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J Thorac Cardiovasc Surg 2007;133:1311-1317
© 2007 The American Association for Thoracic Surgery

Surgery for Congenital Heart Disease

Intermediate-term results of repair for aortic, neoaortic, and truncal valve insufficiency in children

^a Division of Cardiothoracic Surgery, Departments of Surgery and Pediatrics, Primary Children’s Medical Center, and the University of Utah, Salt Lake City, Utah
^b Division of Cardiology, Departments of Surgery and Pediatrics, Primary Children’s Medical Center, and the University of Utah, Salt Lake City, Utah.

Presented at the Thirty-second Annual Meeting of the Western Thoracic Surgical Association, Sun Valley, Idaho, June 21-24, 2006.

Received for publication June 20, 2006; revisions received October 31, 2006; accepted for publication November 6, 2006.

^_* Address for reprints: John A. Hawkins, MD, Cardiothoracic Surgery, Primary Children’s Medical Center, 100 North Medical Drive, Salt Lake City, UT 84113. (Email: jhawkins{at}hsc.utah.edu).

	Abstract

Top Abstract Introduction Patients and Methods Results Discussion Conclusions References

 
Objective: Repair of aortic valve insufficiency is difficult, and durability is relatively unknown in children. This study evaluates the intermediate-term results of repair of the systemic semilunar valve, including the native aortic valve, neoaortic valve (anatomic pulmonary), and truncal valve.

Methods: We reviewed the records of 54 children (aged 2 days to 18 years) who underwent repair of the functional aortic valve for moderate or greater insufficiency from 1991 to 2005. Valve anatomy was tricuspid aortic in 26 patients, bicuspid aortic in 11 patients, tricuspid neoaortic in 9 patients, bicuspid neoaortic in 1 patient, and truncal valve in 7 patients. Multiple surgical techniques were used in most of the 54 patients, including leaflet plication in 17, leaflet repair in 15, commissuroplasty in 32, pericardial cusp augmentation in 8, and sinus of Valsalva reduction in 3.

Results: There was 1 early death and no late deaths. Actuarial freedom from reoperation was 68% at 5 years and 58% at 10 years. Freedom from aortic valve replacement was 82% at 5 years and 73% at 10 years. Duration of cardiopulmonary bypass was the most significant risk factor for reoperation with multivariate analysis. Of the 40 patients who have not undergone reoperation, 37 have had follow-up echocardiograms with the latest study (4.5 ± 4.2 years) demonstrating trace to 1+ insufficiency in 23 patients, 1 to 2+ in 12 patients, 2 to 3+ in 1 patient, and 3 to 4+ in 1 patient.

Conclusion: Repair of the insufficient systemic semilunar valve offers acceptable 10-year freedom from reoperation and functional results, and should be considered for most children.

	Introduction

Top Abstract Introduction Patients and Methods Results Discussion Conclusions References

 
The timing of surgical intervention and the optimal surgical approach for insufficiency of the systemic semilunar valve in infants and children are difficult issues. Although repair of aortic insufficiency in adults has gained popularity over the last 10 years, there is limited experience in children and long-term results are unknown. The ability to repair an insufficient valve may be even more important for the growing child to avoid the need to replace or up-size a prosthetic valve or allograft. In addition, valve repair avoids the need for anticoagulation, which can be more difficult to achieve and associated with higher risk of complications in the pediatric population.

This study examines our intermediate-term results with repair of native aortic valves, truncal valves, and anatomic pulmonary valves functioning as systemic neoaortic valves, such as those after the arterial switch for transposition, single-ventricle palliation, or the pulmonary autograft (Ross) procedure. We also sought to determine risk factors for predicting failure of valve repairs.

	Patients and Methods

Top Abstract Introduction Patients and Methods Results Discussion Conclusions References

 
Patients
The database of Cardiothoracic Surgery at Primary Children’s Medical Center and the University of Utah was reviewed to identify children aged less than 18 years who had at least 3+ (moderate to severe) insufficiency of the aortic, truncal, or systemic semilunar valve and underwent operation between July 1991 and January 2006. Of these patients, those who underwent repair of the systemic semilunar valve form the basis for this study. The institutional review board at Primary Children’s Medical Center and the University of Utah approved this retrospective review. Records were reviewed to obtain age, weight, additional cardiac anomalies, valve morphology, operative times, additional procedures, techniques used, presence and degree of left ventricular outflow tract obstruction, and details at reoperation. End points included reoperation on the valve, late valve function defined by echocardiography, and death.

Echocardiography
Complete two-dimensional, Doppler, and M-mode transthoracic echocardiograms were performed preoperatively to evaluate cardiac anatomy, systemic semilunar valve stenosis, and regurgitation. Stenosis was evaluated by measuring the peak instantaneous gradient across the systemic semilunar valve obtained using continuous-wave Doppler interrogation from all available views. Valve regurgitation was graded from trace to 4+ using a combination of previously described measurements, including the ratio of diameter of the regurgitant color Doppler jet to the aortic annulus diameter from the parasternal long-axis view, the ratio of the area of the color Doppler regurgitant jet to the area of the aortic annulus from the parasternal short-axis view, the presence or absence of holodiastolic flow reversal in the descending aorta below the diaphragm, and the presence or absence of left ventricular enlargement measured by M-mode.¹

Additional guidance and assessment of the valve morphology was performed by transesophageal echocardiography (TEE) in the operating room both before and after surgical repair. TEE imaging and color Doppler allowed further assessment of leaflet abnormalities along with localization of coaptation abnormalities and specific areas of regurgitant jets. The short-axis view was routinely used to measure and localize the area of the regurgitant jet for grading purposes and direction of specific leaflet therapies.

Operative Techniques
All patients underwent operation with moderate hypothermia (24°C-28°C) and blood cardioplegia. Cardioplegia was routinely reinfused at 20- to 30-minute intervals, with retrograde or antegrade direct coronary instillation. Techniques used to repair the valve varied according to the anatomy of the valve and the cause of insufficiency. Each valve was inspected to determine the anatomy and cause of insufficiency and the anatomic defects at 3 levels: the annulus, valve leaflets, and sinuses and sinotubular junction. Attempts were made to normalize the anatomy as much as possible at all 3 levels, depending on the anatomic abnormality. Annular dilation with failure of cusp coaptation was corrected by circumferential annuloplasty techniques² or subcommissural plication in the interleaflet triangle to reduce the annulus size and improve coaptation.³ The leaflets were evaluated for size, leaflet damage or defects, and coaptation area. Defects or damage to valve leaflets were repaired by previously described techniques.^4,5 Elongated or prolapsing leaflets were repaired by plication using the technique described by Trusler and colleagues.⁶ Inadequate leaflet tissue was augmented using autologous, glutaraldehyde-treated pericardium.⁷ Sinus enlargement or sinotubular junction enlargement that was thought to be contributing to valve insufficiency was treated with sinus reduction techniques.⁸

Follow-up
All 54 patients underwent follow-up by their individual cardiologist, and information regarding reintervention and survival was obtained on all patients between March and June 2006. Postoperative echocardiography was used to evaluate postoperative results, and identical criteria were used to evaluate valve insufficiency or stenosis as used on preoperative evaluation.

Valve-related reintervention was defined as any reoperation for recurrent or persistent systemic semilunar valve regurgitation. The most common indications for reintervention, determined by the child’s cardiologist, were 3+ systemic semilunar valve insufficiency accompanied by symptoms, systemic ventricular enlargement, or reduced systemic ventricular function.

Statistical Methods
Normally distributed data are presented as mean ± standard deviation. Non-normally distributed data are described as median and range. Survival and event-free survival were calculated and presented in Kaplan–Meier format.⁹ Factors associated with reintervention for late significant valve insufficiency were analyzed in a univariate analysis using Cox’s proportional hazard model.¹⁰ After this univariate analysis, factors that showed a trend toward influencing the need for later reintervention (P < .2) were entered into a multivariate forward stepwise regression analysis. Factors analyzed included age at operation, date of operation, age less than 4 years, valve type, need for additional procedures, anatomic diagnosis, previous or concurrent left ventricular outflow tract surgery, valve anatomy and number of cusps, crossclamp time, cardiopulmonary bypass duration, and repair techniques used.

	Results

Top Abstract Introduction Patients and Methods Results Discussion Conclusions References

 
A total of 93 patients underwent operation and had at least 3+ systemic semilunar valve insufficiency during the study period. This included 39 patients (39/93, 42%) who underwent valve replacement and 54 patients (54/93, 58%) who underwent their first operation for repair of at least 3+ systemic semilunar valve insufficiency and form the basis for this study. Diagnoses for the patients undergoing repair are listed in Table 1. The indication for operation was solely insufficiency of the semilunar systemic valve in 17 patients and a combination of valve insufficiency and other anomalies requiring surgical attention in the remaining 37 patients. The insufficient valve was morphologically a tricuspid aortic valve in 26 patients, a bicuspid aortic valve in 11 patients, a tricuspid neoaortic (anatomic pulmonary) valve functioning in situ in 6 patients, a tricuspid neoaortic valve functioning as an autograft in 3 patients, a bicuspid systemic neoaortic valve functioning in situ in 1 patient, and a truncal valve in 7 patients. Of the 7 truncal valves, 5 were quadricuspid and 2 were tricuspid. Multiple techniques were used in most patients (30/54, 56%) and included commissuroplasty³ in 32, leaflet plication⁶ in 17, leaflet repair^4,5 in 15, cusp augmentation⁷ in 8, circumferential annuloplasty² in 7, and sinus of Valsalva reduction plasty⁸ in 3. Patient age at operation ranged from 4 days to 18 years (mean = 8.5 ± 6.5 years) and included 10 infants less than 1 year of age, 8 patients aged between 1 and 4 years, 11 patients aged between 4 and 10 years, and 25 patients aged between 10 and 18 years.

Thirteen of the 54 patients required reintervention (13/54, 23%) 48 ± 28 months after aortic valve repair. No patient required aortic valve replacement during the same hospitalization after aortic valve repair. Reintervention was for pure systemic semilunar valve regurgitation of 3+ with evidence of left ventricular enlargement and/or symptoms in 11 patients and for mixed aortic insufficiency and aortic stenosis in 2 patients. Both patients with mixed stenosis and insufficiency had aortic insufficiency associated with leaflet avulsion after balloon valvuloplasty for aortic stenosis.⁴ Reintervention consisted of re-repair in 5 patients, aortic valve replacement with a mechanical prosthetic valve in 4 patients, and pulmonary autograft aortic valve replacement (Ross procedure) in 4 patients. There were no early or late deaths after reoperation. The 8 aortic valve replacements occurred 53 + 31 months after repair. The actuarial rate of reoperation for valve-related causes including replacement or re-repair and the actuarial freedom from valve replacement are shown in Figure 1. Overall, reoperation free survival was 98% at 1 year, 68% at 5 years, and 58% at 10 years. Freedom from aortic valve replacement was 100% at 1 year, 82% at 5 years, and 67% at 10 years.

View larger version (16K): [in this window] [in a new window]   Figure 1. Actuarial freedom from reintervention for aortic valve replacement or reoperation for any valve-related cause after first repair of moderate or greater insufficiency of aortic, neoaortic, or truncal valves in 54 children. Patients free of aortic valve replacement (dashed line); patients free of any reintervention, valve replacement, or re-repair (solid line); 67% confidence intervals (error bars). The number of patients (in parentheses) followed at that interval.

Univariate analyses of risk factors associated with reintervention are listed in Table 2. The most significant factors associated with reintervention were longer cardiopulmonary bypass times (P = .013) and year of procedure after 2000 (P = .018). We did not find complex repairs (vs simple repairs), number of valve cusps, anatomic type of valve, anatomic diagnosis, specific repair technique, or age to be an independent predictor of reintervention. Multiple stepwise forward regression analysis showed that only duration of bypass was an independent predictor of reintervention.

	Discussion

Top Abstract Introduction Patients and Methods Results Discussion Conclusions References

Previous reports of repair of aortic insufficiency in children are relatively few and have conflicting results. Hasaniya and associates¹² reported a 47% reoperation rate at 6 years in 21 children after aortic valve repair and did not recommend repair as a viable alternative for children with aortic insufficiency. Tweddell and associates¹³ reported their experience with aortic valve repair in a group of heterogenous patients. Freedom from reoperation at 10 years for the group requiring "complex" repairs (analogous to those in this report) was 44%. Although others have reported long-term results of repair of aortic insufficiency in young patients, most have combined both adult and pediatric patients, making careful analysis of results in the infant and young child difficult.^14-17

The repair of the aortic, truncal, or neoaortic valve needs to be individualized to the specific valve anatomy and disease, but the approach can be standardized to systematically evaluating the annulus, the leaflets, and the sinuses and sinotubular junction.¹⁸ Once the abnormal anatomy is clearly understood at each level, individual techniques can be used to restore valve competence. Annular enlargement is dealt with by commissuroplasty³ or circumferential annuloplasty² to restore a normal or near normal annular size. We have found circumferential annuloplasty to be particularly useful when repairing neoaortic regurgitation after the pulmonary autograft (Ross) procedure.¹⁹ The subsequent circumferential suture line with this technique may inhibit annular growth in the child, and we attempt to minimize this risk by using an absorbable stitch (polydioxanone). Regardless, careful follow-up is needed because long-term growth of the annulus with this technique is unknown and left ventricular outflow tract obstruction may develop. Leaflet abnormalities can be repaired by direct suture, patch, reattachment, or even cusp augmentation with pericardium. Cusp reattachment and repair with pericardium are particularly applicable in patients with aortic insufficiency after balloon valvuloplasty.^4,8 Cusp augmentation is useful for centrally located insufficiency from cusp thickening and retraction, characteristic of rheumatic aortic insufficiency. Cusp augmentation may also be applicable when 1, 2, or all leaflets appear to have an inadequate surface area for coaptation. We have found glutaraldehyde-treated autologous pericardium to work best, but others have found untreated autologous or bovine pericardium to be equally efficacious.^20,21 The immediate results of pericardial augmentation are good, but the long-term durability and its potential for growth are unknown. This approach will allow many, if not most, children to have valve repair rather than replacement. It also has resulted in 5- and 10-year freedom from reoperation rates in our series of 89% and 64%, respectively, comparable to rates reported in adults in a recent review.¹¹

In this study, the length of operation was found to be an independent predictor of the subsequent need for reintervention for systemic semilunar valve insufficiency. Others have found that the amount of residual regurgitation when the patient either leaves the operating room or the hospital is strongly predictive of the need to reoperate.^3,12 We did not analyze this as a risk factor because, in our practice, patients with more than mild insufficiency were returned to bypass and further repair was performed to achieve competence. The longer duration of bypass as a major factor associated with reintervention may reflect more complex repairs that are more likely to fail. It may also indicate that as our experience has grown, we have extended indications for valve repair into an area in which success is not as likely or as durable.

Although non-aortic valves, such as truncal valves, bicuspid valves, or pulmonic valves functioning as neoaortic valves, approach significance (P = .057) as a factor predicting the need for reintervention in the univariate analysis, they were not independent predictors in the multivariate model. No particular technique was associated with reintervention, implying that the specific method of repair is probably not as important as the valve itself for determining the need for reoperation.

There are few reports of operation for neoaortic regurgitation after the arterial switch, and in the largest series to date, 13 of 16 patients had aortic valve replacement and only 3 patients had repair, with 1 failure.²² Despite this, we and others would argue that repair should be attempted in the majority of patients with significant neoaortic insufficiency after the arterial switch, unless there is significant root enlargement and root replacement is warranted.²³ Truncal valve repair has been reported in several small series with mixed results.^24-26 Similarly, others have shown that the neoaortic valve can be repaired after the Ross procedure.^19,27 To our knowledge, repair of anatomic pulmonary valves in a single-ventricle circulation after a Damus–Kaye–Stansel or Norwood-type procedure has not been reported to date. The 3 patients in this series have all been followed less than 2 years, so it is impossible to draw conclusions about long-term durability of the neoaortic valve in the single-ventricle situation, although it should not be significantly different than the situation after the arterial switch procedure for transposition of the great arteries. Despite the unknown durability, we believe that the neoaortic or truncal valve functioning in the systemic circulation should undergo attempts at repair, because our early and mid-term results are promising and there is no option of a pulmonary autograft procedure in these subgroups.

This study is limited by several factors. First, this is a relatively large number of pediatric patients with systemic semilunar valve insufficiency, but the numbers of patients in each diagnostic group are small. Thus, specific recommendations or conclusions regarding the advisability of repair for specific lesions or valve morphology type cannot be made. This heterogeneity and small sample size also make inferences regarding the efficacy of specific valve repair techniques difficult. Although this series represents a relatively large number of patients over a 15-year period, the type of valve considered for repair has evolved over the years. Early in the experience, most patients had aortic insufficiency after balloon valvuloplasty or associated with a ventricular septal defect. More recently, repair has been attempted in any child with systemic semilunar valve insufficiency, including hypoplastic left heart syndrome, post-Ross procedure, truncus arteriosus, rheumatic heart disease, or even a previous valve repair. This change in patient selection along with a shorter duration of follow-up for the more complex cases make long-term evaluation of current techniques and indications difficult. In addition, there is no way to compare the groups who had replacement without an attempt at repair in the early part of the series with those who were routinely repaired toward the end of the series.

	Conclusions

Top Abstract Introduction Patients and Methods Results Discussion Conclusions References

 
We demonstrated a diverse group of pediatric patients with aortic, neoaortic, or truncal valve insufficiency who underwent repair of the existing semilunar valve with acceptable intermediate-term results. An individualized approach tailored to the anatomic abnormalities noted at the annular, valvar, and sinotubular regions can be effective, with less than half the patients requiring reoperation in the decade after repair.

	References

Top Abstract Introduction Patients and Methods Results Discussion Conclusions References

 

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This Article Abstract Full Text (PDF) Alert me when this article is cited Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to Personal Folders Download to citation manager Author home page(s): John A. Hawkins Peter C. Kouretas Richard V. Williams Lloyd Y. Tani Linda M. Lambert Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Hawkins, J. A. Articles by Minich, L. L. Search for Related Content PubMed PubMed Citation Articles by Hawkins, J. A. Articles by Minich, L. L. Related Collections Valve disease Related Article