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Department of Pediatric Cardiac Surgery, Sakakibara Heart Institute, Tokyo, Japan
Accepted for publication June 1, 2007.
* Address correspondence to Dr Ando, Department of Pediatric Cardiac Surgery, Sakakibara Heart Institute, 3-16-1 Asahi-cho, Fuchu-si, Tokyo, 183-0003, Japan (Email: maando{at}shi.heart.or.jp).
Presented at the Forty-third Annual Meeting of The Society of Thoracic Surgeons, San Diego, CA, Jan 29–31, 2007.
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Abstract |
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 Top Abstract IntroductionPatients and MethodsResultsCommentDiscussionReferences |
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Methods: Between 1989 and 2006, 49 patients with tricuspid or common atrioventricular valve regurgitation (moderate or greater) associated with single ventricle underwent valve repair 53 times. Edge-to-edge repair was performed in 5 of 23 with tricuspid valve and 17 of 30 with common atrioventricular valve.
Results: In the edge-to-edge group, more patients had severe regurgitation (59.1%) compared with the non–edge-to-edge group (32.3%) before operation (p = 0.0906). The postoperative degree of regurgitation was mild or less in 95.5% of the edge-to-edge group compared with 48.4% of the non–edge-to-edge group (p = 0.0003). Tricuspid or atrioventricular valve stenosis was not observed in any of the patients after repair. The degree of regurgitation on follow-up echocardiogram was mild or less in 77.3% of the edge-to-edge group at 2.6 ± 4.0 years after repair, compared with 38.7% of the non–edge-to-edge group at 1.9 ± 3.2 years (p = 0.109).
Conclusions: Edge-to-edge repair was an effective adjunctive in repairing tricuspid or common atrioventricular valve associated with functionally single ventricle. An excellent reduction of regurgitation was observed in most of the patients, and may lead to preservation of ventricular function during interim stage and successful completion of the Fontan operation.
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Introduction |
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TopAbstractIntroductionPatients and MethodsResultsCommentDiscussionReferences |
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A variety of techniques have been used to repair tricuspid and common AV valves including circular annuloplasty, leaflet resection, repair of cleft leaflets, and chordal shortening [5–7]. Edge-to-edge repair (E-E) has been advocated as an option to repair mitral valve with anterior leaflet prolapse in adult patients [8]. The use of this technique in growing children may not be recommended because of the potential valve stenosis. We hypothesize that tricuspid or common AV valves in the setting of single ventricle (frequently associated with significant annular dilation) may be amenable to repair by this technique, without causing stenosis.
The purpose of this study was to assess the efficacy of E-E applied for tricuspid and common AV valves associated with functionally single ventricle.
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Patients and Methods |
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TopAbstractIntroductionPatients and MethodsResultsCommentDiscussionReferences |
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Clinical data of 236 consecutive patients who underwent either cavopulmonary anastomosis or modified Fontan operation for functionally single ventricle between October 1989 and March 2006 were retrospectively reviewed. We identified 63 patients who were diagnosed with a common AV valve, with 26 (41.3%) requiring valve repair owing to moderate or severe regurgitation. For the purpose of this study, a common AV valve was defined as the absence of primum atrial septum and attachment to the interventricular septum (ie, complete AV septal defect/common inlet AV valve) [9]. Forty-four patients were diagnosed with double-inlet ventricle (two separate AV valves supported by a single ventricle, without an attachment to the ventricular septum), and 7 (15.9%) of them required valve repair. The affected valve was documented to be morphologic tricuspid valve in 5 patients (11.4%) and mitral valve in 2 (4.5%). Sixty-seven patients were diagnosed with tricuspid atresia or severe stenosis, with 2 (3.0%) undergoing mitral valve repair. Among 29 patients with mitral atresia or severe stenosis, 16 (55.2%) underwent tricuspid valve repair. Thirty-three patients were documented to have two well-developed AV valves that were separated by the ventricular septum (eg, double outlet right ventricle with noncommitted ventricular septal defect); 2 patients (6.1%) required tricuspid valve repair and 1 (3.0%) required mitral valve repair.
Overall, 49 patients who had important (moderate or greater) regurgitation of either common AV valve (26) or tricuspid valve (23) underwent valve repair. Eight patients required reoperation, including four prosthetic valve replacements, owing to recurrent regurgitation after initial operation. Therefore, 53 AV valve repairs were performed in these 49 patients, who were the cohort of this study. On the other hand, mitral valve repair was done in 5 patients with no reoperation. There were 25 females and 24 males. Heterotaxia syndrome was present in 21 patients, and Down’s syndrome in 2 patients.
The definitions of anatomical variants of single ventricle and hypoplastic left heart syndrome were guided by the report of The Society of Thoracic Surgeons Nomenclature and Database Project [10, 11]. In cases with double-inlet single ventricle, the tricuspid valve was generally the right-sided valve in d-loop and the left-sided valve in l-loop [12].
Edge-to-edge repair was performed by suturing the free-floating segments of the opposing leaflets using a technique much similar to that described for adult mitral valve disease. In the common AV valve, the superior and inferior leaflets were sutured together. In the tricuspid valve, the anterior and septal leaflets were sutured together.
The method of AV valve repair was selected according to the discretion of each operative surgeon, guided by preoperative echocardiographic data. The decision was also influenced by the patient’s size, with a propensity for a less complicated or innovative approach (such as circular annuloplasty) in smaller patients. The severity of regurgitation was graded semiqualitatively as none, trivial, mild, moderate, or severe by Doppler color echocardiography. A postoperative echocardiogram was performed in all patients, and all data were available for analysis. Clinical follow-up of patients included review of office and hospital charts, correspondence letters, and direct telephone contact. Survival beyond the last patient contact was not assumed.
The SPSS statistical software for Windows (version 11.0; SPSS, Chicago, Illinois) was used for data analysis. Values were expressed as mean ± SD or median. Differences between groups were examined by Student’s t test (continuous variables) or Fisher’s exact test (categorical variables). Cumulative survival was estimated using Kaplan-Meier product limit method. Intergroup comparison was made using the log-rank test. Linear regression equation was calculated to assess chronological transition of age at Fontan operation. All probability values were two-tailed.
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Results |
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TopAbstractIntroductionPatients and MethodsResultsCommentDiscussionReferences |
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30 days) deaths, 3 patients in the E-E group and 3 in the non–E-E group (p = not significant). Patient demographics and morphologic variables for the E-E and non–E-E groups are listed in Table 1. Atrioventricular valve repair was performed at the time of preparatory operation before cavopulmonary anastomosis in 18, cavopulmonary anastomosis in 21, interim operation between cavopulmonary anastomosis and Fontan in 3, Fontan in 9, and after Fontan in 2. The repair method chosen at each stage is listed in Table 2.
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2, could be inserted into both orifices. In 3 patients, two commissures of the ipsilateral side were closed, leaving a single orifice on the contralateral side. In the first case of this series, a circular annuloplasty was performed around the left-sided orifice. The procedures utilized for common AV valve repair are summarized in Figures 1 and 2.
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In the E-E group, more patients had severe degree of valve regurgitation (13 patients = 59.1%) compared with the non–E-E group (10 patients = 32.3%) before operation (p = 0.0906). The postoperative degree of regurgitation was mild or less in 21 patients (95.5%) of the E-E group compared with 15 patients (48.4%) of the non–E-E group (p = 0.0003). Tricuspid or AV valve stenosis was not observed in any of the patients after repair.
The degree of regurgitation on follow-up echocardiogram was mild or less in 17 patients (77.3%) of the E-E group at 2.6 ± 4.0 years after repair, compared with 12 patients (38.7%) of the non–E-E group at 1.9 ± 3.2 years (p = 0.109). However, there was no difference in the degree of regurgitation or the need for reoperation in the long term between the two groups. Actuarial percentage of patients having AVVR mild or less was 56.6% at 5 years, 42.5% at 5.8 years, and remained unchanged thereafter in the E-E group. It was 36.4% at 2.4 years and remained unchanged for as long as 12.7 years in the other group (p = 0.0215; Fig 5). Reoperation for AVVR was required in 8 patients, with 4 of them undergoing prosthetic valve replacement. The median interval from initial operation to reoperation was 3.0 (0.1 to 7.9) years, and the median age at reoperation was 5.4 (0.4 to 11.5) years. Freedom from reoperation for AVVR at 10 years was 65.0% in the E-E group and 64.5% in the non–E-E group (p = not significant). The actuarial survival was 75.7% at 5 and 10 years of age and 60.5% at 15 and 20 years of age in the E-E group. It was 75.8% at 5 and 10 years of age, 60.7% at 15 years of age, and 40.4% at 20 years of age in the non–E-E group (p = not significant).
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Comment |
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TopAbstractIntroductionPatients and MethodsResultsCommentDiscussionReferences |
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The affected valve frequently has abnormal morphology, with extensively-reduced leaflet coaptation, making reparative procedure difficult [13]. A variety of techniques of valve repair for tricuspid or common AV valve regurgitation have been reported, including reduction annuloplasty, chordal shortening, partial commissure closure, cleft closure, and others [5–7].
Mitral valve plasty with anchoring of the prolapsing free edge of the anterior leaflet to the posterior leaflet was reported by Alfieri and coworkers [8], and has been used as a reliable and reproducible modification in adult patients (E-E). This technique is also applicable to the tricuspid valve by suturing the free edge of septal and anterior leaflets [14, 15]. Application of this technique to growing children may be problematic because it reduces the effective orifice area. However, patients with functionally single ventricle usually have a significantly dilated annulus. In fact, AVV stenosis was not seen in any of our series undergoing E-E.
The technique of E-E repair for common AV valve regurgitation has been previously reported by our group [16], and involved the suturing of superior and inferior bridging leaflets. Similar techniques have been reported from other centers with excellent postoperative reduction in the amount of regurgitation. Using this technique for common AV valve associated with right atrial isomerism, Van Son and colleagues [17] reported no recurrence of regurgitation with a follow-up of as long as 17 months. This confirms the findings of this report, which demonstrates the efficacy of the E-E technique in preventing AVV valve regurgitation for several years after operation. Successful management of AVVR and reduction in volume work accomplished during staged reconstruction is reported to be associated with preserved ventricular function at the time of Fontan operation [5, 6].
Our long-term evaluation also showed that moderate or greater degree of AVVR develops after E-E in a similar percentage of patients when compared with the non–E-E group at 5.8 years. Three patients in the E-E group and 5 patients in the non–E-E group required reoperation for AVVR, with no significant difference in freedom from reoperation in the long term.
It had been our policy in the past to perform Fontan completion at an older age, with the mean age of 10.4 years at the beginning of this study period. Also, a single-stage Fontan operation (without interim cavopulmonary anastomosis) was more common. A staging operation, in which volume reduction is achieved by performing a cavopulmonary anastomosis during infancy, is a common practice in the current era [18]. Following this general trend, Fontan completion is currently performed in our center at 3 years of age or younger in the majority of cases, with a mean age of 2.8 years at the end of this study period. Our results show that durability of E-E repair may not be as long lasting in the setting of pulmonary overcirculation. However, with the current strategy, encompassing a shorter period when the ventricle can carry an excess volume load as a result of parallel pulmonary and systemic circulation, the incidence of recurrent regurgitation and reoperation might be reduced.
Some patients may have a dysmorphic AV valve with severe regurgitation present during infancy, and valve replacement becomes inevitable. Even in this clinical scenario, it is in our opinion of enormous significance to maintain competence of AV valve and preserve ventricular function for several years after birth, as an adult-sized prosthesis can usually be implanted by 2 to 3 years of age in patients with functionally single ventricle. Mahle and coworkers [19] reported that AV valve replacement among children younger than 2 years of age carried a 66% mortality, whereas mortality among children older than 2 years was 8%. The technique described herein might be especially effective in improving survival of patients with severe abnormality of the AV valve, lowering the operative risk at the time of valve replacement.
In conclusion, E-E was an effective adjunct in repairing tricuspid or common AV valve associated with functionally single ventricle. An excellent reduction of regurgitation was observed in most of the patients, and that may lead to preservation of ventricular function during the interim stage and successful completion of Fontan operation.
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Discussion |
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TopAbstractIntroductionPatients and MethodsResultsCommentDiscussionReferences |
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We’ve had a number of patients where they had excellent repair at the time of surgery, but then 2, 3 years down the road, they’ve just had moderate or severe because it’s a common AV valve. It’s a big annulus.
DR JEFFREY P. JACOBS (St. Petersburg, FL): I myself have no experience with that. I wonder, has anybody in the room done anything like that? Doctor Mayer?
DR JOHN E. MAYER (Boston, MA): Well, that is actually what I’ve begun doing in the last couple of years for particularly the heterotaxy type hearts with a complete common AV canal/AV valve morphology, and that is to sort of think about it in the same way that we do with a common AV valve in a Down’s with two ventricles, and that is to try to put something in to fix the distance between the most anterior part of the annulus and the posterior part of the annulus back by the coronary sinus when it’s there.
Actually, what I’ve done is also not only close what would be the equivalent of the cleft with an edge-to-edge type repair, but also to actually put a piece of Dacron, sort of like a ventricular septal defect (VSD) patch, but just put it in the area under the cleft so that then there’s a—essentially you’re limiting the excursion of the AV valve sort of in and out of the ventricle, if you will. It’s almost like putting a cord—or a set of cords—in there, except it’s a little piece of patch.
The experience is small, and the short-term results have been good. But I think whether or not this will help solve this annular dilation and late development or redevelopment of AV valve regurgitation I think remains to be seen.
DR ANDO: Yes. Actually, I have one question for Dr Mayer. Do you put the stitch on the annulus if you put the strip on the leaflet?
DR MAYER: No. I would put it in just in the same way that Dr Backer described putting his VSD patch in except I would just sew a piece, usually a strip of Dacron, just up through the leaflets and then up through the Dacron. So it’s just like you were putting an ASD patch in except that it has no height.
DR ANDO: I think it’s a very fascinating technique, but I think in order for this technique to be more effective, I think it’s better to fix the annulus by the strip because otherwise you—
DR MAYER: Yes. No, I think that’s right. I mean, at the most anterior and posterior parts, you’re actually coming up through—
DR ANDO: So then the problem will be the AV block when you—especially if you have a heterotaxy. Sometimes you cannot tell the exact—
DR MAYER: Right. But see, in that area, you’re still just sewing in leaflet, and you can get the leaflet right up next to where it joins into the atrioventricular groove.
DR CHRISTOPHER A. CALDARONE (Toronto, Ontario, Canada): That leads to a question I actually wanted to ask. You didn’t say this, but what was the mode of failure in the edge-to-edge repairs when you inspected the valve afterward? Was it dehiscence of the edge-to-edge approximation or was it new regurgitation in a previously nonregurgitant portion of the orifice?
DR ANDO: Yes. Actually, I had to speculate from the documentations made by prior surgeons. I think there are some patients who have a very dysplastic valve, and this dysplasia of the valve just progresses with time. So even if you can reduce the regurgitation effectively, this patient is going to come back with regurgitation anyway. But if the leaflet is still pliable and have a significant regurgitation, the one thing that’s possible is that the stitches get cut into the leaflet because it’s very fragile. And this mode of failure could have happened in several cases in our series.
DR JOSEPH J. AMATO (Chicago, IL): That was an excellent presentation. I just have a question that perhaps is a non sequitur question. You showed beautiful pictures of the leaflets. Did the substrate or the presence of either a single-papillary muscle or a double-papillary muscle change your technique at all? I always looked at the modification of the leaflets underneath to observe for the number of papillary muscles as to how I was going to repair my valves.
DR ANDO: I didn’t see the case with an obvious single-papillary muscle in this series. But I did see some cases that had a hypoplasia on one side of the leaflet, and at that part, the papillary muscles were fused beneath the valve. And in those cases, as I showed in a slide, we just completely closed that side of the commissures to create a single orifice.
DR SHUNJI SANO (Okayama, Japan): We have also a very similar experience with the edge-to-edge repair of common AV valve. Although the AV valve incompetence was almost none or mild postoperatively, it gradually increased within 2 or 3 years’ time. So in the recent 2 or 3 years, I do put glutaraldehyde-treated pericardium or a small strip of Gore-Tex to reinforce the annulus. As you said, in the heterotaxy patient, complete annuloplasty is not possible owing to abnormal conduction system. So only two thirds of the annulus was plicated. Other than heterotaxy, probably more than 80% of the annulus is plicated with these Gore-Tex strips or glutaraldehyde-treated pericardium. However, we don’t know the long-time follow-up result yet.
DR ANDO: Thank you very much.
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TopAbstractIntroductionPatients and MethodsResultsCommentDiscussionReferences |
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This article has been cited by other articles:
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  J. H. Shuhaiber, S. Y. Ho, M. Rigby, and B. Sethia Current options and outcomes for the management of atrioventricular septal defect Eur. J. Cardiothorac. Surg., May 1, 2009; 35(5): 891 - 900. [Abstract] [Full Text] [PDF]
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 F. Kerendi, Z. B. Kramer, W. T. Mahle, B. E. Kogon, K. R. Kanter, and P. M. Kirshbom Perioperative risks and outcomes of atrioventricular valve surgery in conjunction with Fontan procedure. Ann. Thorac. Surg., May 1, 2009; 87(5): 1484 - 1488. [Abstract] [Full Text] [PDF]
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