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Ann Thorac Surg 1997;63:1713-1717
© 1997 The Society of Thoracic Surgeons

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Original Article: Cardiovascular

Clinical Outcome and Left Ventricular Function After Pulmonary Autograft Implantation in Children

Departments of Cardiopulmonary Surgery, Pediatric Cardiology, and Cardiology, University Hospital Rotterdam, Rotterdam, the Netherlands

Accepted for publication December 24, 1996.

	Abstract

Top Footnotes Abstract Introduction Material and Methods Operation Follow-up Echocardiography Statistical Analysis Results Echocardiography Comment Acknowledgments References

 
Background. Aortic root replacement with a pulmonary autograft is an alternative treatment for children with aortic valve or root disease, or both.

Methods. Twenty-six patients (18 boys and 8 girls) with a mean age of 10.9 years (range, 0.3 to 16.9 years) underwent this procedures in a 7-year period. The mean follow-up period was 3.2 years (range, 0.2 to 7.5 years).

Results. During follow-up 3 patients died and one autograft was replaced with a mechanical valve. The actuarial survival and actuarial event-free survival rates were 87% and 79%, respectively, at both 5 and 7 years. None of the surviving patients had complaints, and all have done well and are living normal lives. Electrocardiographic signs of myocardial ischemia and left ventricular hypertrophy were not present. Echocardiography showed autograft valve regurgitation to be absent or trivial (n = 17) or mild (n = 5). Stenosis was not present. Increasing autograft annulus diameters were noted during follow-up, but this was not related to the severity of autograft regurgitation. Left ventricular dimensions and function were within normal limits later than 1 year after the operation. Only 2 patients had a moderate pulmonary stenosis without right ventricular hypertrophy.

Conclusions. The surgical results, clinical outcome, valve function, and left ventricular function in our patients have been good. This procedure is recommended as a method of aortic valve replacement in children.

	Introduction

Top Footnotes Abstract Introduction Material and Methods Operation Follow-up Echocardiography Statistical Analysis Results Echocardiography Comment Acknowledgments References

 
The use of the pulmonary autograft for aortic root replacement has become a well-established treatment for aortic valve and root disorders in children [1–3]. The advantages of this technique are obvious: there is no risk of thromboembolism, no need for anticoagulants, and no immunologic degeneration of the autograft; perhaps most importantly, the autograft has growth potential [1, 4], thereby avoiding the need for reoperation in the growing child. However, the root replacement technique is a technically less demanding technique than the implantation of a pulmonary autograft within the aortic root (intraaortic cylinder or subcoronary implantation) [5]. We reported our initial experience with this aortic root replacement technique in children in 1992 [3]. The present study incorporates a larger follow-up period and thus an improved view of the midterm results with regard to clinical outcome, left-sided and right-sided pulmonary allograft function, and the increase in the diameter of the pulmonary autograft. A further goal of this study was to evaluate the results of the pulmonary autograft procedure with regard to clinical outcome and left ventricular function.

	Material and Methods

Top Footnotes Abstract Introduction Material and Methods Operation Follow-up Echocardiography Statistical Analysis Results Echocardiography Comment Acknowledgments References

 
Patients
From November 1988 to September 1995, 26 children (18 boys and 8 girls) underwent aortic root replacement in which a pulmonary autograft was used. The mean age of the children was 10.9 years (range, 0.3 to 16.9 years). The aortic valve lesion was congenital in origin in 23 patients; 21 of them had bicuspid valves, and 2 also had subaortic stenosis. The cause of the lesion in the remaining 3 patients was endocarditis (1 patient), juvenile rheumatoid arthritis (1 patient), and rheumatic fever (1 patient). Previous cardiac operations and procedures performed in the patients are listed in Table 1; surgical valvotomy and balloon valvuloplasty were the most frequent procedures. The actual indication for aortic valve replacement was aortic stenosis (5 patients), aortic regurgitation (9 patients), and both (12 patients). Altered left ventricular morphology, increased left ventricular wall thickness, or left ventricular dilatation, or a combination of these, was present in all patients, depending on the cause of the valve lesion.

	Operation

Top Footnotes Abstract Introduction Material and Methods Operation Follow-up Echocardiography Statistical Analysis Results Echocardiography Comment Acknowledgments References

	Follow-up

Top Footnotes Abstract Introduction Material and Methods Operation Follow-up Echocardiography Statistical Analysis Results Echocardiography Comment Acknowledgments References

 
Our follow-up protocol consisted of a yearly physical examination performed in the outpatient clinic. The clinical condition of adolescents and adults was scored using the New York Heart Association classification for dyspnea. A comparable classification was used for children. Height, weight, and blood pressure were noted. Auscultation was performed to detect important valvular stenoses or regurgitation. Electrocardiography was performed to evaluate the cardiac rhythm and determine whether there was left ventricular hypertrophy [7]. Thirteen patients underwent bicycle exercise testing, and the results were described as the percentage of the mean values in a normal population of the same age using the protocol of Godfrey and associates [8]. Endocarditis, thromboembolism, structural deterioration, and nonstructural dysfunction were defined as valve-related events, according to the criteria of Edmunds [9]. Total heart block was included as a cardiac event.

	Echocardiography

Top Footnotes Abstract Introduction Material and Methods Operation Follow-up Echocardiography Statistical Analysis Results Echocardiography Comment Acknowledgments References

 
Echocardiography was performed regularly postoperatively. Precordial two-dimensional color Doppler echocardiography was used to semiquantitatively classify aortic and pulmonary regurgitation by the length and width of the regurgitation jet in the parasternal long-axis view. Aortic regurgitation was graded as none if there was no regurgitation jet; trivial if a short, narrow jet was present just beneath the aortic valve; mild if the jet was limited to the left ventricular outflow tract; moderate if the jet reached halfway to the ventricle; and severe if the jet reached more than halfway to the ventricle and there was also left ventricular dilatation and considerable diastolic back flow in the aortic arch. Pulmonary regurgitation was graded as none if there was no regurgitation; trivial if a short, narrow jet was present just beneath the pulmonary valve; mild if a narrow regurgitation jet was visible in the right ventricular outflow tract; moderate if a broad regurgitation jet was seen extending into the right ventricular outflow tract; and severe if the retrograde flow started within the right and left pulmonary arteries and there was also considerable right ventricular dilatation. Continuous-wave Doppler echocardiography was used to detect aortic and pulmonary stenosis by measuring the maximal blood flow velocity. Stenosis was graded as none, trivial, moderate, or severe.

Two-dimensional echocardiography was used to measure the diameters of the pulmonary autograft annulus. The inner diameter of the autograft annulus was measured at the hinge points of the valve leaflets in an early systolic, parasternal, long-axis view of every initial postoperative precordial two-dimensional echocardiogram (less than 10 days postoperatively) and one or two subsequent echocardiograms obtained during follow-up. Only those subsequent echocardiograms obtained from patients more than 1 year after the autograft procedure were measured. To compare the autograft annulus diameters with normal ones we used the results of Snider and colleagues [10], who used two-dimensional echocardiography to measure pulmonary trunk diameters in 110 normal subjects, aged 1 day to 18 years.

The dimensions of the left ventricle during systole and diastole were determined, and the fractional shortening was calculated from these. The septal and posterior wall thicknesses at end-diastole were also measured, but only in echocardiograms obtained from patients more than 1 year after the autograft procedure. Measurements were compared with those noted for normal Dutch children [11]. The relative end-diastolic wall thickness was expressed as the ratio of the end-diastolic posterior wall thickness to the diameter of the left ventricle [12]. Echocardiography in 2 children was performed at another hospital using the same protocol.

	Statistical Analysis

Top Footnotes Abstract Introduction Material and Methods Operation Follow-up Echocardiography Statistical Analysis Results Echocardiography Comment Acknowledgments References

 
Estimated survival and event-free survival curves were made using the Kaplan-Meier method [13] with 70% confidence limits.

	Results

Top Footnotes Abstract Introduction Material and Methods Operation Follow-up Echocardiography Statistical Analysis Results Echocardiography Comment Acknowledgments References

 
There were no early deaths. Early morbidity occurred in 1 patient, who suffered from a complete atrioventricular block after extensive resection of a subvalvular stenosis; a pacemaker was implanted in this patient. The mean follow-up time was 3.2 years and ranged from 0.2 to 7.5 years. Except for the patient followed up for 0.2 year, follow-up in all patients was longer than 1 year.

Three patients died during follow-up. Two patients died of heart failure caused by restrictive cardiomyopathy with pulmonary hypertension, which was documented by cardiac catheterization. A severe Candida sepsis was present in 1; she died 2 months postoperatively. The second patient died after 22 months. The third patient died 6 months postoperatively as the result of relapse of chronic juvenile rheumatoid arthritis, resulting in autograft and mitral valve destruction [14]. One patient was reoperated on 22 months postoperatively because of relapse of acute rheumatic fever that led to the destruction of the pulmonary autograft [15]. There were no reoperations necessitated by technical reasons or structural degeneration. Endocarditis did not occur. Thromboembolic complications were not observed. The survival and reoperation-free survival curves are shown in Figure 1. The estimated 5- and 7-year survival rate was 87% (70% confidence limits at 5 years, 75 to 99), and the event-free survival rate was 79% (70% confidence limits at 5 years, 66 to 92). The patient with the postoperative complete atrioventricular block was reoperated on for mitral and tricuspid regurgitation 6.5 years after the pulmonary autograft procedure. The mitral valve was replaced with a mechanical valve and valvuloplasty was performed on the tricuspid valve. Concomitantly his endocardial pacemaker was replaced with an epicardial pacemaker.

View larger version (14K): [in this window] [in a new window]   Fig 1. . Kaplan-Meier curves of survival (solid line) and reoperation-free survival (dotted line) for children after the pulmonary autograft procedure (numbers are equal for both curves). (CL = confidence limits.)

	Echocardiography

Top Footnotes Abstract Introduction Material and Methods Operation Follow-up Echocardiography Statistical Analysis Results Echocardiography Comment Acknowledgments References

 
Aortic and pulmonary valve function were described semiquantitatively at the last follow-up (Table 2). Moderate or severe aortic regurgitation or stenosis was not seen. The 5 patients with mild aortic regurgitation had been found to have trivial aortic regurgitation early after operation. These regurgitation jets had become mild by 3 to 5 years after operation. However, the clinical status of these patients was good. Moderate or severe pulmonary regurgitation was also not found. Moderate pulmonary allograft stenosis was noted in 2 patients, with blood flow velocities of between 3.5 and 4.0 m/s along the right ventricular outflow tract. It occurred in 2 patients with cryopreserved pulmonary allografts inserted at 5 and 16 years of age; the gradients became mild 3 and 4 years postoperatively. The clinical status of these patients was good.

View larger version (16K): [in this window] [in a new window]   Fig 2. . Diameters of the pulmonary autograft annulus during follow-up in 19 children who had undergone the pulmonary autograft procedure. The dotted lines represent the 80% prediction interval of normal pulmonary trunk diameters measured by Snider and associates [10] in 110 normal children (aged, 1 day to 18 years) using two-dimensional echocardiography. (BSA = body surface area.)

	Comment

Top Footnotes Abstract Introduction Material and Methods Operation Follow-up Echocardiography Statistical Analysis Results Echocardiography Comment Acknowledgments References

There were no early deaths in this group of patients with complex abnormalities. Two patients died during follow-up for reasons not related to the autograft procedure. Another patient died as the result of recurrent chronic juvenile rheumatoid arthritis that destroyed the autograft [14]. Reoperation was necessary in a patient with recurrent acute rheumatic fever that destroyed the autograft [15]. Chronic juvenile rheumatoid arthritis is regarded as a contraindication to the pulmonary autograft procedure, and acute rheumatic fever is regarded as a relative contraindication, depending on the adequacy of antibiotic prophylaxis for the disease.

The clinical outcome of the remaining patients in this series was good. They are doing well and showing normal growth patterns. Only 1 patient is taking medication. Other studies have also shown improvement in the functional class in patients who receive pulmonary autografts [1, 2], but the assessment in these studies did not include exercise capability testing. The children undergoing this exercise testing in our study performed excellently, with capabilities in accordance with those of age-matched normal children. Electrocardiographic signs of left ventricular hypertrophy or myocardial damage were not found in our patients after 1 year of follow-up, contrasting with the electrocardiographic changes found in 20 adult patients, 3 of whom had a right bundle-branch block and 2 of whom had signs of severe ischemia [17]. Pulmonary autograft valve stenosis was not found, and any regurgitation was trivial or mild. This good valve function in the midterm has been reported by others as well [1]. Oury and Mackey [18], in their report regarding the Ross Procedure Registry, also noted good valve function in most of the patients, both children and adults, in the root replacement group.

The allograft implanted in the right ventricular outflow tract may be of more concern. The pulmonary regurgitation in such patients has not been found to be important, but 2 of the patients in our series have important gradients across the allograft in this location. Although right ventricular hypertrophy has not been found and the clinical function is good in these patients, it may be that structural degeneration will occur in these right-sided pulmonary allografts and necessitate reoperation in the future. In this regard, however, the long-term function of right-sided pulmonary allografts must be distinguished from the function of allografts used for congenital right heart defects [19].

As has been reported before, the greatest advantage to the use of a pulmonary autograft is its ability to increase in diameter over time [1]. Our study confirmed the increasing diameter of the pulmonary autograft in patients who had undergone their operation more than a year before. As compared with the 80% prediction interval cited by Snider and colleagues [10], however, most of the diameters of the grafts in our patients are above this interval, indicating that the measurements of native pulmonary annuli are questionable as reference values. Although in most patients this increase was less than 20%, there were 3 patients with an increase of more than 50% (more than 10 mm). Elkins and associates [1] reported a greater increase in the diameter of freestanding pulmonary autografts than in the diameter of autografts placed using the intraaortic cylinder technique, and they attributed this "extra" increase to dilatation of the graft. Long-term follow-up is therefore necessary to come to any conclusions about the behavior of the pulmonary autograft diameter. Its relationship to aortic regurgitation especially warrants an in-depth follow-up. In our series in which the mean follow-up was 3.2 years and the longest follow-up was 7.5 years, only limited aortic regurgitation of some autografts and no important gradient across the autograft valve were noted. Correlations between an increase in the autograft annulus diameter and the severity of aortic regurgitation could not be found.

Functional status and survival rates are not only improved in adult patients who undergo aortic valve replacement [20], there is also a regression in the myocardial mass and an improvement in systolic and diastolic left ventricular function in such patients [21–23]. In children, left ventricular dimensions often remain impaired after aortic valve procedures for congenital stenosis, probably related to the remaining gradient across the valve [24, 25]. All variables related to left ventricular size and function were within normal limits in all our patients except 1, contrasting with the abnormal situation before operation. This is in agreement with the results noted by others, who also found an improvement in the left ventricular dimensions in children, indicating the lack of sequelae from this operation [26, 27]. A limitation to our study, however, is that adequate echocardiographic measurements of the left ventricle could not be obtained in all patients.

We conclude that the surgical results, clinical outcome, valve function, and left ventricular function are good after aortic root replacement with a pulmonary autograft and recommend this procedure for aortic valve replacement in children.

	Acknowledgments

Top Footnotes Abstract Introduction Material and Methods Operation Follow-up Echocardiography Statistical Analysis Results Echocardiography Comment Acknowledgments References

 
We thank Dr Annette Reimers (cardiologist) and Dr Livia Kapusta (cardiologist) of the Radboud Hospital in Nijmegen and Dr Alessandro Frigiola (thoracic surgeon) from the San Donato Hospital in Milan for involving us in the surgical treatment of their patients and for the use of follow-up data. All allografts implanted in patients treated in Rotterdam were distributed by Bio-Implant Services, Leiden, the Netherlands.

	Footnotes

Top Footnotes Abstract Introduction Material and Methods Operation Follow-up Echocardiography Statistical Analysis Results Echocardiography Comment Acknowledgments References

 
Address reprint requests to Dr Hokken, Cardiopulmonary Surgery, BD 156, University Hospital Sophia-Dijkzigt, Rotterdam, Dr Molewaterplein 40, 3015 GD Rotterdam, the Netherlands.

	References

Top Footnotes Abstract Introduction Material and Methods Operation Follow-up Echocardiography Statistical Analysis Results Echocardiography Comment Acknowledgments References

 

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