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J Thorac Cardiovasc Surg 2001;122:162-168
© 2001 The American Association for Thoracic Surgery
Surgery for Congenital Heart Disease (CHD) |
From the Departments of Cardiovascular Surgery,a Cardiology,b and Biostatistics,c Children's Hospital, Harvard Medical School, Boston, Mass.
Received for publication Nov 10, 2000. Revisions requested Dec 13, 2000; revisions received Jan 5, 2001. Accepted for publication Jan 22, 2001. Address for reprints: Richard A. Jonas, MD, Department of Cardiovascular Surgery, The Children's Hospital, 300 Longwood Ave, Boston, MA 02115 (E-mail: richard.jonas{at}tch.harvard.edu).
Abstract
Objective: Aortic regurgitation after balloon dilation of congenital aortic stenosis may be treated with valve repair as an alternative to replacement.
Methods: Charts and echocardiograms of all patients undergoing aortic valve operations after balloon dilation of congenital aortic stenosis at our institution between January 1988 and December 1999 were reviewed.
Results: Twenty-one patients underwent valvuloplasty for predominant aortic regurgitation 9 months to 15 years (mean, 6.1 years) after balloon dilation. The mean ± SD age at the time of the operation was 11 ± 7 years. Aortic regurgitation was caused by a combination of commissural avulsion (10), cusp dehiscence with retraction (9), cusp tear (5), central incompetence (2), perforated cusp (1), or cusp adhesion to the aortic wall (1). Repair techniques included commissural reconstruction with a pericardial patch (8), pericardial patch cusp augmentation (6), primary suture repair (6), raphae release and debridement (4), commissurotomy (4), commissural resuspension with sutures (3), and cusp release (1). There were no deaths. At a mean follow-up of 30.1 months (range, 9 months8 years), all patients were asymptomatic, and the grade of aortic regurgitation had been significantly reduced (P < .001). Left ventricular end-diastolic dimension z scores and proximal regurgitant jet/aortic anulus diameter ratios were significantly reduced (P < .001) and remained so over time. Freedom from reoperation for late failure was 100%, and overall freedom from reintervention was 80% at 3 years.
Conclusion: Aortic valve repair for balloon-induced aortic regurgitation is reproducible and durable at medium-term follow-up.
Significant aortic regurgitation (AR) is a recognized complication after balloon dilation of congenital aortic valvular stenosis (cAVS).
1-5 It has an incidence of 13.2% immediately after balloon dilation, which increases to 38.2% during follow-up in our experience.
5 Given the current use of balloon dilation as the primary approach to cAVS at many centers, including our own, some of these patients now have significant AR. Mechanical valve replacement is complicated by the need for systemic anticoagulation and lack of annular growth. The pulmonary autograft procedure is an alternative to a mechanical prosthesis with potential for annular growth, but disproportionate dilation of the neoaortic root, autograft regurgitation, and the need for pulmonary homograft changes remain concerns.
6-11 The number of patients reported who have undergone a valve-sparing procedure for balloon-induced AR is small.
12-15 We describe our operative approach and efficacy of repair at follow-up in patients with congenital cAVS who have undergone a valve-sparing repair for AR induced by balloon dilation at our institution.
Patients and methods
Methods
All patients who underwent balloon dilation for cAVS between January 1988 and December 1999 were identified through the cardiology catheterization laboratory database. All data procurement was carried out after institutional approval according to guidelines established by the committee on clinical investigation. All charts were retrospectively reviewed up to the last entry, and echocardiograms were read by independent reviewers.
Patients
During the 11-year study period, 335 balloon dilations were performed on 281 patients for cAVS, 59 (21%) of whom eventually required aortic valve operations for residual stenosis, AR, or both(Table 1). A valve-sparing procedure was performed in 21 patients in whom the predominant lesion was AR. These patients form the basis of this report. The mean ± SD age at the time of balloon dilation was 5.1 ± 3.2 years; 4 patients had 2 dilations, and 1 patient underwent 3 such procedures. Postdilation AR, as determined by using angiography, was considered mild in 2 patients, mild to moderate in 11 patients, moderate in 3 patients, and severe in 5 patients. Five other patients underwent valvuloplasties but were excluded from the study group because 4 had predominant aortic stenosis (AS) after unsuccessful balloon dilation, and 1 underwent an associated modified Konno procedure. The remaining 33 patients underwent a Ross procedure with or without the Konno procedure (15), aortic valve replacement (9; homograft: 4; prosthesis: 5), surgical valvotomy (5), or Norwood palliation (4;Table 1
). Four patients had a subaortic membrane, and 3 patients underwent aortic coarctation repair.
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Surgical technique
The operation was performed on cardiopulmonary bypass with moderate systemic hypothermia for all patients. A pericardial patch was harvested and fixed in 0.6% glutaraldehyde for 20 to 30 minutes. The fixation time was chosen to improve the handling and increase the strength of the pericardial patch. A left ventricular vent was placed through the right superior pulmonary vein. An aortotomy was made transversely and extended toward the noncoronary sinus of Valsalva. Cold cardioplegia was infused selectively into both coronary ostia. A decision to perform valve reconstruction was at the surgeon's discretion and was a judgment made on the basis of preoperative echocardiography, intraoperative TEE, and direct inspection of the valve. Anatomic elements necessary for valve repair include a sufficient annular diameter to not require left ventricular outflow tract enlargement, mobile cusps or cusps that can be made mobile by resection or shaving of excess fibrous tissue, and an ability to achieve coaptation without inducing stenosis. A variety of valvuloplasty techniques were used.
12-16 The valve was studied by using fine forceps to approximate the length, depth, and mobility of the cusps. The diameter of the central opening of the valve, as well as the location and mobility of the commissures and raphae, were also noted.
Primary repair
Excess fibrous tissue, which has a tendency to build up around raphae (rudimentary fused commissures), was aggressively removed (so-called shaving), giving the cusp more mobility. Fused commissures with adequate suspension to the aortic wall were opened with a scalpel. Simple tears involving otherwise competent cusps were repaired primarily, usually with a 5-0 polypropylene (Prolene; Ethicon, Inc, Somerville, NJ) running suture. If the native cusps were deemed adequate, tears involving the anterior commissure (right coronary commissure [RCC]-noncoronary cusp commissure [NCC]) in bicuspid valves were repaired by resuspending the commissure with sutures passed through the aortic wall. Otherwise, the commissure was resected and reconstructed. Prolapsed but otherwise competent and pliable cusps were shortened by resuspension of the cusps to the commissures with pledget-supported sutures. In cases of central cusp incompetence with dilation of the sinuses of Valsalva, a sinus of Valsalva reduction plasty was performed to reduce commissural splaying. This was done by resecting a wedge of noncoronary sinus, followed by primary closure of the aortotomy(Figure 1).
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Results
Preoperative data
The mean age at the time of the operation was 11 ± 7 years (range, 9 months15 years). The mean time between the last balloon dilation and the operation was 6.1 ± 4.5 years. Five patients had decreased exercise tolerance. Severe AR was present in 14 patients, and moderate AR was present in 7 patients (median grade = severe). The mean maximum instantaneous gradient was 43 ± 26 mm Hg. Significant left ventricular dilation was present in 11 patients (LVEDD z score >3).
Intraoperative data
Description of aortic valves
The valve was bicuspid in 19 patients, tricuspid in 1 patient, and unicuspid in 1 patient. Fusion of the left coronary commissureRCC was predominant in 15 (71%) patients. Fusion of the RCC-NCC was present in 4 patients, and fusion of both the left coronary commissureRCC and RCC-NCC was present in 1 patient. Predominant factors contributing to AR were a combination of anterior commissural avulsion in 10 patients, cusp dehiscence with retraction (presumably caused by long-standing balloon-induced tear) in 9 patients, simple cusp tear in 5 patients (anterior in 4 patients and posterior in 1 patient), central incompetence in 2 patients (from calcified cusps in 1 patients and from sinus of Valsalva dilation in 1 patient), a perforated cusp in 1 patient, and free cuspal edge adhesion to aortic wall in 1 patient. The RCC was the most frequently involved cusp (20/21 patients).
Aortic valve reconstruction
The various techniques used are summarized in Table 2. No patients required immediate conversion to aortic valve replacement after failed valvuloplasty. Cardiopulmonary bypass was reinstituted in 2 patients after the initial valvuloplasty. In 1 patient this was because of severe pericardial cusp patch prolapse with resulting AR managed by adding a second pericardial patch to the opposing cusp, thereby improving coaptation with resultant trace AR. In the other patient there was evident AS after valve debridement and primary repair of a tear for which anterior and posterior commissurotomies were carried out, with reduction of the peak systolic ejection gradient from 55 to 35 mm Hg.
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At further follow-up, echocardiograms continued to reveal a significant reduction in AR grade (median AR grade = mild, P < .001;Figure 5). Mean LVEDD z scores were also significantly reduced (3.44 ± 1.97 preoperatively vs 1.54 ± 2.27 at immediate follow-up, P = .003; 3.44 ± 1.97 preoperatively vs 1.00 ± 1.73 at late follow-up, P < .001;Figure 6), as were proximal AR jet diameter/aortic anulus diameter ratios (0.53 ± 0.18 preoperatively vs 0.17 ± 0.13 at immediate follow-up, P < .001; 0.53 ± 0.18 preoperatively vs 0.28 ± 0.17 at late follow-up, P < .001). For both LVEDD z scores and AR jet diameter/aortic anulus diameter ratios, immediate follow-up values were not significantly different from late follow-up values (P = .84 and P = .09, respectively). The freedom from increased AR (see "Methods" section) was 100% at 2 years and 75% at 3 years(Figure 7). Preoperative Doppler maximum instantaneous gradient and that at last follow-up did not change (43 ± 26 vs 43 ± 21 mm Hg, P = .95;Figure 8). None of the variables listed above were significantly associated with increased AR in the Cox model (P > .5 for each).
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Balloon dilation has replaced surgical valvotomy as the primary treatment for cAVS in most centers, often with excellent relief of stenosis.
1-5 The present report is the largest series reported to date dealing specifically with balloon-induced AR in cAVS. Several series, including some intraoperative human studies,
17,18 have shown that after successful balloon dilation, new AR is more common than residual AS.
1-3,12 Typically, the balloon-dilated bicuspid aortic valve tears near or at the anterior commissure, with the RCC being most frequently involved. In this study, as in others,
17,18 most patients had an anterior lesion involving the RCC, and AR was the predominant lesion. Although when to surgically intervene in these children is a controversial decision, accepted indications are the presence of symptoms, increasing significant LV dilation with or without dysfunction, or both in the setting of moderate or worse AR.
Aortic valve repair versus pulmonary autograft
Growing children would benefit if the balloon-damaged aortic valve could be repaired in a reproducible and durable fashion, rather than being replaced.
15 Prosthetic valves usually require anticoagulation and reoperations and have a small risk of endocarditis. The initial enthusiasm for the pulmonary autograft procedure, currently accepted as the best choice for replacing the aortic valve in children,
6 has been dampened by clinical,
7,11,19 as well as experimental,
8 observations of early dilation of the neoaortic root, sometimes leading to significant regurgitation.
9,10 Although freedom from homograft replacements after the Ross procedure has been greater than 85% at 8 years,
10 homografts undergo more rapid degeneration in the very young child.
20 Elkins and colleagues
10 showed that intra-aortic pulmonary autograft valve replacements tended to follow a normal diameter increase, whereas root replacements tended to dilate. Because the latter technique is usually used in children, progressive dilation remains a serious concern.
Deciding whether to replace or repair an aortic valve remains a difficult task, largely influenced by preoperative and intraoperative 2-dimensional and 3-dimensional echocardiography,
21 as well as direct inspection. Another factor that needs to be accounted for when trying to decide whether to replace or repair is the size of the aortic anulus and the degree and mechanism of AS. In the present cohort aortic valve repair did not result in increased AS. The preoperative aortic anulus z scores were in the range of 0.9 to 2.4. On the basis of these data, it is our clinical impression that long-standing AR-induced volume load will promote growth of the often hypoplastic anulus of a congenitally stenotic aortic valve, thus allowing for a subsequent repair with less risk of postoperative AS. A patient with mixed severe AS and AR might benefit more from a pulmonary autograft than a repair, despite an operative mortality of 4% with the root replacement.
10 If the decision that a valve can be repaired could be made with certainty preoperatively, asymptomatic children with moderate AR and no left ventricular dilation could undergo valvuloplasty in a proactive effort to prevent further myocardial damage.
22
We currently use any increase in LVEDD z scores as an indication for operation in asymptomatic children. A recent report on 22 valvuloplasties
14 noted that repair of a regurgitant bicuspid aortic valve was significantly less effective than a pulmonary autograft replacement. This is in contradistinction with the results of this report, in which bicuspid or unicuspid valves were present in 91% of patients. After a mean follow-up of 31 months, only 1 patient has progressed to severe AR, with no change in LVEDD z score. A longer period of follow-up was not associated with increased AR. Only 3 patients with increasing AR at follow-up also have increased LVEDD z scores, suggesting that aortic valve repair did reliably prevent or reduce further myocardial dilation in the majority of patients.
Type of repair
Repair of insufficient bicuspid valves, albeit not balloon-damaged valves, was addressed specifically in 2 recent reports.
16,23 The described repair techniques are similar to ours but are more designed to deal with redundant prolapsing cusps. Balloon-damaged valves often exhibit a localized tissue defect at the site of the previous tear, making it amenable to reconstruction with pericardial patch extension. One recent report
12 focused exclusively on balloondamaged valves. Repair techniques included only primary repair and debridement performed an average of 35 months after balloon dilation, whereas the patients in the present study were operated on an average of 69 months after balloon dilation. The free cusp edge of the balloon-torn valve has a tendency to roll onto itself, retracting and becoming fibrotic over time. This would explain why pericardial patch augmentation was used in a much larger scale in our cohort and why AS was not a problem postoperatively. The pericardial patch has to counteract the inherent shallowness of the congenitally stenotic valve by giving it more depth (Spinnaker effect).
21 We have not seen the complication of development of AS caused by crowding of the ventriculoaortic junction with the augmented cusps described in a recent study,
15 which is perhaps related to our less extensive use of patch augmentation.
Conclusion
This study suggests that aortic valve repair for balloon-induced AR can be recommended. It is reproducible and durable at medium-term follow-up in the majority of patients.
Acknowledgments
We thank John E. Mayer, Jr, MD, and Pedro J. del Nido, MD, for allowing us to include their patients in this study.
Footnotes
Part of this work was done while Dr Moran was the Tommy Kaplan Fellow in the Children's Hospital Cardiovascular Program. ![]()
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