TY - JOUR
AU - Carpentier,, A.
AB - Abstract Objective: ‘Classical’ repair of Ebstein's anomaly is usually performed with transverse plication of the atrialized chamber. However, the anterior leaflet has restricted motion which is an important factor of the tricuspid valve insufficiency. We studied the long term results of mobilization of the anterior leaflet associated with longitudinal plication of the right ventricule. Methods: From 1980 to July 2002, 191 patients (mean age 24.4±15 years (1–65)) were operated on. Anterior leaflet function was assessed on pre-op echocardiography and on surgical examination. Conservative surgery was possible in 187 patients (98%) and included mobilization of the anterior leaflet, longitudinal plication of the right ventricle and prosthetic annuloplasty in adults. Bidirectional cavo-pulmonary shunt was associated in 60 patients. Four patients had valve replacement. Results: Hospital mortality occurred in 18 patients: 9% (95%CL: 6–15%) due to right ventricle (RV) failure in nine patients. Mean follow-up was 6.4 years (0.07–22). Actuarial survival was 82% at 20 years. Tricuspid valve insufficiency was 1 or 2+ in 80% of the cases. Reoperation occurred in 8% (16 patients). A successful second repair was obtained in ten patients. Electron beam computerized tomography (20 patients) demonstrated improved left ventricle ejection fraction 56–66% (P<0.05). Supraventricular tachycardia and pre-excitation syndromes were reduced from 23 to 5%. Conclusion: Conservative surgery is indicated for all symptomatic patients. The incidence of valve repair is high when leaflet mobilization is performed. Valve replacement can be avoided in most cases. Functional and hemodynamic results are excellent. Ebstein's anomaly, Tricuspid valve 1 Introduction Conservative techniques of Ebstein's anomaly are numerous [1–3]. Most are based on the plication of the tricuspid annulus associated or not with a transverse plication of the atrialized chamber. However, anomalies of the anterior leaflet [4] are not treated and could explain the need for valve replacement [5]. Mobilization of anterior leaflet was introduced in 1980 by Carpentier et al. [6]. In the current study, we analyze the results and feasibility of the technique, the incidences of residual or recurrent tricuspid valve insufficiency and reoperation. 2 Material and methods From January 1980 to July 2002, 191 consecutive patients (pts) were included in this series with the intent of performing reconstructive surgery. Mean age was 24.7±15 years (range 1–65). Our center has no activity in neonate surgery and subsequently neonate were no referred to our center. Cyanosis was present in 82 patients (43%). Cyanosis was defined by an arterial saturation at rest lower than 92%. Preoperative functional class (New York Heart Association) was one in 2% (four pts), two in 39% (73 pts), three in 44% (84 pts) and four in 16% (30 pts). Mean cardiothoracic ratio was 0.65±0.06 (0.5–0.8). Tricuspid valve insufficiency (TVI) was assessed by echocardiography and/or angiocardiography in our early experience. Anterior leaflet motion was studied (Fig. 1 ) with echocardiography (172 pts), computerized tomography (20 pts), and magnetic resonance imaging (four pts). TVI was graded 1–4+. It was 1+ in 2% (three pts), 2+ in 12% (23 pts), 3+ in 48% (93 pts) and 4+ in 38% (72 pts). Tricuspid valve stenosis was present in 31 pts (16%). Fig. 1 Open in new tabDownload slide Restricted anterior leaflet motion; (a) Echocardiography: the anterior leaflet is tethered to the ventricular wall; (b) Computerized tomography: a muscular band restricts the anterior leaflet; (c) Magnetic resonance imaging: the papillary muscle is included in the infundibulum; RA, right atrium; RV, right ventricle; LV, left ventricle. Fig. 1 Open in new tabDownload slide Restricted anterior leaflet motion; (a) Echocardiography: the anterior leaflet is tethered to the ventricular wall; (b) Computerized tomography: a muscular band restricts the anterior leaflet; (c) Magnetic resonance imaging: the papillary muscle is included in the infundibulum; RA, right atrium; RV, right ventricle; LV, left ventricle. According to Carpentier's classification [6], Type A was present in 7% (14 pts), Type B in 34% (64 pts), Type C in 53% (101 pts) and Type D in 6% (12 pts). Associated anomalies are listed in Table 1 : the most frequent was the presence of an atrial septal defect (ASD). Twelve pts (6%) had a previous operation (Table 2 ). Indications for surgery were functional disability (60%), cyanosis (38%), and rhythm disturbances not improved by medical treatment (45%). Table 1 Open in new tabDownload slide Associated anomalies Table 1 Open in new tabDownload slide Associated anomalies Table 2 Open in new tabDownload slide Previous operation Table 2 Open in new tabDownload slide Previous operation The surgical technique performed has been previously described in detail [6,7]. The goals were to restore normal tricuspid function and to preserve right ventricular contractility. Conservative surgery was possible in 187 pts (98%). The steps were mobilization of the anterior leaflet in all cases, longitudinal plication of the atrialized chamber: 79% (150 pts), prosthetic tricuspid Carpentier ring: 63% (120 pts), papillary muscle reimplantation: 8% (15 pts). Mobilization of the anterior leaflet was obtained by detachment of the leaflet from the annulus, from the antero-septal commissure to the junction with the posterior leaflet when present. An overview of the right ventricular chamber was thus obtained. All abnormal muscular adhesions from the leaflet tissue to the right ventricular wall were dissected with scissors as far as possible in the right ventricle (Fig. 2 ). A lateral papillary muscle was individualized when possible. Fig. 2 Open in new tabDownload slide Operative view. The anterior leaflet is detached from the annulus, allowing the access to the infundibulum. Muscular bands are divided with scissors in order to mobilize the anterior leaflet. Fig. 2 Open in new tabDownload slide Operative view. The anterior leaflet is detached from the annulus, allowing the access to the infundibulum. Muscular bands are divided with scissors in order to mobilize the anterior leaflet. Since 1994, a bidirectional cavopulmonary shunt (BPCS) was performed under cardiopulmonary bypass at the end of the valvular procedure in patients with decreased right ventricle contractility. The vena cava anastomosis was wide, along the total length of the right pulmonary artery from its origin to division at the pericardial reflection. An associated BPCS was performed in 60 pts (36 %). At the end of the procedure the mean pressure in the superior vena cava was 16±5 mmHg (max 20 mmHg, min 12 mmHg). Associated valvular techniques were fenestration of the anterior leaflet (four pts), anterior leaflet enlargement (four pts) with a glutaraldehyde autologous pericardial patch. The infundibulum of the RV was plicated externally from the pulmonary annulus to the apex in four pts. The atrial septal defect was always closed except in two pts. Mapping and cryofulguration of the accessory pathways were performed in two pts. Ventricular septal defect (two pts), mitral valve insufficiency (two pts), pulmonary stenosis (one pts) and subaortic stenosis (one pts) were treated during the same operation. Four patients underwent tricuspid valve replacement by a bioprosthesis. Indications for replacement were: leaflet tissue totally adherent to the ventricular wall (Type D): three cases, partial AV defect with hypoplasia of leaflet tissue: one case. In all but three pts, the aorta was cross clamped at 28°C. Blood or cold cristalloïd cardioplegia was injected in the aortic root and repeated if necessary every 30 min. Slush ice was placed on the right and left ventricles. Three patients with poor left and right ventricular function were operated on without aortic cross clamping on a beating heart. A morphological study by electron beam computerized tomography (Imatron C100 and Siemens Evolution) with EKG triggered, 3 mm contiguous axial slices, after contrast injection was performed on 20 pts. The cine study was performed at 12 levels, with 12–20 views per level in short axis view. Measurements of right and left ventricular volumes were performed independently by two examiners, before and after surgery. Statistical analysis: hospital mortality was defined as death during surgery or within 30 days of surgery. The Kaplan–Meier method was used to calculate the actuarial survival and the probability of remaining free from reoperation. Confidence limits (95%) were calculated. Continuous variables were evaluated with Student's tests, values of P less than 0.05 were considered to be statistically significant. The association of qualitative risk factors with perioperative death was evaluated with χ2 test. 3 Results Hospital mortality was 9% (18 pts) (CL 95%: 6–15%). Prior to 1994 (introduction of BPCS in selected patients), hospital mortality was 12% (10/82) (CL 95%: 6–22%). After 1994, hospital mortality decreased to 7% (8/109) (CL 95%: 3–14%) and the difference was statistically significant. The causes of hospital deaths are listed in Table 3 . The most frequent cause of operative death was right ventricular failure which associated low cardiac output with no improvement following volume replacement and inotropic support. Systemic acidosis and anuria appeared very early after surgery: 6–8 h. On echocardiography, the left ventricle appeared small, empty and compressed by the RV. The right ventricle was very dilated and hypokinetic. Table 3 Open in new tabDownload slide Causes of hospital death Table 3 Open in new tabDownload slide Causes of hospital death Before 1994 hospital death was due to right ventricular failure in 6/12 (50%) patients. After 1994 hospital death was due to right ventricular failure in 3/8 (37%) patients. Non-lethal perioperative complications were AV dissociation with pace maker implantation (five pts), pericardial effusion (seven pts), thrombosis of the bioprosthesis (one pt). Information concerning clinical status were obtained by examining patients and reviewing charts during surgical consultation in our own center or through the referring physician. Three patients were lost to follow-up. Mean follow-up was 6.37±5.1 years (y) (0.01–22). Seven patients died at a mean delay of 2.8±0.7 y (0.36–13). Three patients died of sudden death at home of unknown cause follow a delay of 0.73, 0.36 and 2.2 y. These patients were all doing well up to the time of death. Post mortem examinations were not performed. These sudden deaths were attributed to rhythm disturbances. Other causes of death were ventricular fibrillation (one pt), seizure (one pt), reoperation for tricuspid valve insufficiency and RV failure (one pt) and late deterioration of ventricular contractility (one pt). The actuarial (Kaplan–Meier) survival rate was 86.6±2.6% at 10 y and 82.2±4.7% at 20 y (Fig. 3 ). Fig. 3 Open in new tabDownload slide (a) Actuarial survival, (b) freedom from reoperation of the surviving patients. Fig. 3 Open in new tabDownload slide (a) Actuarial survival, (b) freedom from reoperation of the surviving patients. Among the 156 pts followed for more than 1 year and free from reoperation, 147 pts were in functional class (NYHA) I and nine pts in class II. Sixteen patients were reoperated, followed by a mean delay of 2.6 years (1 day–9.4 y) (Table 4 ). The reoperation was performed for residual tricuspid insufficiency in 14 pts, stenosis in one pt, and bioprosthesis thrombosis in one pt. Conservative surgery was performed in 12 pts using various associated techniques: extensive mobilization of the anterior leaflet in six pts, plication of the annulus in four pts, papillary muscle transfer in one pt, patch extension of the anterior leaflet in three pts, and a prosthetic ring was implanted in six pts. A BPCS was performed in three pts associated with valvular surgery in two pts and isolated in one pt (the indication being TV stenosis). Two patients underwent valve replacement and one pt underwent a cardiac transplantation. Four patients died following reoperation. The actuarial rate of freedom from reoperation, calculated on the surviving patients, was 88.6±4 % at 10 and 20 y (Fig. 3). Table 4 Open in new tabDownload slide Reoperations Table 4 Open in new tabDownload slide Reoperations Evaluation of the tricuspid valve function was assessed by echocardiography at the last follow-up (Table 5 ). This evaluation included all patients alive with conservative surgery, patients who were reoperated on with a repetitive repair, and also patients who died suddenly. Sixty-eight patients had minimal (1+) regurgitation, with the jet localized along the anterior part of the ventricular septum. Six patients had residual tricuspid valve insufficiency (3+) on echocardiography, well tolerated clinically except in one pt. Five of these patients had undergone an associated BPCS. Among these six patients, three had dilatation of the right ventricle. Table 5 Open in new tabDownload slide Degree of tricuspid valve insufficiency Table 5 Open in new tabDownload slide Degree of tricuspid valve insufficiency Tricuspid valve stenosis resolved in all patients with a velocity not exceeding 1 m/s. No deliterious effect of the BPCS was noted except in one pt who had very mild swelling of the upper part of the body with normal pulmonary artery and superior vena cava pressures. The study of ventricular volumes demonstrated decreased contractility of the atrialized chamber with a mean ejection fraction of 37±28%. In two of the 20 patients studied, the atrialized chamber was dyskinetic. Ejection fraction of the effective right ventricle was decreased after surgery from 56±10% to 41±12% (P<0.01). Ejection fraction of the left ventricle was increased after surgery from 56±10% to 68±8% (P<0.01). 4 Discussion It is well known that the clinical and anatomic spectrum of Ebstein's anomaly is very large [4,8,9]. Factors involved in tricuspid valve insufficiency are the enlargement of the tricuspid annulus, the restricted and downward septal leaflet, and the absence or restricted posterior leaflet [10]. In addition, the anterior leaflet is rarely normal and tethering is frequent [4,5]. The classical conservative surgical technique was elaborated by Hunter [11], Lillhei [12] and Hardy [13] with plication of the atrialized chamber. This technique was refined by Danielson with the association of tricuspid annulus plication [1]. However, in a large series of 324 pts [5], half had undergone tricuspid valve replacement. This could be due to the inability to restore normal leaflet coaptation with the plication of the atrialized chamber as an isolated technique. Similar techniques also report a high rate of valve replacement [14]. Simple conservative surgical techniques were published with good mid-term results in short series [2,15,16]. However, in these reports, patients were not consecutive and failures of the technique were not included in the results. The goal of reconstructive valvular surgery is to restore a normal surface of coaptation of the leaflet tissue. In Ebstein's anomaly the anterior leaflet is the only effective leaflet capable of reaching the ventricular septum. Subsequently, the surface of coaptation is between the anterior leaflet and the ventricular septum. Techniques of valve enlargement were developed [17] with some success, and unknown long term results. Repair of the septal leaflet is technically demanding [18]. Mobilization of the anterior leaflet is based on the concept of a restricted anterior leaflet. This functional analysis was assessed by echocardiography. With extensive mobilization, we were able to perform conservative surgery in 98% of patients. Similar results were obtained by others centers [19]. In ten pts of our series, reoperated on for residual tricuspid insufficiency, it was found that mobilization had been minimal and could be improved with success. These patients of our early experience were initially interpreted as technical failures, retrospectively confirming the validity of the concept. Mobilization of the anterior leaflet has been extended to infants when the lesions were complex [20]. The surgical limit of valve repair appears to be Carpentier Type D disease, in which the individualization of the anterior leaflet from the RV wall is not complete. Valvular replacement in such cases appears prudent and effective [5]. Ebstein's anomaly is also a right ventricular disease [21] with most of the operative mortality being due to underestimation of right ventricular contractility. With the introduction of BPCS as an associated technique, operative mortality was reduced and the post-operative course simplified [22]. Decreasing right ventricle preload is an important part of the management of depressed RV contractility [23,24]. Indications for associated BPCS are not clear and are based on clinical data. At the present time we associate BPCS when the right ventricular wall is thin and dilated, when there is a long history of tricuspid valve insufficiency and when the anterior leaflet is widely attached to the ventricular wall. However, isolated BPCS can not resolve the consequences of massive tricuspid insufficiency, as was the case in two of our patients. The atrialized chamber participates in the evolution of the right ventricular impairment. In some patients, we found that contractility was decreased and an anevrysm could be present with dyskinetic motion. Exclusion of the atrialized chamber can be obtained by resection [25] or plication. Longitudinal plication associates exclusion of the atrialized right ventricle and plication of the tricuspid annulus to normal size. In such cases no deleterious effect could be demonstrated. Assessment of RV functions (diastolic and systolic) is still difficult irrespective of the method used. Echocardiography could not explore all the segments of a deformed right ventricle. Computed tomography was useful but the duration of the investigation (10–15 min) is long for children. Magnetic resonance imaging is promising and under evaluation. Reconstructive surgery for severe Ebstein's anomaly requires the association of several surgical techniques corresponding to each individual anatomic and functional aspect. Mobilization of the anterior leaflet is one of them, participating to the increased rate of successful valve reconstruction. Dr A. Corno (Lausanne, Switzerland): I'm very pleased that you mentioned the addition of a bidirectional cavopulmonary shunt to improve the results. We followed your suggestion. Of course, we have very limited experience in Lausanne. We are applying blindly to all the adult patients with Ebstein’s anomaly the cavopulmonary shunt. The question is, do you use this addition in all the patients now or do you select the patients, and in this case, what are the criteria to use or not to use the bidirectional cavopulmonary shunt? Dr Chauvaud: The bidirectional cavopulmonary shunt is not performed in all patients. Selection is made on preoperative echocardiography when there is huge dilatation of the infundibulum or an akinetic ventricular wall. It means that we need investigation of the right ventricular function. During the operation, when we start on bypass, if the right ventricular wall appears very thin, it means that the post operative course will be very difficult and that decreasing the preload of the right ventricle will be mandatory. So indications of BPCS are a combination of several parameters. Dr C. Mavroudis (Chicago, IL, USA): I'm not sure that we have very many good questions to ask you, because you are, obviously, the last word on this. But let me ask you, what did you do in those 15% or 20% of patients that had some kind of an arrhythmia, either accessory connections or reentry tachycardia? I know you didn't say it and maybe you didn't have time to, but do you approach that problem at the same time, and what kind of energy dissipation do you use? Dr Chauvaud: We are effective on Wolff-Parkinson-White syndrome, supraventricular tachycardia, and we are totally ineffective on atrial fibrillation. By using this technique, with detachment of the anterior leaflet from the annulus and using longitudinal plication of the right ventricle, we cut most of the accessory pathways which are located on the right side of the heart behind the coronary sinus. At the present time I associate a limited maze procedure of the right atrium, but I don't have the result of this associated technique. Dr D. Metras (Marseille, France): At the last meeting in Toronto last year on pediatric cardiac surgery and cardiology we heard an enormous series presented by Dr. Danielson from the Mayo Clinic, and he showed in the last series the technique they do that does not include any plication of the atrialized portion of the ventricle, arguing that this plication may interfere with the coronary circulation, do some stenosis or kinking, and may make problems on the right ventricular function. I see that you plicate this chamber, of course, each time. What do you think of this large experience presented by the Mayo Clinic? Dr Chauvaud: I totally disagree. Dr Metras: Not with me. I’m not a representative of the Mayo Clinic. I’m just asking a question. Dr Chauvaud: Indeed there is a risk for coronary circulation with the plication of the atrialized chamber and the tricuspid annulus reduction. I don't want to enter into the details, but insist on the fact that the suture is very superficial, only in the endothelium. We performed isotope investigations of the myocardial vascularization, before and after surgery, and could see that there is no deleterious effect of the plication on the right ventricular function. Dr. C Schreiber (Munich, Germany): I would like to stress the same aspect as my colleague. We were looking at specimen a few years ago with Professor Anderson and found that it is not always necessary to restore the size of the right ventricular cavity; the right ventricle is often big enough, and we in the German Heart Center Munich have repaired almost 100 hearts now with a much simpler technique, just using the anterior leaflet and creating a monocusp valve, with good results. We do have though a slightly higher incidence of reoperations, but the overall outcome is comparable to your series. 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TI - Ebstein's anomaly: repair based on functional analysis
JO - European Journal of Cardio-Thoracic Surgery
DO - 10.1016/S1010-7940(02)00836-9
DA - 2003-04-01
UR - https://www.deepdyve.com/lp/oxford-university-press/ebstein-s-anomaly-repair-based-on-functional-analysis-srvIB80JC3
SP - 525
EP - 531
VL - 23
IS - 4
DP - DeepDyve
ER -