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Ann Thorac Surg 1999;68:1356-1359
© 1999 The Society of Thoracic Surgeons

Original Articles: Cardiovascular

Long-term results of surgical valvuloplasty for congenital valvar aortic stenosis in children

Claude C. Chartrand, MDa, Enrique Saro-Servando, MDa, Jitka Suzanne Vobecky, MDa

a Division of Cardiovascular Surgery, Sainte-Justine Hospital, Montreal, Quebec, Canada

Address reprint requests to Dr Chartrand, Division of Cardiovascular Surgery, Hôpital Sainte-Justine, 3175 Côte Sainte-Catherine, Montreal, Quebec PQ, Canada H3T 1C5
e-mail: chirhsj{at}point-net.com

Presented at the Thirty-fifth Annual Meeting of The Society of Thoracic Surgeons San Antonio, TX, Jan 25–27, 1999.


   Abstract
Top
 Abstract
Introduction
 Material and methods
 Results
 Discussion
 References
 
Background. To question the validity of surgical aortic valvuloplasty for congenital aortic valve stenosis, a retrospective study was undertaken to determine the long-term survival, the incidence of valve restenosis or insufficiency, and the freedom of reoperation or valve replacement.

Methods. From January 1960 through 1992, 67 consecutive children diagnosed with congenital aortic valve stenosis underwent an open aortic valvuloplasty at our institution. Ages at operation ranged from 6 to 228 months (mean 105.7 ± 52 months). The mean follow-up of these patients has been 127.5 ± 66.7 months.

Results. There was no hospital mortality, but two late valve-related deaths occurred. Eight patients developed aortic regurgitation 5 to 125 months (mean 66.6 ± 35 months) after surgical valvuloplasty, and 1 of them required aortic valve replacement. Because of restenosis, 16 patients required a second operation. Of them, 5 children underwent a second aortic valvuloplasty without mortality and, in 4 of them, the functional result has been excellent after a mean follow-up of 75.4 ± 12 months. Eleven patients required an aortic valve replacement 62 to 208 months postop (mean 100.9 ± 50.8 months). Mechanical prosthesis were used in 7 and bioprosthesis in 4. Two patients required a Konno and 1 required a Ross procedure. There were no early nor late deaths after reoperations. The probability of 20-year survival after the first valvuloplasty was 94%, the freedom of reoperation 63%, and the freedom of aortic valve replacement 73% for the same time period.

Conclusions. Our results demonstrate that congenital aortic valvar stenosis in children can be surgically well controlled until adulthood. Our study also shows that surgical valvuloplasty is a safe and efficacious procedure and that its benefical effect is maintained over 20 years in the majority of children.


   Introduction
Top
 Abstract
 Introduction
Material and methods
 Results
 Discussion
 References
 
Aortic valve stenosis is a relatively frequent congenital heart defect. Most often it is asymptomatic, but with time produces important physiopathological consequences, and carries the known risk of sudden death if the transvalvular gradient exceeds 50 mm Hg [1]. Open aortic valvuloplasty may, most of the time, alleviate adequately the valvular stenosis with a low morbidity and mortality [2]. However some long-term studies indicate that reoperation will be necessary in a significant number of patients due to restenosis or insufficiency [3–5].

In order to question the validity of surgical aortic valvuloplasty, a retrospective study was undertaken to determine the long-term survival, the incidence of valve restenosis or insufficiency, and the freedom of reoperation or valve replacement.


   Material and methods
Top
 Abstract
 Introduction
 Material and methods
Results
 Discussion
 References
 
From January 1960 through January 1992, 67 consecutive patients diagnosed with congenital aortic valve stenosis underwent an open aortic valvuloplasty at Sainte-Justine Hospital. Newborns and infants younger that 6 months were reported previously and are excluded from this study [6]. There were 40 boys and 27 girls. Ages at operation ranged from 6 to 228 months (mean 105 ± 52 months). Three patients had four previous aortic valve balloon dilatations 1 to 7 months before surgery.

Cardiac catheterization was done in all patients, and indication for surgery was based on the pressure gradient between the left ventricle and the ascending aorta. Aortic valve gradients ranged from 40 to 163 mm Hg (mean 83 ± 28 mm Hg). Some patients with low aortic gradient were operated upon because of additional criteria: progressive left ventricular hypertrophy, alteration in repolarization phase on electrocardiogram (ECG), or cardiovascular symptoms.

Operation was performed through a median sternotomy using cardiopulmonary bypass with moderate hypothermia. The heart was arrested with cold crystalloid cardioplegic solution and myocardial protection was completed by topical cooling. Through an oblique aortotomy, the fused commissures were incised and, when required, resection of fibrocalcic nodules and trimming of the cusps were undertaken. Bicuspid aortic valve was present in 53 patients. At the end of surgery, the pressure gradients were recorded by direct left ventricular and aortic punctures. Associated cardiac lesions were also corrected in 8 children (persistent ductus arteriosus, 2; atrial septal defect, 2; supravalvar aortic stenosis, 2; subvalvar aortic stenosis, 1; partial anomalous pulmonary venous return, 1; and fibroelastosis, 1).

All patients were followed at a regular outpatient clinic by the referring cardiologist until transfer to an affiliated adult hospital. In our earlier experience, the follow-up was based essentially on physical examination ECG and heart catheterization. Afterwards, the echo Doppler became the standard examination. Aortic valve insufficiency, assessed on echo Doppler, was considered mild when regurgitant flow reached less than 25% of the distance between the aortic valve and the apex of left ventricle, moderate if it was 25% to 50%, and severe if it reached more than 50% of this distance [7]. The peak systolic gradient between the left ventricle and the ascending aorta indicated the degree of stenosis [8].

Statistical analysis were done calculating means, standard deviations, and actuarial Kaplan-Meir analysis for expression of survival and freedom of reoperation.


   Results
Top
 Abstract
 Introduction
 Material and methods
 Results
Discussion
 References
 
There was no operative or hospital mortality in all 67 first open aortic valvuloplasties. The mean preoperative aortic gradient of 83 ± 28 mm Hg was decreased to 24 ± 16 mm Hg after surgery (Fig 1). One patient presented postoperative bleeding disorder requiring exploration, and 1 developed a transient neurological deficit after an hypertensive crisis. No significant aortic insufficiency has been noted before discharge.



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  		Fig 1. Gradients before and after the first valvuloplasty. The results are expressed as mean ± standard deviation.

 
The mean follow-up of these children has been 127.5 ± 66 months. There were three late deaths (4.5%). One 7-year-old child presenting severe left ventricular fibroelastosis associated with mitral insufficiency and systemic pulmonary pressure at the time of valvotomy died 42 months later from congestive heart failure; at necropsy, no significant aortic restenosis nor insufficiency was found. Two other patients in whom a gradient of 60 and 80 mm Hg had been documented at their last follow-up visit, 7 and 9 years postoperatively, died suddenly during physical activities.

Figure 2 illustrates the actuarial survival of all patients after the first open aortic valvotomy. The probability of survival 20 years after the initial valvuloplasty is 94% (confidence limit [CL] 86% to 100%).



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  		Fig 2. Actuarial survival after initial valvuloplasty (67 patients). The vertical bar indicates 70% confidence limits. The numbers in parenthesis are the numbers of patients for each interval.

 
Eight patients developed moderate to severe aortic valve insufficiency 5 to 125 months after surgery (mean 66.6 ± 35 months). Only in 1 child was the aortic regurgitation severe enough to require a valve replacement with a mechanical device. The remaining 7 patients are under medical therapy alone.

Because of recurring and progressing valvular stenosis, 16 patients underwent a second therapeutic procedure from 47 to 208 (mean 78 ± 35.6) months after the first valvuloplasty. In 3 patients, balloon valvotomy was performed; however, 2 of these eventually needed a valvular replacement.

In 5 children, a second valvuloplasty was carried out. The mean preoperative aortic gradient of 105 ± 5 mm Hg has been lowered to 40 ± 12 mm Hg after surgery (Fig 3). From 13 to 118 months (mean 73 ± 53 months) after this second procedure, no recurrent stenosis nor insufficiency was evidenced in 4 of the 5 children.



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  		Fig 3. Gradients before and after the second valvuloplasty. The results are expressed as mean ± standard deviation.

 
In 10 patients, due to the severity of valvular disease, the reoperation consisted of a valvular replacement. Their ages at the time of reoperation were from 139 to 216 months (mean 182 ± 20 months). Six mechanical prosthesis (four Medtronic, one Omniscience, one Lillehei Kaster) and three porcine bioprosthesis (two Carpentier Edwards, one Hancock) were implanted. The mean prosthetic valve diameter was 23 ± 2 mm, ranging from 19 to 27 mm. In 2 patients, a Konno aorto-ventriculoplasty was necessary, and in 1, a Ross procedure was preferred. There was no early or late mortality after valvular replacement. One tamponade was explored and 1 patient presented a femoral artery thrombosis. There has been no late complication among patients with mechanical devices. In 1 of the patients with bioprosthesis, an aortic gradient of 45 mm Hg was documented 19 months after implantation. The actuarial analysis of this cohort of patients demonstrates that, after the initial valvuloplasty, the probability of freedom of reoperation at 5, 10, and 20 years is 97% (CL 94% to 99%), 71% (CL 65% to 77%), and 63% (CL 50% to 76%), and freedom of aortic valve replacement is 97% (CL 94% to 99%), 82% (CL 76% to 87%), and 73% (CL 60% to 86%) for the same time period (Fig 4).



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  		Fig 4. Actuarial freedom from aortic valve replacement and reoperation after initial valvuloplasty. The vertical bar indicates 70% confidence limits.

 

   Discussion
Top
 Abstract
 Introduction
 Material and methods
 Results
 Discussion
References
 
In accordance with many other reports, our results demonstrate that open aortic valvuloplasty is a safe and efficacious treatment for children with congenital aortic valve stenosis [1, 9, 10]. Indeed, there has been no hospital mortality and the morbidity has been minimal. Also, direct vision allows to proceed to an adequate commissurotomy along with resection of fibrous nodules and trimming of the valve cusps, when required, and to avoid trauma of the valve leaflets [9, 11, 12]. In the majority of cases, the transvalvular gradient could be substantially decreased, and no iatrogenic valvar insufficiency could be identified in the immediate postoperative period.

Therefore, our present attitude favors open aortic valvuloplasty in pure aortic valve stenosis in children even when bicuspid valve is present [9, 13]. This therapeutic option offers the children many years with adequate valvular function because 63% (CL 50% to 76%) are free of reoperation 20 years after their initial valvuloplasty. Finally, this initial management allows to postpone aortic valve replacement because 73% (CL 60% to 86%) of the patients have been free of valve replacement at 20 years postop, as well as the prosthetic valve-related complications: outgrow of the device, complications related to anticoagulation, risk associated with pregnancy, deterioration of the bioprosthesis, and the risks of subsequent replacements [14–16].

However, the outcome of this congenital anomaly is unpredictable, for some will develop restenosis, valvular insufficiency, or both. Hence, 16 children presented predominant restenosis severe enough to require a reoperation. In 3 instances, balloon valvotomy was performed but was successfull only once. We now attempt this procedure only when the leaflets are not dysplastic. In 5 cases, a second valvuloplasty was feasible and carried out without mortality. After the procedure, the aortic gradient decreased significantly, there has been no postoperative complication, and the long-term result has been satisfactory in 4 of them. Because of this experience, we favor a second valvuloplasty if the valvular pathology is favorable.

Postoperative valvular insufficiency developed in 8 patients. Because no obvious regurgitation occurred in the immediate postoperative period, this may be related to the careful limitation of the commissurotomy, believing that a mild residual stenosis is less deleterious than an aortic insufficiency. The relatively late postoperative appearance of valvular regurgitation allows us to think that it is more related to the progression of the disease itself [11, 12]. In the presence of significant valvular regurgitation, we still favor medical therapy, and defer reoperation unless there is progressive ventricular dilatation.

Severe progression of the valvular disease made further valvuloplasty impossible in 11 patients who required aortic valve replacement. Early in our experience, three bioprosthesis were implanted. However, because of their fast degeneration as documented by others, we abandoned their further use in 1980 [17–19]. In all but one of the remaining patients, mechanical valves were used. It is noteworthy that our surgical approach allowed us to delay for many years valvular replacement in order that the aortic annulus grows sufficiently to accomodate a prosthesis of adult size. In three instances where the aortic annulus was too small, we have favored a Konno or a Ross procedure in order to avoid a second valvular replacement. In accordance with our previous results, there has been no early or late mortality and minimal morbidity after valve replacement [20].

Sudden death in children with aortic stenosis has been well documented in the literature [1, 17]. This complication was encountered twice, 84 and 101 months after the valvuloplasty. At the last outpatient clinic, the aortic gradients were 65 and 85 mm Hg, respectively. Considering our results and according with others, we believe that these two complications might have been avoided by another surgical procedure, and that all patients presenting a transvalvular gradient above 50 mm Hg or progressive left ventricular hypertrophy or changes in repolarization should be offered a surgical relief even if asymptomatic [9, 17, 21]. Also, it seems possible that the risk of permanent myocardial lesions associated with hypertrophy and ischemia could be decreased with earlier surgical intervention.

Our results demonstrate that aortic valve stenosis in children can be surgically well controlled until adulthood. In our experience, surgical valvuloplasty has been safe and efficacious, and its beneficial effect has been maintained over 20 years in two-thirds of our patients. Hence, we believe that open aortic valvuloplasty should be the first therapeutic procedure in children with pure congenital aortic valve stenosis. In many patients with recurring stenosis, a second open valvotomy can be as beneficial as the first one. Finally, if the pathological process is too severe with or without insufficiency, valvular replacement should be carried out as well as an annular enlargement procedure when required.


   References
Top
 Abstract
 Introduction
 Material and methods
 Results
 Discussion
 References
 

  1. Wheller J.J., Hossier D.M., Teske D.W., Craenen J.M., Kilman W. Results of operation for aortic stenosis in infants, children and adolescents. J Thorac Cardiovasc Surg 1988;96:474-477.[Abstract]
  2. Kirklin J.W., Barratt-Boyes B.G. Congenital aortic stenosis. In: Kirklin J.W., Barratt-Boyes B.G., eds. Cardiac Surgery, 2nd ed. New York: Churchill Livingstone, 1993:1195-1237.
  3. Caspi J., Ilbawi M.N., Robertson D.A., Piccione W., Monson D.O., Najafi H. Extended aortic valvuloplasty for recurrent valvular stenosis and regurgitation in children. J Thorac Cardiovasc Surg 1994;107:1114-1120.[Abstract/Free Full Text]
  4. Sommerville J., Presbitero P., Ross D. Open aortic valvotomy in the young. Br Heart J 1981;45:351.
  5. DeBoer D.A., Robbins R.C., Maron B.J., McIntosh C.L., Clark R.E. Late results of aortic valvotomy for congenital valvar aortic stenosis. Ann Thorac Surg 1990;50:69-73.[Abstract/Free Full Text]
  6. Vobecky J.S., Chartrand C., Angaté H., Stanley P. Surgery for critical aortic stenosis in newborn is still good therapy after 25 years. Canadian J Surg 1992;5:489-491.
  7. Ilbawi M.N., DeLeon S.Y., Wilson W.R., et al. Extended aortic valvuloplasty. Ann Thorac Surg 1991;52:663-668.[Abstract/Free Full Text]
  8. Sander S.P. Echocardiography. In: Fyler D.C., ed. Nadas’ Pediatric Cardiology. Philadelphia: Hanley & Belfus Inc, 1992:159-186.
  9. Dobell A.R.C., Bloss R.S., Gibbons J.E., Collins G.F. Congenital valvular aortic stenosis. J Thorac Cardiovasc Surg 1981;81:916-921.[Abstract]
  10. Sandor G.G.S., Olley P.M., Trusler G.A., et al. Long-term follow-up of patients after valvotomy for congenital valvular aortic stenosis in children. J Thorac Cardiovasc Surg 1980;80:171-176.[Medline]
  11. Chiariello L., Vlad P., Subramanian S. Surgical treatment of congenital valvular aortic stenosis. Thorax 1976;31:398-404.[Abstract/Free Full Text]
  12. Brown J.W., Stevens L.S., Holly S., et al. Surgical spectrum of aortic stenosis in children. Ann Thorac Surg 1988;45:393-403.[Abstract/Free Full Text]
  13. Van Son J.A.M., Reddy V.M., Black M.D., Rajasinghe H., Haas G.S., Hanley F.L. Morphologic determinants favoring surgical aortic valvuloplasty versus pulmonary autograft aortic valve replacement in children. J Thorac Cardiovasc Surg 1996;111:1149-1157.[Abstract/Free Full Text]
  14. Ilbawi M.N., Idriss F.S., DeLeon S.Y., Muster A.J., Duffy C.E., Gidding S.S. Valve replacement in children. Ann Thorac Surg 1987;44:398-403.[Abstract/Free Full Text]
  15. Russo R., Bortolotti U., Schivazappa L., Girolami A. Warfarin treatment during pregnancy. A clinical note. Haemostasis 1979;8:96-98.[Medline]
  16. Sareli P., England M.J., Berk M.R., et al. Maternal and fetal sequelae of anticoagulation during pregnancy in patients with mechanical heart valve prosthesis. Am J Cardiol 1989;63:1462-1465.[Medline]
  17. Williams D.B., Danielson G.K., McGoon D.C., Puga F.J., Mair D.D., Edwards W.D. Porcine heterograft valve replacement in children. J Thorac Cardiovasc Surg 1982;84:446-450.[Abstract]
  18. Kanazawa H., Miyamura H., Watanabe H., et al. Surgical management of valve replacement in children. Kyobu Jeka Jpn J Thorax Surg 1991;44:887-892.
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