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J Thorac Cardiovasc Surg 1994;107:1114-1120
© 1994 Mosby, Inc.

SURGERY FOR CONGENITAL HEART DISEASE

Extended aortic valvuloplasty for recurrent valvular stenosis and regurgitation in children

Chicago, Ill.

From The Heart Institute for Children, Christ Hospital and Medical Center, and Rush-Presbyterian-St. Luke's Medical Center, Chicago, Ill.

Address for reprints: Michel N. Ilbawi, MD, The Heart Institute for Children, Christ Hospital and Medical Center, 4440 West 95th St., Oak Lawn, IL 60453.

Abstract

Recurrent significant aortic valvular stenosis or regurgitation, or both, after balloon or open valvotomy in pediatric patients often necessitates aortic valve replacement. In an attempt to preserve the aortic valve, we performed extended aortic valvuloplasty in 21 children with recurrent aortic valve stenosis or regurgitation from January 1989 to March 1993. Previous related procedures were one open aortic valvotomy or more (n = 15), balloon valvotomy (n = 4), balloon valvotomy after surgical valvotomy (n = 1), and repair of iatrogenic valve tear (n = 1). Mean age at the time of the extended aortic valvuloplasty was 6 ± 3.4 years. Mean pressure gradient across the aortic valve was 56 ± 12 torr. Regurgitation was moderate (grade 2 to 3) in nine and severe (grade 4) in 12 patients. Extended aortic valvuloplasty techniques consisted of thinning of valve leaflets (n = 15), augmentation of scarred and retracted leaflets with autologous pericardium (n = 11), resuspension of the augmented leaflet (n = 14), release of the rudimentary commissure from the aortic wall (n = 5), extension of the valvotomy incision into the aortic wall on both sides of the commissure (n = 20), patch repair of the sinus of Valsalva perforation (n = 1), reapproximation of tears (n = 5), and narrowing of the ventriculoaortic junction (n = 2). No operative deaths occurred. The postoperative mean pressure gradient, assessed by most recent Doppler echocardiography or cardiac catheterization at a follow-up of 18 ± 6 months, was 19 ± 6 torr (p < 0.01 versus the preoperative gradient). Aortic regurgitation was absent in 13, mild in 6, and moderate-to-severe, necessitating subsequent aortic valve replacement, in 2. This short-term experience indicates that extended aortic valvuloplasty is a safe and effective surgical approach that minimizes the need for aortic valve replacement in children with significant recurrent aortic valve stenosis or regurgitation. (J THORAC CARDIOVASC SURG 1994;107:1114-20)

Several surgical procedures have been used for the treatment of children with congenital aortic stenosis, including open aortic valvotomy, ¹ closed transventricular dilation of the aortic valve, ² and balloon valvotomy. ³ However, the long-term results remain unsatisfactory, with residual valvular stenosis or regurgitation necessitating subsequent valve replacement in the majority of cases. ^4-6

Recently, a new technique of extended aortic valvuloplasty as the initial approach for management of congenital aortic stenosis in children was described. ⁷ Relief of the obstruction was better and less residual valvular regurgitation was observed with the new approach than with standard valvotomy. On the basis of that experience, the same technique in conjunction with various other valvuloplasty procedures was applied in children with significant recurrent aortic valvular stenosis or regurgitation, or both, who otherwise would have required valve replacement.

This study presents our experience with extended aortic valvuloplasty techniques for management of recurrent or residual abnormalities. It analyzes the efficacy of the described techniques in preserving native valve function and deferring valve replacement.

PATIENTS AND METHODS

Between January 1989 and March 1993 the records of all children who required reoperation for recurrent aortic valvular stenosis or regurgitation, or both, at The Heart Institute for Children were reviewed. Patients with associated intracardiac lesions were excluded. Twenty-one patients are included in this study. All patients had two-dimensional echocardiograms with Doppler studies and cardiac catheterization before the operation. Previous procedures included one open aortic valvotomy or more (n = 15), balloon valvotomy as the initial intervention (n = 4), balloon valvotomy for restenosis after surgical valvotomy (n = 1), and repair of iatrogenic valve leaflet tear after closure of ventricular septal defect (n = 1).

Indications for reoperation included a peak systolic aortic valve gradient greater than 50 torr in 13 patients and the presence of moderate-to-severe aortic regurgitation resulting in progressive increase in left ventricular dimensions in 21 patients.

The degree of aortic regurgitation was graded as mild when the regurgitant jet reached 25% of the distance from the valve to the left ventricular apex, moderate when the jet reached 50% of the distance, and severe when the jet reached more than 50% of the distance.

Structure of the aortic valve at operation.
The valve was bicuspid in 19 patients and tricuspid in two patients. The valve leaflets displayed various degrees of dysplasia, thickening, and myxomatous nodularities. Fused commissures were present in 20 patients. Lack of cusp coaptation was the predominant factor contributing to aortic regurgitation in 16 patients. In five patients the free edge of the cusp was torn near the commissure. One patient had a cusp perforation. The left ventriculoaortic junction was dilated in two patients and supravalvular narrowing was present in three patients.

Surgical techniques.
The operation was performed with cardiopulmonary bypass with ascending aorta and bicaval cannulation. A left ventricular vent was introduced through the right upper pulmonary vein and systemic moderate hypothermia (28° C) was used in all patients. After aortic crossclamping, the aorta was opened with an oblique incision extending into the noncoronary sinus of Valsalva. Cold blood cardioplegic solution was selectively infused into the coronary ostia in all patients, combined with retrograde cardioplegia in two patients.

Extended valvotomy.
The fused commissures were incised to the ventriculoaortic junction and the incision was extended in a circumferential fashion beyond and above the commissures into the aortic wall around the insertion of the leaflet for a distance of 1 to 2 mm on both sides, splitting the aortic wall into two layers, as previously described (n = 20) (Fig. 1, A). ⁷ The incision was also deepened into the muscle tissue underneath the commissure between the right and left leaflets (Fig. 1, B). A small cube of muscle tissue was resected at that area in four cases to free tethered cusps. Myxomatous nodularities and areas of leaflet thickening were thinned out to decrease the bulk obstructing the orifice and to improve mobility and coaptation (n = 16) (Fig. 1, C). The rudimentary commissure was released from its attachment to the aortic wall by splitting of the aortic wall in that area for a distance of 3 to 5 mm (n = 5) (Fig. 1, D). In three patients, the narrowed supravalvular area was enlarged with a diamond-shaped polytetrafluoroethylene patch (Fig. 1, E).

View larger version (108K): [in this window] [in a new window]   Fig. 1. Details of extended aortic valvuloplasty in children. A, The fused commissures are incised and the incision is extended in a circumferential fashion beyond and above the commissures into the ventriculoaortic junction. B, The incision is deepened in the muscle underneath the commissure between the right and left leaflets.C, Myxomatous nodules and areas of leaflet thickening are thinned out. D, Releasing of the rudimentary commissure.E, Enlargement of the supravalvular area with a polytetrafluoroethylene patch.

View larger version (84K): [in this window] [in a new window]   Fig. 2. Details of valve reconstruction. A, Reapproximation of tears in the free edge of the cusp. B, The retracted and scarred leaflet is augmented with autologous pericardium and then resuspended to the aortic wall with pledget-supported fine sutures. C, Patch repair of sinus of Valsalva perforation. D, Narrowing of ventriculoaortic junction with a 2-0 polypropylene suture, which is superficially placed underneath the valve in a circumferential fashion. The suture is tightened while the degree of leaflet coaptation is assessed. Great care is taken to avoid subvalvular narrowing.

Follow-up and statistical methods.
All patients were examined for signs and symptoms of heart failure. Two-dimensional echocardiography with color Doppler studies were performed before discharge and every 6 months after the operation. Data are expressed as mean ± standard deviation and are analyzed by means of Student's t test. A p value less than 0.05 was considered significant.

RESULTS

Mean age at the time of valvuloplasty ranged between 1.8 and 13 years (mean 6 ± 3.4 years). The interval from previous operation or balloon valvotomy to present intervention ranged from 9 months to 9 years (mean 5 ± 2 years). Preoperative mean pressure gradient across the aortic valve was 56 ± 12 torr. Regurgitation was moderate (grade 2 to 3) in nine patients and severe (grade 4) in 12 patients.

No operative deaths occurred, but one patient had a large pericardial effusion 2 weeks after the operation and required pericardiocentesis.

Follow-up, ranging from 2 to 51 months (mean 18 ± 6 months), was available on all patients. Echocardiographic evaluation demonstrated an increase in valve orifice and improvement in cusp coaptation in all patients. Postoperative mean pressure gradient, assessed by the most recent continuous wave Doppler study, was 19 ± 6 mm Hg (p < 0.01 versus the preoperative value) (Fig. 3). The aortic valve was competent in 13 patients and mildly incompetent in six patients. Fig. 4 depicts the change in severity of valvular regurgitation. In all patients, echocardiography and Doppler study showed no progression of valve stenosis or incompetence over the time of follow-up. The actuarial freedom from valve replacement after extended aortic valvuloplasty is 90% at 51 months (Fig. 5). Two patients had significant residual aortic stenosis and regurgitation necessitating aortic valve replacement 6 and 8 months after the valvuloplasty procedure. One patient with hypoplastic ventriculoaortic junction and failed cusp augmentation underwent aortoseptal enlargement (Konno) with insertion of a 21 mm Medtronic Hall prosthetic valve (Medtronic, Inc., Minneapolis, Minn.) at 6 years of age. In the other patient the valve was replaced with a 20 mm Medtronic Hall prosthetic valve at age 7.5 years after failure of cusp extension and resuspension.

View larger version (88K): [in this window] [in a new window]   Fig. 3. Comparison of preoperative and postoperative peak systolic aortic valve gradients after extended aortic valvuloplasty as measured by Doppler echocardiography. Part of the decrease in peak pressure gradient may be due to reduction in the degree of regurgitation and resultant decrease in stroke volume.

View larger version (86K): [in this window] [in a new window]   Fig. 4. Scheme showing the postoperative change in the degree of aortic valve regurgitation in 21 children after extended aortic valvuloplasty.

View larger version (28K): [in this window] [in a new window]   Fig. 5. Actuarial analysis of freedom from reoperation. After 51 months 90% of all patients (n = 21) had not required valve reoperation (AVR).

Recurrent aortic valve stenosis or regurgitation is common after open valvotomy or balloon dilation in pediatric patients. ^5,7-9 The combination of pressure and volume overload in these patients is associated with a poor prognosis. ¹⁰ Repair of the aortic valve for the management of this residual abnormality has not received much attention, especially in children. Most patients with this combination were treated with valve replacement, especially when regurgitation was the principal lesion. ⁵ This approach was justifiable because of lack of reliable techniques that could minimize stenosis and restore competence and because of the consistent results obtained from valve replacement. Awareness of the drawback of valve replacement and experience obtained from repair of the atrioventricular valves, as well as the good results of aortic valvuloplasty in adults with rheumatic heart disease, ¹¹ stimulated interest in aortic valve repair rather than replacement in the pediatric age group.

Failure of previously used techniques to restore competence and minimize stenosis after balloon dilation or commissurotomy procedures stems from incomplete understanding of the underlying disease and the salient morphologic features of the aortic valve that influence the surgical approach in this condition. These features have come into focus as experience has evolved in repairing these valves.

One feature is the eccentric opening of the congenitally stenotic valve. This valve opening is limited, even after an adequate commissurotomy. Extension of the incision beyond the commissure into the aortic wall and to both sides of the valve cusp, as performed in this series, augments the valve orifice considerably. In the early experience, the incision was extended for several millimeters on both sides and might have contributed to aortic regurgitation in the postoperative period. However, as more experience was gained, the incision has been limited to 1 to 2 mm on both sides of the commissure, and with this revision, incompetence has been rendered less prevalent.

Another feature of congenital valvular stenosis is the crowding of the cusps at the commissures and their tethering to the immediate subvalvular area. Cautious undermining of cusp attachment to the ventriculoaortic junction and release of any tethering tissue in the subcommissural area improves mobility of the cusps and further enlarges the orifice. This approach has been aggressively applied only after realization that no true aortic valve anulus exists ¹² and that undermining the cusp does not result in its detachment.

A shallow valve cusp is a third characteristic feature of the congenitally stenotic valve, and in some cases one or two cusps are almost flat plates with a shallow cavity. Release of rudimentary commissure fusion, a procedure frequently used, results in an unsupported shallow cusp, at least on one side of the orifice. This cusp tends to prolapse into the ventricular cavity and has poor coaptation with the opposite cusp. The techniques used in this series address the problem. Release of the rudimentary commissure from the aortic wall without perforating the cusps allows stretching of the cusp tissue adjacent to the aortic wall and increases its depth. More important, however, is augmentation of the shallow cusp with a piece of pericardium and resuspension of its edges to the aortic wall at a level that provides depth to the cusp and good coaptation with the other leaflet. The pericardial patch used for this purpose should be taut enough to prevent prolapse of the leaflet into the ventricular cavity during diastole or to obstruct the coronary ostium during systole. A pericardial patch has been used frequently to resuspend the leaflet in the later part of this experience. The durability of the pericardium in this setting has been questioned in the past, and results were poor when large pieces were used to replace a complete cusp. More recently, several authors have used glutaraldehyde treated bovine or calf pericardium with encouraging long-term results. The recent report of Chachques and associates ¹³ concerning the advantages and clinical use of glutaraldehyde-treated autologous pericardium in the mitral position encouraged its use in this series of patients. Obviously, longer follow-up is needed to assess the durability of autologous unstented pericardium in the aortic valve position.

Another problem frequently present in patients with recurrent regurgitation and stenosis is a supravalvular ridge, which can limit free leaflet motion and decrease systole. This supravalvular ridge is probably part of the pathologic spectrum of this lesion. It is also exaggerated by a previous aortotomy, especially when performed in the neonatal period for the initial open valvotomy. In a few of these cases, enlargement of that area by cutting across the stenotic ridge and placement of a patch can increase the space available for free motion of the leaflets.

Valvuloplasty technique also has to address yet another morphologic problem, namely, thickening and nodularities of the aortic valve leaflets. Thinning of these cusps and aggressive removal of myxomatous nodules helps to increase the mobility of the leaflet and decreases the extent of obstruction.

The data from this experience suggest that the short-term results of valvuloplasty techniques in children with recurrent stenosis or regurgitation are encouraging. Avoiding valve replacement in this age group is advantageous and allows an earlier and aggressive attempt at treating residual stenosis or regurgitation, thus decreasing the deleterious effects of long-standing pressure and volume overload. However, the approach is evolving and long-term results are needed to evaluate its efficacy in avoiding, or at least delaying, aortic valve replacement.

Appendix: DISCUSSION

Dr. Serafin Y. DeLeon (Maywood, Ill.).
I admire Dr. Caspi and his colleagues for their attempt to preserve the aortic valve in these children, but I have some reservations about this procedure.

Going around the aortic commissure seems to make sense because the opening during systole will be a lot bigger. Because the structure of the cusp is preserved, apposition is expected during diastole. However, in some cases, probably because of hemodynamic changes, the cusps no longer appose at the commissural level. Additionally, lengthening and redundancy of the valve occurs, leading to prolapse and further insufficiency.

We have seen on echocardiography the gap at the commissure and redundancy of the cusps producing insufficiency not only at the commissure but also throughout the whole valve. We have already encountered three patients in whom significant aortic insufficiency developed: two required valve replacement and the remaining patient is being observed because of young age.

Dr. Caspi, please address my concerns. Can you suggest some modifications that we can use so that insufficiency will be minimized in these children?

Thomas L Spray (St. Louis, Mo.).
You have been using intraoperative echocardiography to assess the value of your repair. Have there been times when the initial repair was unsuccessful and you reestablished bypass and then modified the repair? If so, what kind of anatomy was there when that was required?

Second, in the two patients who had severe regurgitation that did not improve with repair, was there something unusual about the anatomy?

Dr. Caspi.
Thank you for the comments. Dr. DeLeon, this is an evolving technique. We published some of our early experience a couple of years ago. At that time, we were more liberal with the incision above the commissure and initially extended it for at least 3 or 4 mm around the commissure. This long incision may be the reason that aortic regurgitation developed in some of your patients later on. In our recent experience, which is included in this study, we limit our incision to only 1 or 2 mm at the most. This approach has been shown to decrease the prevalence of postoperative valvular incompetence.

Furthermore, we check the valve routinely after the repair. If there is evidence of aortic valve regurgitation, we go a step further and augment the leaflet with a small piece of pericardium, as I described.

Dr. Spray, we never needed to reinstitute bypass and redo the procedure. Actually, two patients required aortic valve replacement. Follow-up in those two patients revealed severe regurgitation. In addition, one of them had a small ventriculoaortic junction and underwent a Konno procedure.

Footnotes

Read at the Seventy-third Annual Meeting of The American Association for Thoracic Surgery, Chicago, Ill., April 25-28, 1993.

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