then evacuation of an intramyocardial hematoma the following day. There were no other significant complications. Intraoperative transesophageal echocardiography after bypass showed decreased tricuspid regurgitation in all patients, including the 4 in whom no valve intervention was performed (Table 1 ) (Table 1). No patient had evidence of tricuspid stenosis or residual intracardiac defects. Echocardiography before discharge showed no change from the transesophageal echocardiogram. All patients were discharged within 11 days after operation, and the median postoperative hospital stay was 7 days.

	Comment

Top Abstract Introduction Patients and methods Results Comment References

 
Ebstein’s malformation is a challenging surgical lesion, for which a variety operations have been developed [9–13]. The central pathologic feature of this anomaly is the tricuspid valve, and it is here that most surgical treatments have been focused [9–12]. However, Ebstein’s malformation is more than simply a defect of the tricuspid valve. The right ventricle is equally important in the dysfunction of the right heart in these patients, and it is essential to consider this factor in designing an approach to repair.

It is well known that altering the loading conditions of the morphologic right ventricle can have a significant effect on tricuspid valvar function [19–21]. With a substantial reduction in volume load, as is achieved with bidirectional cavopulmonary anastomosis, alteration in tricuspid valvar function is probably attributable largely to a combination of geometric changes in the ventricle and valvar apparatus [19]. In the setting of Ebstein’s malformation with an atrial septal defect, the interatrial defect allows for decompression of the right heart preoperatively. In such patients, closure of the defect may in effect induce right ventricular volume overload. Depending on the presence of an atrial defect and the magnitude of right to left shunting at the atrial level, a bidirectional cavopulmonary anastomosis may either reduce an existing right ventricular volume load or prevent the development of such a load when the interatrial defect is closed. In patients with Ebstein’s malformation, in which the geometry of right ventricle and tricuspid valve are abnormal at baseline, the altered right heart geometry and mechanics induced by reduction of volume load may be substantial. As the results of this series demonstrate, reduction of right ventricular preload alone may reduce tricuspid regurgitation to the point where no tricuspid valve intervention is required. In our series, this was the case only in patients with mild regurgitation preoperatively. However, by this same principle, patients with moderate regurgitation can experience a significant reduction in regurgitation with reduction of volume load along with minimal tricuspid valve interventions such as minor suture annuloplasty or commissuroplasty procedures. Therefore, the native structure of the valve can be maximally preserved, with no need for major right heart reconstructive techniques [10, 13], even in patients with moderate or severe forms of the disease. Furthermore, supplementary bidirectional cavopulmonary anastomosis may allow for more aggressive tricuspid annuloplasty in patients requiring a more extensive procedure, insofar as reduced transtricuspid valve flow minimizes the risk of iatrogenic tricuspid stenosis. This may obviate prosthetic valve replacement or annuloplasty rings, which are required in a substantial number of patients in other series [1, 10]. The efficacy of this approach in patients with severe tricuspid regurgitation cannot be ascertained from this study. The 1 patient with severe regurgitation required a more substantial procedure on the tricuspid valve, but this was confounded by the presence of a previous, incorrectly placed annuloplasty ring. Although ventricular unloading alone is almost certainly insufficient for complete relief of severe tricuspid regurgitation, we predict that it will facilitate less aggressive tricuspid valve intervention, as with the patients in our series who had moderate preoperative regurgitation.

Right ventricular unloading is likely to have salutary effects not only on tricuspid valve function, but on right ventricular function as well [19]. Reduced preload decreases the obligatory stroke work of an entire cardiac output, and is likely to contribute to normal right ventricular function after closure of all intracardiac defects. The concept of partial biventricular repair in patients with hypoplastic right heart has recently achieved an increasingly prominent role in the management of complex congenital heart disease [22–25]. Ebstein’s malformation is one of the most suitable lesions to which to apply this concept. In addition to the factors discussed above regarding the benefits of ventricular unloading for tricuspid valve and right ventricular function, reduction of right ventricular volume load may also lead to improved left ventricular function in patients with Ebstein’s malformation by minimizing the deleterious effects of right to left ventricular interaction, which have been shown to interfere with normal left ventricular mechanics in this lesion [6].

There are several concerns with bidirectional cavopulmonary anastomosis in the setting of antegrade flow from the right ventricle. The most significant of these is the effect of elevated pressure and pulsatility in the superior vena cava. Indeed, superior vena cava syndrome is a concern in patients undergoing partial biventricular repair with a bidirectional superior cavopulmonary anastomosis, and vigilance regarding this complication is required in the early postoperative period. Although none of the patients in this series suffered from superior vena cava syndrome, it has occurred in 1 of our patients who underwent partial biventricular repair with a bidirectional cavopulmonary anastomosis for pulmonary atresia with intact ventricular septum. The most obvious options for managing this complication are reduction of pulsatility and pressure in the superior vena cava by banding the right pulmonary artery between the pulmonary trunk and the cavopulmonary anastomosis, or if this is unsuccessful, conversion to functional univentricular palliation.

In considering which approach to Ebstein’s malformation is most appropriate for any given patient, careful echocardiographic and hemodynamic evaluation is imperative. In the planning of this procedure, we have not relied on measurement of right ventricular volume, which can be difficult to measure accurately, especially in patients with the distorted right ventricular geometry characteristic of moderate to severe Ebstein’s malformation. Rather, we consider a variety of factors assessed by preoperative echocardiography, including severity of downward displacement of the septal and posterior leaflets, degree of tricuspid regurgitation, degree of right to left shunting, right ventricular function, geometry and mechanics of the interventricular septum, and the status of the right ventricular outflow tract. Since we adopted this approach in late 1995, there have been 5 patients with Ebstein’s malformation who did not undergo partial biventricular repair, all of whom had severe right ventricular hypoplasia and pulmonary atresia. Operative and intraoperative transesophageal echocardiographic findings can also significantly affect our decision. For example, only 8 of the patients in the present series had the bidirectional cavopulmonary anastomosis performed during the initial bypass run. In the other 2 patients, initially we performed biventricular repair, after which bypass was discontinued and right ventricular and tricuspid valve function were evaluated. Pressure monitoring and transesophageal echocardiography showed evidence of persistent tricuspid valve or right ventricular dysfunction, which led us to return to bypass and perform the bidirectional cavopulmonary anastomosis. Our threshold for adding a bidirectional cavopulmonary anastomosis in patients in whom biventricular repair is initially attempted is a right atrial pressure of approximately 12 mm Hg or at least twice left atrial pressure. However, it is essential to augment this information with intraoperative transesophageal echocardiography.

In conclusion, we have presented an approach to the management of Ebstein’s malformation that focuses on both the tricuspid valve and the right ventricle. Just as tricuspid valve repair and reduction of regurgitation will likely improve right ventricular performance, reducing the volume load on the ventricle may improve both ventricular (right and left) and tricuspid valvar function. Thus, the two aspects of this approach may function synergistically. Long-term follow-up is not yet available on these patients, but they have all demonstrated improved exercise tolerance and right heart function at follow-up of up to 24 months.

	References

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