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Ann Thorac Surg 1998;65:1758-1762
© 1998 The Society of Thoracic Surgeons
a Divisions of Pediatric Cardiothoracic Surgery and Cardiology, Departments of Surgery and Pediatrics, Primary Childrens Medical Center and the University of Utah, Salt Lake City, Utah, USA
Accepted for publication January 22, 1998.
Address reprint requests to Dr Hawkins, Pediatric Cardiothoracic Surgery, Primary Childrens Medical Center, 100 N Medical Dr, Salt Lake City, UT 84113
e-mail: (jhawkins{at}med.utah.edu)
| Abstract |
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Methods. From 1986 to 1996, 37 infants in the first 3 months of life underwent open aortic valvotomy for critical aortic stenosis. All patients underwent cardiopulmonary bypass, valvotomy, and valve debridement under direct vision with standard techniques.
Results. Early mortality was 11% (4 of 37, 70% confidence limit 7% to 20%) and all early deaths were in neonates less than 2 weeks of age. Late death occurred in 6 patients a mean of 10 ± 12 months (range, 2 to 36 months) after valvotomy. Actuarial survival, including operative deaths was 92% ± 6% at 1 month, 78% ± 9% at 1 year, and 73.4% ± 10% at 10 years. In a multifactorial regression analysis, the best predictors of death were the presence of endocardial fibroelastosis and small body surface area and the best predictor of the need for late reintervention was preoperative aortic annular size. Thirteen patients required reintervention: repeat operation in 7 patients, balloon valvuloplasty in 3 patients, and both balloon valvuloplasty and reoperation in 3 patients. Actuarial freedom from reintervention postoperatively is 97% ± 3% at 1 month, 73% ± 9% at 1 year, and 55% ± 11% at 10 years. Reintervention was for recurrent left ventricular outflow obstruction in 9 patients and mixed aortic stenosis and aortic insufficiency in 4. Echocardiography 4.3 ± 2.5 years after aortic valvotomy in survivors who have not required reintervention (n = 20) revealed a Doppler peak instantaneous systolic gradient of 37 ± 14 mm Hg and mild or less aortic regurgitation in 16 patients and moderate aortic regurgitation in 4 patients.
Conclusions. Current surgical results with critical aortic stenosis in the neonate and young infant are acceptable in terms of both late survival, reintervention, and functional results in the majority of patients. Newer interventions, such as balloon valvuloplasty, should be carefully evaluated for long-term results and should be compared more appropriately to current surgical results to determine the best treatment modality for the neonate and infant with critical aortic stenosis.
| Introduction |
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Our approach to the neonate and small infant with critical AS over the years has been almost exclusively with open surgical valvotomy. The purpose of this study was to take advantage of our relatively uniform approach and examine late functional results and reintervention after surgical valvotomy. This will allow a reference point for comparison of newer interventions for critical aortic stenosis such as balloon valvuloplasty, transventricular closed valvotomy, and neonatal pulmonary autograft procedures.
| Patients and methods |
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90 days of age who underwent open aortic valvotomy at Primary Childrens Medical Center between January 1986 and December 1996. The only patients excluded from this study were those that had preoperative recognition of left ventricular (LV) hypoplasia sufficient to preclude a biventricular repair and patients who underwent primary balloon aortic valvuloplasty. In general, if the patient had both an aortic valve annulus diameter less than 5 mm and a calculated LV end-diastolic volume less than 20 mL/m2 [14], then the patient did not have an attempt at biventricular repair with a valvotomy. During the time period of this study a total of 8 additional patients less than 90 days of age underwent primary balloon aortic valvuloplasty for critical AS. Six of these patients were chosen for primary balloon valvuloplasty early in the total experience and at the discretion of the attending cardiologists, with 4 survivors. Two additional patients underwent balloon valvuloplasty because of an annulus size of 4 mm or less and adequate LV volume and function. Both of these patients suffered a dissection of the ascending aorta and underwent urgent ascending aortic repair and aortic valvotomy. These two patients subsequently died 24 and 43 days postoperatively of chronic low cardiac output states with multisystem organ failure. A total of 37 consecutive patient records were reviewed for the 11-year study period. Twenty-four of the patients were in the first month of life and 13 patients were between 1 and 3 months of age at operation. The mean age at operation was 26 days and the median age at operation was 14 days (range, 2 to 86 days). Other patient characteristics are included in Table 1. Patients with severe hypoplastic left heart structures were generally selected for univentricular repair, but there were 3 patients with an aortic annulus less than 6 mm, 4 with a mitral valve diameter less than 8 mm, 4 with an LV ejection fraction less than 0.20, and 5 patients with an LV end-diastolic volume less than 20 mL/m2. All patients underwent preoperative two-dimensional and Doppler echocardiography and many early patients underwent preoperative cardiac catheterization. Operation was undertaken if the infant had critical AS as defined by ductal dependency or congestive heart failure with severe symptoms.
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Postoperative follow-up was obtained in all patients by direct visits with attending cardiologists. Complete two-dimensional and Doppler echocardiograms were obtained in all survivors. Aortic valve gradient was estimated using continuous wave Doppler interrogation from multiple views. Postoperative aortic regurgitation was evaluated by color Doppler mapping and graded as mild, moderate, or severe according to previously described methods [15]. Echocardiographic indices were determined based on the preoperative echocardiogram and included determination of LV volume, aortic annulus and root diameters, mitral valve area, LV mass and mass/volume ratio, LV shortening fraction and ejection fraction, and end-systolic wall stress. Reintervention was defined as any reoperation or balloon aortic valvuloplasty and the earliest reintervention was chosen when 1 patient had both operative and balloon aortic valvuloplasty reintervention.
Statistical methods
Normally distributed data are presented as mean ± standard deviation. Nonnormally distributed data are described as median and range. Survival and event-free survival are calculated and presented by the method of Kaplan and Meier [16]. Coxs proportional hazard model was used in both a single factor and multifactorial analysis of survival and event-free survival (freedom from reintervention) [17]. Factors analyzed for effect on survival and event-free survival included aortic annulus size, preoperative Doppler LV outflow gradient, mitral valve area, presence of mitral insufficiency LV end-diastolic dimension, LV end-diastolic volume, LV mass, LV ejection fraction, age at operation, end-systolic wall stress, presence of echocardiographic evidence of endocardial fibroelastosis, and presence of coexisting cardiac anomalies (exclusive of patent ductus arteriosus).
| Results |
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| Comment |
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The early mortality rate for surgical aortic valvotomy has ranged between 9% and 59%, even in recent years [18]. The mortality rate in our series is certainly quite low by older standards, but undoubtedly there is some degree of selection that has occurred over the years as we have learned which patients have ventricular size and volume sufficient to undergo valvotomy rather than Norwood reconstruction or transplantation. We found small body surface area and the presence of echocardiographic evidence of endocardial fibroelastosis to be the strongest predictors of mortality, either early or late. This is in agreement with other studies that have found small patient size or young age and endocardial fibroelastosis [8, 19] to be predictors of death. We did not find LV volume to be an independent predictor of death as others have [20], but this is likely due to the fact that we did not have many patients with small LV volumes who underwent this attempt at a biventricular repair and the relative weakness of ventricular volume as a definitive predictor in this entity [20]. Kitchiner and associates [7] found a small aortic annulus to be a predictor of an adverse outcome as we found it to be a predictor of late reintervention. The late survival in this study was good with a 73% survival at 10 years, including operative mortality. This compares favorably to the late survival reported by others using surgical valvotomy with Messmer and colleagues [5] reporting a 10-year survival of 78% and Gildein and colleagues [8] reporting a 5-year actuarial survival of 66%, both inclusive of operative mortality. Others have examined late survival rates for neonates and infants after surgical valvotomy reporting 10-year survival of 70% to 100% (exclusive of operative deaths), but with early deaths ranging from 36% to 59% [4, 7, 3].
Reintervention after surgical aortic valvotomy was predictably high in this study (13 of 33 survivors). Actuarial freedom from reintervention for hospital survivors was 68% at 5 years and 55% at 10 years, almost identical to those reported by Karl [6] and Gildein [8] and their colleagues. Perhaps surprisingly, more than 68% of hospital survivors are free of any adverse event (death, reintervention, endocarditis) at 5 years and about half are alive and free of any reintervention at 10 years (Fig 2). These numbers are remarkably similar to those reported by others including Messmer [5], Karl [6], Gildein [8], Elkins [21], and Kitchiner [7] and their colleagues. Like others, we also found that reintervention was largely for recurrent LV outflow tract obstruction and only rarely for severe aortic regurgitation [3, 8]. In most patients with little or no aortic insufficiency we have opted to proceed to balloon aortic valvuloplasty as the first reintervention. If the patient has moderate or severe aortic valve insufficiency or if there is a significant degree of subaortic stenosis, we have proceeded to surgical intervention. Over the years our surgical reintervention method has changed almost exclusively to the Ross or Ross/Konno procedure [2123] rather than allograft or prosthetic valve insertion. We have found this to be most reliable, of relatively low risk, and perhaps the best long-term alternative for the growing infant or child who requires operative reintervention after a valvotomy in infancy.
This study was instituted specifically to examine our results so we could carefully examine the late results of surgical aortic valvotomy and make an educated decision regarding the institution of balloon aortic valvuloplasty as the procedure of choice for neonates and infants with critical AS. The 30-day procedure mortality for balloon aortic valvuloplasty has been reported to be in the range of 9.6% to 40%, but has reached low rates in recent years [13]. Although long-term results of balloon aortic valvuloplasty for older infants and children have been well described, late results for balloon valvuloplasty in neonates and young infants have been lacking. BuLock and associates [24] demonstrated an intermediate survival of 50% at 3 years in a series of 14 infants less than 2 months of age at the time of balloon aortic valvuloplasty. In another large study of infants <30 days of age, Egito and associates [13] demonstrated survival of 63% a mean of 4.3 years after balloon valvuloplasty. When patients with a Rhodes score of 2 or greater were excluded from their series, survival improved to 88% at 8 years. In the large Valvuloplasty and Angioplasty of Congenital Anomalies study, age less than 3 months was a strong predictor of early and late mortality, but the actuarial results were not really delineated [12]. As in surgical series, LV hypoplasia and endocardial fibroelastosis appear to be important determinants of survival after balloon valvuloplasty [25]. It is difficult to compare directly the two modalities as the results of both operation and valvuloplasty are so affected by patient selection and favorable anatomy and it is often difficult to discern from an individual series how much patient selection is present [26].
Surgical valvotomy offers some advantages over both balloon aortic valvuloplasty and transventricular surgical valvuloplasty. Open aortic valvotomy offers the ability to inspect directly the valve and incise the commissures accurately and under controlled circumstances. It also offers the ability to resect dysplastic tissue or nodules, which we believe may contribute to a favorable long-term outcome by more completely relieving LV outflow tract obstruction. Indeed, a more extended aortic valvuloplasty has been reported to offer excellent results in some young infants [27] and our exact technique has been successfully used previously by others treating infants with critical AS [5, 6, 8]. Balloon valvuloplasty or transventricular valvotomy is a relatively uncontrolled situation where the tear may occur incompletely or even through one cusp, resulting in an unsatisfactory result with respect to residual LV outflow obstruction or aortic insufficiency [18].
In this study we had a relatively high rate of valve replacement with a prosthetic valve or Konno type reconstruction of the LV outflow tract. This reflects partially the time period and our own past preference for prosthetic valve insertion over allograft valve replacement. Certainly, since 1993, we have exclusively chosen pulmonary autograft insertion when aortic valve replacement is needed and the valve cannot be repaired or salvaged. In general, our approach to the first reintervention after aortic valvotomy is balloon aortic valvuloplasty if the reason for reintervention is primarily recurrent stenosis and aortic insufficiency is less than moderate. If balloon valvuloplasty fails we have then proceeded to pulmonary autograft insertion rather than repeat attempts at balloon valvuloplasty, as others have [13]. If the patient has moderate or greater aortic valve insufficiency we would proceed directly to a pulmonary autograft insertion. Although the long-term efficacy of pulmonary autograft insertion in infants is not proven, we consider it the best choice for aortic valve replacement in the growing child [21].
In summary, we consider aortic valvotomy still to be a valuable tool in the treatment of neonates and young infants with critical AS. The long-term results with respect to both survival and reintervention are good and compare favorably with other forms of neonatal congenital heart disease. We would consider surgical valvotomy the standard for comparison, despite the success of recent reports of balloon aortic valvuloplasty [13] and transventricular aortic valvotomy [2]. Neonates and young infants of small size or with evidence of severe endocardial fibroelastosis are not good candidates for surgical valvotomy and may be better candidates for conversion to a univentricular physiology or cardiac transplantation. Only with time, careful follow-up, and perhaps a randomized trial will we be able to determine accurately which therapysurgical valvotomy, balloon aortic valvuloplasty, transventricular valvotomy, or even neonatal pulmonary autograftis the best long-term solution to critical AS in the neonate and young infant.
| References |
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