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Eur J Cardiothorac Surg 2004;26:54-59
© 2004 Elsevier Science NL

Repair of atrioventricular valve regurgitation in the modified Fontan operation

Ahmad Sallehuddin*, Ziad Bulbul, Frank Otero, Khaled Al Dhafiri, Zohair Al-Halees

King Faisal Specialist Hospital and Research Centre, Riyadh 11211, Saudi Arabia

Received 1 November 2003; received in revised form 17 February 2004; accepted 22 March 2004.

* Corresponding author. Tel.: +966-1-442-7470; fax: +966-1-442-7482
e-mail: asallehuddin{at}kfshrc.edu.sa


   Abstract
Top
 Abstract
1. Introduction
 2. Materials and methods
 3. Results
 4. Discussion
 5. Conclusions and limitations
 References
 
Objective: To evaluate the prevalence, impact and outcome of repair of atrioventricular valve regurgitation (AVVR) in Fontan patients. Methods: We retrospectively reviewed 340 Fontan patients from 1986 to 2001. Twelve patients with valve closure or replacements were excluded. AVVR was graded by transthoracic echocardiography. Patients were divided into group O (no AVVR), group A (1+ to 2+) and group B (3+ to 4+). Results: AVVR was present in 129 (39.3%) patients. Repair was carried out in 37 (11.3%) with no difference in mortality as compared to no repair (18.9 vs 10.9%, P=0.16). Mean follow-up was 44 months (1–197 months) with 14 patients lost to follow-up. No valve repair was carried out in group O (192 patients) and no clinical change in AVVR was observed (pre-op 0.00, post-op 0.78). In group A (85 patients) 6 patients had valve repair without significant change in the degree of AVVR after valve repair (pre-op 1.8, post-op 2.2, P=0.18). In group B (44 patients) 31 had valve repair and a significant reduction was observed (pre-op 3.28, post-op 2.44, P<0.001). A similar improvement was also observed when the valve was not repaired (pre-op 3.25, post-op 2.44, P=0.003). Survival at 10 years was comparable (83% repaired, 89% not repaired, P=0.165). There was no difference in the incidence of long-term complications (26% repaired, 29.7% no repair, P=0.64). Conclusions: Trivial to mild AVVR remains stable and their repair during the Fontan operation provides no additional benefits. Valve repair in patients with moderate to severe AVVR improved the regurgitation with comparable operative mortality and long-term outcome; however, similar benefits could be achieved without repair of the atrioventricular valve. We should not deny patients with similar AVVR the Fontan operation.

Key Words: Fontan operation • Total cavo-pulmonary anastomosis • Atrioventricular valve regurgitation • Atrioventricular valve repair • Heart defects • Congenital


   1. Introduction
Top
 Abstract
 1. Introduction
2. Materials and methods
 3. Results
 4. Discussion
 5. Conclusions and limitations
 References
 
Atrioventricular valve regurgitation (AVVR) is a known perioperative risk factor of the modified Fontan operation [1] and has been considered as one of the factors contraindicating the procedure [2]. AVVR is nonetheless concomitantly repairable during the Fontan operation at a low risk [3]. With increasing numbers of patients at high risk being subjected to the Fontan operation, including patients with significant AVVR, it is important to know the results of atrioventricular valve repair in these patients. In this review we sought to determine the prevalence of AVVR in a large number of Fontan patients at our institution. In particular we wish to evaluate the impact of atrioventricular valve repair on the degree of regurgitation and assess the long-term outcome of these patients following the procedure. By evaluating the stability of the repair we attempt to define the group of patients who would or would not benefit from the procedure.


   2. Materials and methods
Top
 Abstract
 1. Introduction
 2. Materials and methods
3. Results
 4. Discussion
 5. Conclusions and limitations
 References
 
We reviewed the medical records, electrocardiograms, operation notes, cardiac catheterization and echocardiography reports of 340 consecutive patients undergoing the modified Fontan operations from 1986 to 2001 at King Faisal Specialist Hospital and Research Centre, Riyadh, Saudi Arabia. Six patients had previous atrioventricular valve repair during a staged bidirectional cavopulmonary shunt (BCPS). Twelve patients who had closure or replacement of the atrioventricular valve were excluded. This population of 328 patients ranges in age from 5.3 to 203.8 months (mean 50.8±34.7) with 138 females and 190 males. Four patients were less than 12 months of age and 51 patients less than 2 years old (15.5%). There were no patients of more than 18 years in age.

There were 211 patients with dominant left and 92 patients with dominant right ventricles. The ventricular morphology was indeterminate in six patients. The modified Fontan operation was also performed in 19 patients with biventricular hearts wherein the intra-cardiac anatomy did not allow for a biventricular repair. The distributions of malformations are shown in Table 1 .


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  		Table 1. Distribution of malformations

 
Pre-operative echocardiography was not available in seven patients. The remaining 321 patients available for evaluation were divided into three groups based on the degree of AVVR prior to the Fontan operation. Group 0 were patients without any AVVR, group A with AVVR grades 1+ to 2+, and group B with 3+ to 4+. The types of atrioventricular valves in each of the three groups are shown in Table 2 . There was a statistical difference in the probability of the valve types being repaired (P=0.001), where 35% of the valves repaired were common atrioventricular valves.


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  		Table 2. Types of atrioventricular valves and distribution between groups

 
2.1. Surgical technique
All procedures were carried out on moderate hypothermia (32 °C) facilitated by periods of low-flow perfusion or deep hypothermic circulatory arrest (16 °C) as needed. Intermittent cold blood cardioplegia with topical hypothermia were used for myocardial protection. Two main variations of the modified Fontan operation were used namely direct atriopulmonary anastomosis and total cavopulmonary connection (TCPC). The latter included the lateral tunnel modification and the total extracardiac cavopulmonary connection. The lateral tunnels were fashioned mainly from autologous atrial wall re-arranged in a reversed Senning's method, while the extracardiac tunnels were created from polytetrafluoroethylene grafts of 18–22 mm in diameter.

Fenestrations in the Fontan partitions were not routine. They were created in patients with relative contraindications of age less than 2 years old, mildly elevated pulmonary vascular resistance and the repair of AVVR.

The atrioventricular valves were tested by saline injection into the ventricles and the decision to repair was based on the degree of these intra-operative leaks as well as the pre-operative echocardiographic findings and the surgeon's assessment of the reparability of the lesions. Valves were repaired when there were signs of long-standing and significant regurgitation. These include thickened leaflet edges, endocardial jet lesions and annular dilatation. Following repair the atrioventricular valves were re-tested both intra-operatively as well as with transoesophageal echocardiography after separation from cardiopulmonary bypass.

2.2. Chordal repair
Elongated chordae were shortened by folding and burying their lower ends into trenches made in their respective papillary muscles. The buried ends were held down with pledgetted 5-0 polypropylene sutures. The trenches were then closed with pledgetted 4-0 polyester sutures. This was carried out in four patients. More recently and with the introduction of polytetrafluoroethylene sutures (Goretex, WL Gore and Associates, Inc., Flagstaff, AZ), the repair by chordal replacements using these sutures was preferred (two patients). No chordal transfer procedures were carried out.

2.3. Leaflet repair
Simple clefts in the atrioventricular valves were repaired using interrupted 5-0 polypropylene sutures that were secured in a figure-of-eight fashion. This was carried out in three patients who had common atrioventricular valves. In the presence of regurgitation adjacent to commissures, the leaflet edges in these areas were approximated using one or two interrupted 4-0 polyester sutures. This was carried out in six patents. None of the patients had any forms of leaflet resection performed.

2.4. Annuloplasty
Conventional Kay annuloplasty was carried out at the commissures using pledgetted 4-0 polyester sutures. This annuloplasty were carried out in four patients with double-inlet and one with absent-right atrioventricular valves. Purse-string annuloplasties were done using 3-0 polypropylene suture passed in a counter-clockwise direction as a double circumferential stitch deep into the junction of the leaflet and the annulus. These purse-string sutures were not completely encircling to avoid injury to the conduction tissue and the area between the posterior insertion of the interventricular septum and the posterior leaflet of the right atrioventricular valve was spared. The purse-string annuloplasty was performed in five patients with common and two with absent-left atrioventricular valves. Ring annuloplasties were not performed in any of the patients.

In addition, four patients had a combination of annuloplasty and leaflet repair. All these patients had common atrioventricular valves. Data on the type of repair were not available in 14 patients.

2.5. Echocardiographic evaluation
A pediatric cardiologist who was blinded to whether the patient had undergone atrioventricular valve repair and to the previous echocardiograms carried out the echocardiographic evaluation. All patients underwent two-dimensional echocardiography.

2.6. Statistical analysis
The {chi}2 square test was used to compare categorical variables and the t-tests for continuous variables. Logistic regression analysis was carried out to evaluate risk factors of operative mortality and the Kaplan–Meier curve to evaluate the late survival rates. Significant results will be reported at type I error rate of 5%. The SAS statistical system software was used throughout.


   3. Results
Top
 Abstract
 1. Introduction
 2. Materials and methods
 3. Results
4. Discussion
 5. Conclusions and limitations
 References
 
The demographic and operative variables of patients with or without atrioventricular valve repair are compared in Table 3 . There were no major differences in the two groups except in four pre-operative variables. The pulmonary artery pressure and pulmonary vascular resistance were both higher in the group with atrioventricular valve repair. The majority of patients in this group also had lesions other than tricuspid atresia and a higher proportion had undergone a staged BCPS prior to the Fontan operation. Despite the addition of a concomitant atrioventricular valve repair, the perfusion and ischemic times were not significantly prolonged as compared to the isolated Fontan procedure.


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  		Table 3. Table 3 Demographic and operative variables of all patients classified into those with and without atrioventricular valve repair

 
3.1. Prevalence of atrioventricular valve regurgitation and its influence on outcome
Pre-operative AVVR was present in 129 patients giving a prevalence rate of 39.3% in our Fontan patients. Thirty-seven patients underwent repair of the atrioventricular valve. The overall mortality rate was 11% (n=36). The mortality of patients with AVVR was comparable to those without AVVR (12.7 and 10.6%, respectively, P=0.57). The mean follow-up time was 44 months (range 1–197 months) with 14 patients lost to follow-up. There were seven late deaths (2.4%). The survival curves following the modified Fontan operation are depicted in Fig. 1 . The 10-year survivals for patients with and without AVVR were 84 and 90%, respectively (P=0.165, Log rank test).



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  		Fig. 1. Kaplan–Meier estimated overall survival function of patients with and without atrioventricular valve regurgitation.

 
3.2. Influence of valve repair on atrioventricular valve regurgitation
There were 192 patients in group O and none had valve repair. In group A there were 85 patients where 6 had valve repair, and in group B there were 44 patients where 31 patients had valve repair. The operative mortality rate in patients with repair was 18.9%, which was not significantly different from those without atrioventricular valve repair (10.9%, P=0.16).

Table 4 shows the change in grades of AVVR. There was a slight increase in the degree of regurgitation in group O from 0 to 0.78. The majority of patients in this group (152/192, 80%) had the lateral tunnel type of modified Fontan operation. The increase in grade of AVVR following the lateral tunnel Fontan in this sub-group was from 0 to 0.71 (P<0.001). Following the other forms of the modified Fontan operation, the increase was similarly significant and therefore it was arguable that the lateral tunnel modification could be implicated for the increased regurgitation.


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  		Table 4. Pre-operative and post-operative degrees of atrioventricular valve regurgitation

 
In group A the grade of AVVR remained stable despite the lack of repair in a large proportion of patients. In group B however, there was a significant improvement in the degree of regurgitation overall (from 3.27 to 2.27, P<0.001). This reduction was nonetheless seen regardless of the surgical repair of the regurgitant valves (Table 4).

3.3. Influence of atrioventricular valve repair on outcome
Patients who had repair of the atrioventricular valve required more days on mechanical ventilation (5.2 vs 1.9, P=0.001) as well as in the intensive care unit (9.4 vs 4.7, P=0.001). The total length of hospital stay nonetheless was not prolonged in spite of the repair (15.6 vs 14.9, P=0.83). Table 5 shows several short- and long-term complications common to Fontan patients and in spite of repair of the atrioventricular valve these complications were not any more prevalent. On further analysis of the subset of patients with moderate to severe AVVR (group B), we were unable to elicit any significant differences in the duration of ventilation, length of ICU and hospital stays, as well as in the incidence of late complications of pleural effusions, protein losing enteropathy and dysrhythmias between those who had their valves repaired and those who had not.


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  		Table 5. Early and late complications in patients with or without atrioventricular valve repair

 
By univariate analysis the risk factors significant for operative mortality were perfusion time, mean pulmonary artery pressure, pulmonary vascular resistance, biventricular hearts and ventricular filling pressures. Using stepwise logistic regression analysis, only perfusion time and mean pulmonary artery pressure were significant risk factors for operative mortality. Neither the presence of AVVR nor the repair of atrioventricular valve was a risk factor of operative mortality in our present study.

Fig. 2 shows that the 10-year survival of patients following atrioventricular valve repair was 83% and was comparable to 89% for patients with AVVR that was not repaired (P=0.165, Log-rank test). The incidence of late complications of arrhythmias and protein losing enteropathy were not significantly different during the same period of follow-up as shown in Table 4.



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  		Fig. 2. Kaplan–Meier estimated overall survival function of patients with and without repair of their atrioventricular valve regurgitation.

 
3.4. The role of bidirectional cavopulmonary shunt
There were 173 patients who had prior BCPS and 85 of these had AVVR before the Fontan operation. Twenty-nine of these patients had eventually undergone repair of their atrioventricular valves during the subsequent modified Fontan operation. It is expected that a BCPS would reduce the volume load on the single ventricle and thus improve the AVVR. Mahle and colleagues [4] concluded that atrioventricular valve repair is not justified in all patients with moderate AVVR undergoing the BCPS. Only 16.7% (29/173) of the patients who had prior BCPS eventually required atrioventricular valve repair in our study. These patients nonetheless represented 78% of all patients who required atrioventricular valve repair. This suggests that the incompetence of these valves is structural in origin and the impact of volume reduction of BCPS bears little influence in their status. Three out of six patients with previous repair of their atrioventricular valve during BCPS needed a second repair during the Fontan operation. From this experience we can deduce that the presence of significant AVVR early on should not deter us from proceeding with the plan for a single ventricle repair. Although some patients may still require further atrioventricular valve repair, the outcome will be fairly acceptable.

3.5. The role of fenestrations
Fenestrations were created in 47 patients (14.2%). One-third (29%) of patients less than 2 years of age had a fenestration compared to only 12% in older patients (P=0.002). The mean pulmonary vascular resistance in patients with fenestration was 2.21 Wood units compared to 1.52 Wood units in those without (P=0.04). Of those patients who had repair of the atrioventricular valve, 29.7% were fenestrated while only 12.4% of patients without repair had a fenestration (P=0.008). We observed no correlations between the use of fenestrations and operative mortality or late outcome.


   4. Discussion
Top
 Abstract
 1. Introduction
 2. Materials and methods
 3. Results
 4. Discussion
5. Conclusions and limitations
 References
 
In the modified Fontan operation, AVVR is a risk factor of operative mortality [2] rendering a high operative mortality rate. The mortality rates of patients with AVVR (12.7%) and of patients undergoing concomitant atrioventricular valve repair (18.9%) in our series were well within the rates experienced by others [5]. Although some have found a significant difference in the mortality rates of patients with AVVR as compared to those without [3], our experience suggests otherwise. Additionally the concomitant repair of the atrioventricular did not significantly alter the mortality rate.

We observed a progressive deterioration in the competency of the atrioventricular valve following the Fontan operation in patients without any AVVR prior to the operation. The degree of deterioration was however small (from 0 to 0.78,). The majority of these patients in our study had the lateral tunnel modification that could hypothetically distort the atrioventricular valve and induce regurgitation. The deterioration was nonetheless similarly observed in patients that had other modifications of the modified Fontan operation and we could not positively attribute the deterioration to the type of Fontan procedure carried out.

In patients with trivial to mild regurgitation this deterioration was however unaffected by the surgical correction of the atrioventricular valves. Although Imai and associates [3] had suggested that repair of the atrioventricular valve should be offered to all similar patients undergoing the Fontan operation, our observation suggests that this is not entirely necessary.

On the other hand patients with moderate to severe regurgitation achieved a significant reduction in the degree of regurgitation following surgical repair. This suggests that neither the presence nor the severity of the AVVR should absolutely contraindicate the Fontan operation. It is interesting nonetheless that the patients who were not repaired within the same group had also shown improvement in their regurgitation grade. Further analysis of this small group of patients revealed no significant differences in outcome. It is therefore likely that the benefits achieved from surgical repair of the atrioventricular valve could have been attained even if the repair was omitted. This is of particular significance in circumstances where cross clamping of the aorta is avoided by choice (e.g. off-pump or extracardiac Fontan on beating heart) and the repair of the atrioventricular valve may be left out with no differences in outcome.

There was a noticeable discrepancy between the pre-operative echocardiographic grading of AVVR and that of the intra-operative evaluation. Six patients in group A had mild AVVR but were repaired while 13 patients in group B had moderate to severe AVVR but were not repaired. This had resulted from the intra-operative decision to repair that was based additionally on the signs of significant regurgitation. These signs were not detectable by the pre-operative echocardiography. Furthermore the reparability of the valves may only be properly assessed at surgery by direct visual examination of the physical structure of the valve.

The data regarding the technical aspects of the valve repair in our patients was unfortunately inadequate. We were unable to perform meaningful analysis due to this deficiency. This was a crucial deficit of this report wherein the proper surgical approach could not be determined for these atrioventricular valves that are not the same as those in patients undergoing biventricular repairs.

Although the addition of valve repair must logically add to the ischaemic time, we have found otherwise (see Table 3). There were 33 patients who had an extracardiac TCPC and in 8 of these patients were done on a beating heart. Despite exclusion of the latter patients from the analysis we have found the ischemic time to remain comparable between the repaired and the non-repaired groups (65.1 vs 59.6 min, P=0.26).

The patients with atrioventricular valve repair in this study needed a longer period of ventilatory support and also more days in the intensive care unit. This had not come as a surprise as their mean pulmonary artery pressure and pulmonary vascular resistance were higher. These adverse pre-operative conditions could be attributed to, if not accentuated by, the AVVR itself. Nonetheless with no difference in the rate of pleural effusions and related complications, the overall hospital stay was not significantly different form their counterparts that did not undergo atrioventricular valve repair.

The incidence of late complications and the overall survival were not different in patients with repair of the atrioventricular valve as compared to their counterparts without repair. While others had found that the presence of AVVR was a risk factor for the development of supraventricular arrhythmias [6,7] our data indicated no difference in the overall incidence of arrhythmias. Perhaps the impact of the repair could have reduced the influence of the regurgitation in this regard.


   5. Conclusions and limitations
Top
 Abstract
 1. Introduction
 2. Materials and methods
 3. Results
 4. Discussion
 5. Conclusions and limitations
References
 
We conclude that trivial and mild AVVR remains stable and their repair during Fontan operation provides no additional benefits. Valve repair in patients with moderate to severe AVVR improved the regurgitation with comparable morbidity and mortality rates as well as long-term survival; however, these benefits could well have been achieved despite the repair. We should not deny patients the Fontan operation based neither on the presence nor the degree of AVVR, as long as there are no other absolute contraindications.

This was a retrospective review and the selection process was not randomized. The groups were not free from selection bias as the choice of intervention depends on the degree of AVVR as well as the surgeons' preference. The repair techniques were also not standardized and not evaluated in full. From the outset, some patients with very severe degrees of AVVR may not have been referred for surgery at all and therefore this study group could not be representative of every patient with the problem.


   Acknowledgments
 
We acknowledge the contributions of Dr Mohamed Shoukri and Ms Sunita Bhatia from the department of Biostatistics, Epidemiological and Scientific Computing, King Faisal Specialist Hospital and Research Center in providing the statistical analysis for this study.


   Footnotes
 
Presented at the joint 17th Annual Meeting of the European Association for Cardio-thoracic Surgery and the 11th Annual Meeting of the European Society of Thoracic Surgeons, Vienna, Austria, October 12–15, 2003.


   References
Top
 Abstract
 1. Introduction
 2. Materials and methods
 3. Results
 4. Discussion
 5. Conclusions and limitations
 References
 

  1. Driscoll D.J., Oxfford K.P., Feldt R.H., Schaff H.V., Puga F.J., Danielson G.K. 5–15 year follow-up after Fontan operation. Circulation 1992;85:469-496.[Abstract/Free Full Text]
  2. Choussat A., Fontan F., Besse P., Vallot F., Chauve A., Bricaud H. Selection criteria for Fontan procedure. In: Anderson R.H., Shinebourne E.A., eds. Pediatric cardiology. Edinburgh: Churchill Livingstone, 1978:559-566.
  3. Imai Y., Takanashi Y., Hoshino S., Terada M., Aoki M., Ohta J. Modified Fontan procedure in 99 cases of atrioventricular valve regurgitation. J Thorac Cardiovasc Surg 1997;113:262-269.[Abstract/Free Full Text]
  4. Mahle W.T., Cohen M.S., Spray T.L., Rychick J. Atrioventricular valve regurgitation in patients with single ventricle: impact of the bidirectional cavopulmonary shunt. Ann Thorac Surg 2001;72:831-835.[Abstract/Free Full Text]
  5. Caspi J.C., Coles J.G., Rabinovich M., Cohen D., Trusler G.A., Williams W.G., Wilson G.J., Freedom R.M. Morphological findings contributing to a failed Fontan procedure. Circulation 1990;82(Suppl IV):IV-177-IVhyphen182.[Medline]
  6. Durongpisitkul K., Porter C.J., Cetta F., Offord K.P., Slezak J.M., Puga F.J., Schaff H.V., Danielson G.K., Driscoll D.J. Predictors of early- and late-onset supraventricular arrhythmias after Fontan operation. Circulation 1998;98:1099-1107.[Abstract/Free Full Text]
  7. Stamm C., Friehs I., Mayer J.E., Zurakowski D., Triedman J.K., Moran A.M., Walsh E.P., Lock J.E., Jonas R.A., del Nido P. Long-term results of the lateral tunnel Fontan operation. J Thorac Cardiovasc Surg 2001;121:28-41.[CrossRef][Medline]



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