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Ann Thorac Surg 2003;76:1412-1416
© 2003 The Society of Thoracic Surgeons

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Original article: cardiovascular

Surgical aortic valvotomy in infancy: impact of leaflet morphology on long-term outcomes

^a Department of Cardiac Surgery, Birmingham Childrens' Hospital, Birmingham, United Kingdom
^b Department of Cardiology, Birmingham Childrens' Hospital, Birmingham, United Kingdom

Accepted for publication April 25, 2003.

^* Address reprint requests to Dr Brawn, Department of Cardiac Surgery, Birmingham Childrens' Hospital, Steelhouse Lane, Birmingham B4 6NH, UK.
e-mail: elizabeth.leeson{at}bhamchildrens.wmids.nhs.uk

	Abstract

Top Abstract Introduction Patients and methods Results Comment References

 
BACKGROUND: Surgical valvotomy for critical aortic stenosis in children enables relatively accurate commissurotomies to be fashioned, resulting in the formation of two or three leaflets. We hypothesized that outcomes after surgery may be best in patients in whom three leaflets are produced.

METHODS: A retrospective review of infants undergoing primary surgical valvotomy at our institution during a 12-year period was carried out. Patients who had additional intracardiac defects were excluded. Clinical and echocardiographic follow-up data were analyzed.

RESULTS: Fifty-four patients fulfilled the study criteria. Median age at surgery was 3 weeks (range, 0 to 51 weeks). Commissurotomy resulted in bileaflet anatomy in 41 patients (group A) and trileaflet anatomy in 13 patients (group B). Operative mortality was 5% in group A and 0% in group B (p = 1.0). In group A, 18 patients required one or more aortic valve reinterventions, including valve replacement in 8 patients. In group B, there was only one reintervention (repeat valvotomy). Kaplan-Meier analysis showed that at 10 years, comparisons of group A versus group B were as follows: actuarial survival, 85% versus 100% (p = 0.15); freedom from reintervention, 33% versus 92% (p = 0.01); freedom from aortic reoperation, 45% versus 92% (p = 0.04); and freedom from aortic valve replacement, 57% versus 100% (p = 0.07).

CONCLUSIONS: Long-term outcomes after aortic valvotomy are significantly better in infants in whom surgery results in trileaflet rather than bileaflet anatomy. Preoperative evaluation of valve morphology may enable selection of a group of patients in whom results of surgery are excellent.

	Introduction

Top Abstract Introduction Patients and methods Results Comment References

 
Valvular aortic stenosis in children is usually treated by percutaneous balloon valvuloplasty or surgical valvotomy. Satisfactory short-term and long-term results have been reported in neonates as well as in older children with both percutaneous valvuloplasty [1–4] and valvotomy [5, 6]. The choice of treatment appears to depend on institutional preference rather than evidence-based selection of the most appropriate procedure for an individual patient.

Percutaneous dilatation relieves aortic stenosis by causing rupture along lines of least resistance, either along underdeveloped commissures or into leaflet tissue [7]. Surgical valvotomy, on the other hand, allows more accurate fashioning of commissurotomies and debridement of any excess material on the valve leaflets. Even with surgery, the variability in anatomic abnormality is such that in more-disorganized valves it is only possible to fashion two cusps, whereas in those with better-developed anatomy three cusps can be formed. We hypothesized that outcomes after surgery may be better in the latter situation. To test this hypothesis, we retrospectively studied the effect of number of leaflets created by surgical valvotomy on short-term and midterm outcomes in children operated on in the first year of life.

	Patients and methods

Top Abstract Introduction Patients and methods Results Comment References

 
This study was a retrospective review of case records and echocardiograms of patients operated on at our institution.

Patients
Patients undergoing surgical aortic valvotomy as a primary procedure in the first year of life during a 12-year period between January 1989 and December 2000 were identified. During this period, the policy at our institution was for all infants with critical aortic stenosis to have surgical treatment in the first instance. Exclusion criteria were any previous aortic valve procedure (percutaneous or surgical) and the presence of other major intracardiac defects. Patients requiring concomitant surgery for patent ductus arteriosus or coarctation of the aorta were included.

A total of 89 patients underwent primary aortic valvotomy at our institution during this time period. Of these, 24 were older than 1 year of age, and 1 required mitral valve surgery as well. Ten patients, operated on before 1992, had hypoplastic left ventricles and by current criteria would have undergone univentricular repair (the Norwood procedure) rather than valvotomy. Outcomes in these patients would have been determined by the ventricular size as much as by the aortic valve anatomy, and so they were excluded. The remaining 54 patients formed the study population.

Surgical technique
The operations were performed by one of two surgeons (W.J.B. and B.S.). Valvotomies were carried out using hypothermic cardiopulmonary bypass and crystalloid cardioplegic arrest of the heart; short periods of circulatory arrest were also used when necessary to improve visualization. Through an aortotomy, the aortic valve was inspected and incisions were made into thickened commissural tissue, ensuring that the leaflets thus created were well supported and not liable to prolapse. Any excessive nodular or degenerative material on the valve was debrided. Eleven patients required concomitant ligation of patent ductus arteriosus, and 5 required repair of coarctation of the aorta.

The description of the operation in the case records was used to define the surgical result in terms of valve leaflet number. The patients were divided into two groups depending on whether valvotomy resulted in a bileaflet (group A; n = 41) or trileaflet (group B; n = 13) structure.

Follow-up
After discharge from the hospital, patients were evaluated clinically and by transthoracic echocardiography at regular intervals either at our institute or by the referring cardiologists at other institutions. Up-to-date follow-up was available for all except 1 patient, who had moved abroad and was not traceable. Median follow-up was 81 months (range, 6 to 138 months) for group A and 82 months (range, 21 to 143 months) for group B.

Echocardiography
Recordings of preoperative echocardiograms were available for 40 of the 54 patients and were reviewed by two observers (W.J.B. and R.D.) blinded to the outcome of surgery. The aortic annulus size was recorded, and an attempt was made to identify the native leaflet anatomy and predict how many leaflets could be constructed at surgical valvotomy.

Statistics
Data were analyzed using SPSS for Windows version 10 (SPSS Inc, Chicago, IL). Categorical group characteristics were compared using Fisher's exact test, and continuous variables were compared using the unpaired Student's t test. Analysis of postoperative time-related events was by the Kaplan-Meier method, using the log-rank test for group comparisons. Probability values less than 0.05 were considered significant.

	Results

Top Abstract Introduction Patients and methods Results Comment References

 
Preoperative and operative data are summarized in Table 1. There were no statistically significant differences between groups A and B in terms of age at surgery, sex, and proportion of patients who were neonates, had clinically detectable heart failure, required inotropic support or ventilation preoperatively, or required concomitant ligation of patent ductus arteriosus or repair of coarctation of the aorta. Retrospective measurement of aortic annular diameter in patients whose preoperative echocardiograms were available showed no statistically significant difference between the groups. Cardiopulmonary bypass, aortic clamp, and circulatory arrest times were statistically similar.

Mean (± standard deviation) intensive care unit stay in the remaining 51 patients was 2.9 ± 2.7 days in group A and 4.0 ± 5.6 days in group B (p = 0.35). Two patients had a complicated recovery. One, in group A, experienced renal failure requiring temporary dialysis, necrotizing enterocolitis requiring ileostomy, and lower limb ischemia requiring embolectomy followed by amputation of the foot. The second, in group B, required temporary renal dialysis. None of the other patients had any major complications. Mean postoperative hospital stay was 11.1 ± 6.7 days in group A and 13.1 ± 12.1 days in group B (p = 0.47).

Late mortality
There were 3 additional late deaths in group A. One patient had restenosis of the aortic valve and 6 months after valvotomy underwent uneventful mechanical valve replacement with annular enlargement. Three months later he presented with severe heart failure and was found to have valve thrombosis. An attempt to replace the prosthetic valve was unsuccessful. Another patient died suddenly at home with no cause of death found at postmortem examination, and the third died after a prolonged epileptic fit. There were no late deaths in group B.

Actuarial survival rates are shown in Figure 1. Ten-year survival was 85% ± 6% in group A and 100% in group B (p = 0.15).

View larger version (14K): [in this window] [in a new window]   Fig 1. Actuarial survival in patients with bileaflet (group A) and trileaflet (group B) aortic valves after valvotomy.

View larger version (16K): [in this window] [in a new window]   Fig 2. Actuarial freedom from any reintervention.

View larger version (15K): [in this window] [in a new window]   Fig 3. Actuarial freedom from reoperation.

View larger version (15K): [in this window] [in a new window]   Fig 4. Actuarial freedom from aortic valve replacement (AVR).

	Comment

Top Abstract Introduction Patients and methods Results Comment References

 
This study was a retrospective review of infants who had undergone surgical valvotomy as primary treatment for congenital aortic stenosis. More than half were neonates and more than two thirds were in heart failure. The operative mortality rates of 3.4% overall and 6.5% for neonates compare favorably with other published series, most of which report neonatal mortality rates of 10% to 20% [1, 3, 8–10]. However, as described above, our study excluded neonates with hypoplastic left ventricles operated on in the years before the Norwood procedure became established. In another series (18 patients) in which neonates with small left ventricular size were excluded, an operative mortality rate of 0% has been reported [5]. Overall 10-year actuarial survival was 89%, freedom from reintervention was 49%, and freedom from aortic valve replacement was 69%. These figures, too, compare favorably with other series [1, 5, 9, 10], and provide further evidence supporting a continuing role for surgery in the management of congenital aortic stenosis. Longer-term studies of hospital survivors suggest that the beneficial outcomes of surgical valvotomy are maintained at 15 to 20 years [6, 11, 12].

An alternative form of treatment for congenital aortic stenosis, percutaneous balloon valvuloplasty, has gained popularity in the last decade, and in many institutions is now the procedure of choice. Although a less invasive procedure, early mortality rates are similar to surgical valvotomy in comparable populations [1, 2, 4, 13, 14]. Significant aortic regurgitation is produced in approximately 5% to 20% of patients [1, 2, 4, 13], and other major procedure-specific complications such as femoral artery thrombosis can occur [4, 13]. In the medium term, reintervention and survival rates appear to be of the same order as after surgery [1, 2, 13, 14]. Although the overall results do not show clear superiority of either percutaneous or surgical therapy, it may be possible to establish whether one or the other may be more beneficial in particular patient subgroups.

It is likely that balloon dilatation of a stenotic valve results in splitting of tissues along lines of least resistance. In a study of 10 patients aged 3 to 17 years in whom balloon valvuloplasty was immediately followed by open examination of the valve, only 3 were thought to have a satisfactory result [7]. In 1 patient, the rupture was too extensive, necessitating repair. Three patients required additional commissurotomies, whereas in another 3, the valve leaflet tissue had ruptured and had to be repaired. Open valvotomy, on the other hand, allows the surgeon to debride the leaflets and to perform accurate commissurotomies under direct vision. In addition, an attempt can be made to construct leaflet anatomy as close to normal as possible.

It cannot be assumed that a trileaflet structure created from a congenitally stenotic valve will perform physiologically as well as a normal trileaflet aortic valve. Morphologic studies of valves from infants with critical aortic stenosis have shown that three sinuses could always be identified, delineated either by the attachment of leaflet tissue or by aortic infoldings [15]. However, there was usually only one patent commissure. The height and base of the interleaflet triangles between rudimentary commissures were significantly reduced, with the tops of the triangles not reaching the sinotubular junction. The lengths of the free edge and line of attachment of the leaflets were also reduced. All valves had nodular excrescences on the leaflets composed of myxoid tissue, and the majority had thickened leaflets. Therefore, although it should theoretically always be possible to extend commissurotomies into the thickened aortic folds and thereby construct three leaflets [15], the resulting valve will still have fundamental morphologic abnormalities.

We hypothesized that despite being abnormal, trileaflet valves may still produce better outcomes than bileaflet ones. Only in a quarter of our patients was the commissural tissue quality and root anatomy such that three leaflets could be constructed without producing significant valve regurgitation. In this group of patients, the need for reintervention for recurrent stenosis and the need for reoperation were found to be significantly less than in patients left with bileaflet valves. Perhaps more importantly, there was a trend toward a reduced requirement for valve replacement in childhood, although this did not reach statistical significance. It is possible that these results reflect not merely an effect of leaflet number but the fact that those patients in whom three leaflets can be created have better-developed aortic root anatomy. Even so, this may be a group of patients in whom surgical valvotomy should be the treatment of choice, as it is likely that potentially trileaflet valves treated with balloon valvuloplasty remain bileaflet [7].

A previous study [16], in an older age group of children, many of whom had concomitant cardiac anomalies repaired, also showed lower rates of residual or recurrent stenosis or regurgitation in trileaflet compared with nontrileaflet aortic valves after valvotomy.

It remains to be seen whether it is possible to prospectively identify those patients in whom there is a high likelihood of being able to construct three leaflets. Our attempt to do so retrospectively had a sensitivity of 88% and a specificity of 50%. However, this was from old recordings of echocardiograms that did not always look specifically at leaflet anatomy, and it may be possible with modern echocardiography to more accurately assess native valve structure. Further studies are required to ascertain whether outcomes after balloon valvuloplasty are similarly affected by leaflet anatomy before surgical valvotomy can be recommended as the treatment of choice in patients with potentially trileaflet valves.

	References

Top Abstract Introduction Patients and methods Results Comment References

 

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