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J Thorac Cardiovasc Surg 1998;115:857-868
© 1998 Mosby, Inc.
SURGERY FOR CONGENITAL HEART DISEASE |
From Deutsches Herzzentrum Berlin, Berlin, Germany.
Read at the Seventy-seventh Annual Meeting of The American Association for Thoracic Surgery, Washington, D.C., May 4-7, 1997.
Received for publication May 12, 1997. Revisions requested August 4, 1997; revisions received Sept. 2, 1997. Accepted for publication Oct. 30, 1997. Address for reprints: Professor Dr. Roland Hetzer, Deutsches Herzzentrum Berlin, Klinik für Herz-, Thorax- und Geäßchirurgie, Augustenburger Platz 1, D-13353 Berlin, Germany.
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Abstract |
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 Top Abstract IntroductionPatients and methodsResultsDiscussionAppendix: DiscussionReferences |
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Introduction |
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TopAbstractIntroductionPatients and methodsResultsDiscussionAppendix: DiscussionReferences |
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1 accounts for approximately 0.3% of all congenital heart defects seen in a large pediatric center. 2 At our institution, this type of defect accounts for 1.2% of all cardiac surgical procedures for congenital heart disease. Its most prominent pathologic characteristic is that the septal or posterior leaflets of the tricuspid valve are variably deformed and displaced downward into the right ventricle, without normal attachment to the tricuspid valve anulus. The anterior leaflet is usually enlarged and sail-like but in the normal position. The displacement of the valve toward the apex of the right ventricular cavity divides the right ventricle into "atrialized" and "true" ventricular portions. 3,4 The anulus of the tricuspid valve is almost always dilated, and the valve itself is usually incompetent. An interatrial communication, usually a patent foramen ovale or atrial septal defect (ASD), is a frequent finding, accompanied by right-to-left shunt and systemic arterial desaturation and cyanosis. 5
Since the first surgical attempts, 6-9 operative treatment has been disputed regarding indications and suitable techniques (valve replacement or repair). 3,10-16 We introduced our technique in 1988; since 1994, it has been our standard surgical repair technique for patients with Ebstein's anomaly and tricuspid valve incompetence. Our technique restructures the valve mechanism at the level of the true tricuspid anulus by using the most mobile leaflet for valve closure without plication of the atrialized chamber. This concept was based on the assumption that it may not be necessary to exclude the atrialized chamber, and that it is desirable to restructure the valve mechanism at the level of the true tricuspid anulus by using the most mobile leaflet for valve closure. This study evaluated our midterm results with this therapeutic approach.
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Patients and methods |
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TopAbstractIntroductionPatients and methodsResultsDiscussionAppendix: DiscussionReferences |
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13 where repair was deemed not feasible. The indication for operation was congestive heart failure of various degrees in all patients (Table I). One patient had systemic embolism and 11 had cyanosis with systemic oxygen saturation at rest of between 76% and 92%. All patients had undergone preoperative echocardiographic studies and right and left heart angiography. Diagnosis was established before the operation in all cases, except for that of a 51-year-old-woman in whom a device occlusion of the ASD attempted at the Cardiology Department was followed by dislocation of the device and right heart endocarditis with pulmonary septic embolism. The correct diagnosis of Ebstein's anomaly was made during the emergency operation.
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13) in four patients, and the atrialized chamber took in the larger part of the right ventricle in 15 cases (Carpentier's type B and C anatomy 13), leaving only a smaller trabeculized right ventricle toward the apex, below the anterior leaflet, and in the outflow tract. The anterior leaflet was largest in all cases; however, the posterior leaflet was quite substantial and mobile in eight cases and reached almost the same size as the anterior leaflet in four instances. The anterior leaflet itself showed clefts or deep fenestrations in four instances, and in one case there was a fourth, smaller, accessory leaflet with origin in the anteroseptal commissure. The anterior leaflet in five patients had localized or wide-spread chordal attachment to the right ventricular free wall, restricting leaflet mobility (Carpentier's type C anatomy, 13 Table II).
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3 In this case the huge ostium was divided into halves by passing two strong mattress sutures from the middle of the anterior anulus to the middle of the atrialized septum just below the natural anulus. Through this approximation of the two opposite parts of the anulus, in each of the resulting two ostia the unfenestrated portion of the anterior leaflet could then coapt with the opposing septum. This repair has remained competent now for almost 9 years.
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Since these initial experiences, the technique has been standardized according to the following concept. On careful inspection of all leaflet components, it was determined which leaflet part was the most mobile and substantial and also had sufficient free edge chordae. In two cases of type C anatomy, mobility of a restricted anterior leaflet was enhanced by dissection of the free wall chordae. To ensure sufficient leaflet body, deep fissures or clefts of the anterior leaflet were closed by sutures in two patients. Once the most mobile part was chosen to be used as the main valve-closing leaflet, the remainder of the natural anulus was obliterated at the level of the natural anulus, either by sewing anulus to anulus or by using a large adjacent leaflet part to close the desired ostial portion. In no case was plication of the atrialized chamber attempted. In a large proportion of our patients (10 cases), the posterior half of the tricuspid ostium was closed directly. In two cases the posterior leaflet was sutured with its septal edge to the septum, in three cases most of the anterior part of the anterior leaflet was sewn to the septum, and in five instances the ostium was narrowed at both the anteroseptal commissure and the posterior anulus. In general, 3-0 polypropylene single mattress sutures supported by pledgets of autologous pericardium were used. The sutures on or along the septum were positioned into the muscular part just below the natural anulus, except for those along the posterior septum where sutures were run through the thin fibrous structure representing the natural anulus (Table II
). The repair was evaluated by repeated filling of the ventricle with saline solution to make sure that the leaflet chosen as closing mechanism could in fact fulfill this task satisfactorily. The diameter of the residual valve ostium was measured and was never smaller than 23 to 25 mm. Once the heart was closed and beating, intraoperative transesophageal echocardiography was carried out to assess the valve and heart function.
Additional cardiac procedures
All but two patients had either secundum ASD (nine patients) or patent foramen ovale (eight patients), which were closed directly or with a patch of autologous pericardium in eight cases. One patient had no interatrial communication, and one had partial atrioventricular (AV) canal defect with a cleft in the anterior mitral leaflet. The cleft was closed by direct suture, and the primum ASD was closed with a pericardial patch. In a patient with a perimembranous ventricular septal defect situated just below the anteroseptal commissure, the ventricular septal defect was closed with a patch and the commissure was closed over a 2 cm length above the patch. One patient had an extensive Chiari network in the right atrium, which was resected. Two patients with high-grade valvular and subvalvular pulmonary stenosis received cryopreserved valved homograft conduits implanted between the right ventricular outflow tract and the main pulmonary artery. The patient who was operated on during the course of active endocarditis also showed mitral valve prolapse with moderate mitral incompetence, which was treated during the same operation by posterior anulus shortening with the Paneth annuloplasty. 17 The closure of the right atrial incision was accompanied by a substantial reduction in right atrial volume, which was considered necessary to reduce the size of the heart, which is frequently huge, and give the lung more room in the chest.
Follow-up
All patients have been prospectively followed up on a regular basis at least once a year. Clinical examination, echocardiographic examination, electrocardiography, and Holter monitoring were performed during the regular surveillance. Follow-up was complete in all cases and lasted between 10 and 103 months (median 28 months).
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Results |
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Postoperative rhythm
Two patients had permanent third-degree AV block that necessitated pacemaker implantation. Two more patients had transient AV block that disappeared shortly after the operation. One patient with known Wolff-Parkinson-White syndrome underwent successful catheter ablation of her abnormal pathway after the corrective operation (patient 3). Of five patients who had supraventricular tachyarrhythmic disturbances, one had postoperative third-degree AV block (patient 1) and the tachyarrhythmia disappeared and normal sinus rhythm has since persisted in four patients.
Functional capacities (New York Heart Association functional class)
All long-term surviving patients had improvement of their functional capacity, except for two patients (patients 5 and 6) who had had only mild preoperative symptoms and stayed in the same New York Heart Association class II after the operation, although their preoperative tricuspid valve incompetence was significantly reduced. Improvement was achieved by one class in 12 cases and by two classes in four (Table I). New York Heart Association functional class improved from the preoperative mean of 2.8 to the postoperative value of 1.9.
Cyanosis
Only one patient had no intracardiac communication; all the others displayed cyanosis at rest or during exercise. Arterial oxygen saturation at rest was normal (>95%) in eight patients; cyanosis was present at rest in 11, with arterial oxygen saturation values between 76% and 92% (mean 86%). After the operation, all patients had normal oxygen saturation values (Table I).
Echocardiographic assessment of tricuspid valve function before and after the operation
Tricuspid incompetence, the most dominant clinical finding other than cyanosis, was found in all patients. Tricuspid incompetence was graded II in two patients, III in 14, and IV in three. After the operation tricuspid incompetence regressed significantly in all cases, and there was no instance of tricuspid incompetence graded higher than II. A small jet was seen passing along the septum in all patients; this, however, was estimated to be minimal (0 to I) in 11 cases and I to II in eight.
Because the amount of tricuspid orifice reduction in some of the individual repair techniques was quite substantial, including obliteration of more than half of the originally huge anatomic anulus, a concern was raised regarding whether tricuspid stenosis might ensue. Echocardiographic follow-up studies revealed inflow velocity between 0.5 and 1.3 m/sec in most patients, and velocity was higher than 1.0 m/sec in only three, without clinical relevance (Table I). This corresponded well with the intraoperative evaluation of the residual valve orifice, which was always larger than 23 to 25 mm in diameter, equivalent to a valve orifice area of more than 3.6 cm2.
Although we left the atrialized ventricular chamber unplicated in all patients, echocardiographic serial surveillance revealed no enlargement of the atrialized chamber during the follow-up, and these chambers were mostly the same size as before the operation (Fig. 5).
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Discussion |
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TopAbstractIntroductionPatients and methodsResultsDiscussionAppendix: DiscussionReferences |
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(Table III), and some patients are not candidates for these procedures because of significant abnormalities of the anterior leaflet, such as inadequate size, attachment of the free edge of the leaflet to the ventricular wall, or failure of delamination. 18In contrast, our technique allows good repair even in cases where only the posterior leaflet or part of the anterior leaflet can be remodeled to act as a valve-closing structure. The value of this concept is strongly underlined by the stability of the obtained tricuspid competence, now for as long as 9 years of follow-up. To date, there have been no instances of late functional deterioration.
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6 Lillihei and coworkers, 7 and Hardy and colleagues, 3 repair has not been generally accepted, and valve replacement has been favored as the most straightforward procedure by most surgeons. Danielson's modification of the Hardy technique, applied in more than 300 cases 11,12,18 (Danielson GK, personal communication, 1997), used the mobile anterior leaflet as closing mechanism by obliterating the atrialized chamber in a transverse fashion, bringing the body of the anterior leaflet toward the natural anulus and allowing anterior leaflet coaptation with the atrialized septum. Carpentier's repair technique 13,14 included detachment of the anterior or posterior leaflet at the origin, transection of abnormal chordae if necessary, longitudinal plication of the atrialized chamber, and the reinsertion of detached leaflets to the anatomic anulus above the plicated chamber, also swinging the leaflets toward the posterior septal anulus. Stabilization of this new valve anulus with a prosthetic ring has been proposed. 13,14
The crucial questions that must be raised relate to the importance of the atrialized chamber, and most repair techniques include either transverse plication or longitudinal exclusion of the atrialized chamber. 3,10-15,18 It was argued that when exposed to right ventricular pressure this chamber might have a similarly negative effect on ventricular energy economics as a left ventricular aneurysm on the left ventricle. Furthermore, stasis within the noncontractile sac might promote clot formation. It was supposed that longitudinal plication, resulting in a "ventriculoplasty," might enhance right ventricular function and obliterate the characteristic rhythm disturbances. 13
Unlike in most valve repair techniques, the atrialized chamber remains untouched if the tricuspid valve is to be replaced, without negative sequelae. 19,20 In the only valve repair method with no plication of the atrialized chamber, the base of the anterior leaflet chordal attachment was fixed with a single stitch to the muscular portion of the atrialized septum, creating a "monocusp valve." 16 The Sebening technique, 16 where valve competence is achieved with a "single stitch" tying the tip of the main anterior chordae to the atrialized septum, does not include any reefing procedure on the atrialized chamber. Excellent long-term results have been achieved with this technique in more than 60 cases 16 (Sebening F, personal communication, 1997).
In all our cases, no attempt was made to obliterate or to reduce this chamber. To date, we have not seen any indication of further enlargement of the right ventricle, or of any other undesirable effects. This has been substantiated by results of echocardiographic studies during follow-up. In contrast, our impression from earlier experiences with other techniques is that transverse obliteration, at least in the more pronounced cases of types B and C anatomy, may cause high tension both on the Hardy sutures 3 and the tissue, with unknown effects on the already abnormal septum and the left ventricle. Furthermore, hope has been expressed by some of us that incorporation of the atrialized chamber into the contracting right ventricle may be beneficial in allowing sufficient right ventricular filling during diastole and could stimulate the remaining musculature in the atrialized chamber wall toward hypertrophy and even contribution to right ventricular contraction. This, however, must be examined in further observation and more detailed studies.
Reduction of the tricuspid orifice at the level of the true anulus has not created stenosis in any case. This is confirmed by normal inflow velocity in echocardiography and also by absence of any clinical signs of possible stenosis of the tricuspid valve. Care must certainly be taken to avoid the His bundle area by placing the sutures on the septum below the natural anulus, at least in the anteroseptal area. This was not successful in every case, as demonstrated by two instances of complete heart block necessitating pacemaker insertion. The risk of this complication could, however, be avoided with more experience.
An anatomic classification introduced by Carpentier and colleagues 13,14 appears to be most valuable in surgical decision making regarding the most appropriate procedure. Type A refers to cases with mild to moderate displacement of the septal and posterior leaflet origins, preserving a relatively large trabecularized right ventricular cavity and a smaller atrialized chamber. In type B this relationship is reversed, with a large atrialized chamber and a smaller contracting ventricle. Common in both types, however, is a large and mobile anterior leaflet. This is restricted in motion in type C by adherence to the anterior right ventricular wall of fibrous bands of abnormal chordae. Finally, type D describes those cases where the entire right ventricle is lined by broadly adherent fibrous leaflet tissue, giving rise to what is called a tricuspid sac.
According to this classification, our patients' conditions were all type A, B, or C. However, there was still vast variability, particularly in the appearance and size of the anterior and posterior leaflets. In the first patient of this series there was no distinction between those two leaflets, and this common leaflet showed a multitude of fissures and clefts. Such clefts were encountered in the anterior leaflet in another three instances, and the posterior leaflet had a substantial size and mobility in six patients. In one instance there was even a fourth leaflet in the anteroseptal commissure. It was decided to close the common orifice to allow the most mobile and substantial part to become the closing mechanism. In type C anatomy, where the anterior leaflet was broadly adhering to the right ventricular free wall, part of this leaflet was freed by careful transection of adherent free wall chordae to achieve a greater mobility of at least this section of the leaflet being chosen to play a part in the newly created valve.
Among this group of patients undergoing valve repair, there was no case with the characteristics of the Carpentier type D anatomy. Recently such a case was encountered, in which the entire right ventricular cavity was lined with fibrous leaflet tissue with broad adhesions to the muscle wall and only a circumscribed, stenotic ostium below the right ventricular outflow tract was visible. In fact, the pathologic features of this case were almost identical to those elegantly described by Dr. Ebstein in his original 1866 case presentation. 1 No attempt was made to repair this defect; the shallow remaining cavity under the fibrous lining was opened by incisions in this fibrous sac starting from the restrictive ostium, eliminating the stenosis, and a valve prosthesis was inserted below the level of the natural anulus.
The technique presented here, which focuses on the individual pathologic features of the tricuspid valve in Ebstein's anomaly, certainly needs further follow-up and experiences in a larger patient group. It is our impression so far that this technique offers good clinical results with a small risk, even with a highly abnormal pathology.
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Appendix: Discussion |
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TopAbstractIntroductionPatients and methodsResultsDiscussionAppendix: DiscussionReferences |
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Dr. Tirone E. David (Toronto, Ontario, Canada). Do you refer to type B anatomy, the one with a very small right ventricle and a large atrialized ventricle, or any type of Ebstein's anomaly?
Dr. Kreutzer. No, only for patients with huge right atrium and poor right ventricular function.
Dr. Hetzer. I think this is a valid option. In fact, the first cases of correction of Ebstein's anomaly in the 1950s were such in which a Glenn shunt had been performed to begin with and where the inferior vena cava return was directed toward the right ventricle. I think this may well be an option if you have an extreme case. In our experience we recently saw such type D anatomy and could not do a repair, so we did a valve replacement at the level of the anatomic anulus and just opened the membrane between the atrialized chamber and the trabecularized chamber to alleviate the tricuspid stenosis, which is often present in those cases. But I think it's an option to do a Glenn shunt.
Dr. David. Did you measure the transvalvular gradient by echocardiography after repair?
Dr. Hetzer. Yes, they were all measured. Of inflow velocities on echocardiography, the highest was 1.3 m/sec. Except for three cases, however, the velocity was below 1.0 m/sec in all instances. So although the valve looked quite narrow after repair, there was no functional tricuspid stenosis. I have taken care that after repair there is always an opening at least 2.3 or 2.5 cm in diameter, which gives a valve opening area of at least 3.5 cm 2.
Dr. Dominique R. Metras (Marseille, France). I just would like to know if this was a consecutive series, and whether you consider that the tricuspid apparatus sometimes looks terrible, is totally irreparable, and does need a replacement.
Dr. Hetzer. I did not have time to go into further details. Actually, this series has been consecutive since 1994 in 16 cases. That means the three earliest patients were still operated on according to the impression of the individual case. Because we saw that this concept of creating the valve at the level of the anatomic anulus works well, we have used only this technique since 1994. As I mentioned, however, we recently had a case that we considered not repairable, and we did a valve replacement.
Dr. D. Glenn Pennington (Winston-Salem, N.C.). Many of these patients have very large hearts, atria, and right ventricles. I think this is the situation to which one of the other discussants was referring. In this era of volume reduction, have you plicated a right ventricle? Do you routinely reduce the size of the right atrium, for example? And if you don't plicate the atrialized portion, do you end up with a very large heart? Also, how do you deal with Wolff-Parkinson-White syndrome, or have you?
Dr. Hetzer. Of course, there is concern about the size of the heart. I must say, however, that in patients who have huge hearts the hugest part is the right atrium, not the atrialized chamber. I have not encountered a case in which the right atrial chamber was so huge that I thought it necessary to plicate it. On the other hand, from my earlier experience with the Hardy technique and the Danielson technique, I found that if you have larger atrialized chambers and you plicate them in a transverse form according to Hardy or Danielson, you may place an excessive amount of tension on the remainder of the ventricles, both on the right and on the left. It is to this that I attribute some of the negative outcomes.
I think, of course, the right atrium was reduced massively in all cases, and this certainly gives a lot more space to the heart. On the other hand, if you incorporate this atrialized chamber into the functioning right ventricle you may have better diastolic filling, and there may be even a chance for the scarce musculature in the wall of this atrialized chamber to gain function. At least I think there is some hope for that.
There was one case with a definite Wolff-Parkinson-White syndrome in which we had a catheter ablation done after the operation. But we have seen also in some instances that supraventricular tachycardias disappeared after the correction without any specific treatment.
Dr. Norberto G. De Vega (Malaga, Spain). I notice that in some cases you repaired the valve by creating two orifices. I am very interested in this concept. I have done some cases in the mitral position to repair the mitral valve, creating a two-orifice valve with very good results. Do you have any experience in mitral repair with this concept? I gather this is Dr. Alfieri's technique.
Dr. Hetzer. Yes. When I read Alfieri's article, I of course thought that this is good concept. We have used this only in one case. But as you know, this has become sort of a routine valve procedure for huge left ventricles, for instance in conjunction with the Batista operation. I think it may well be a valuable option for mitral valve repair as well if it is difficult to use one of the more established types of repair. I completely agree with you.
Dr. Joãtao Q. Melo (Carnaxide, Portugal). Dr. Hetzer, for the last few months we have operated on patients with Wolff-Parkinson-White syndrome and concomitant significant coronary artery disease. During the bypass grafting procedure, we successfully used radiofrequency catheter ablation under direct operative vision.
Do you believe that in Ebstein's anomaly we can correct the tricuspid valve and the Wolff-Parkinson-White syndrome in the same operation?
Dr. Hetzer. I am about to have a good electrophysiologist join our group, and I think that he can probably handle these special cases in the same way.
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Acknowledgments |
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Footnotes |
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