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Late results after repair of aortic insufficiency associated with ventricular septal defect
Late results after repair of aortic insufficiency associated with ventricular septal defect The late results of 70 patients aged 1.96 to 35.9 (mean 10.1) years who had repair of ventricular septal defect and aortic insufficiency from 1968 to 1988 were reviewed. The ventricular septal defect was subcristal in 50 and subpulmonary in 20 patients. Two thirds were situated immediately below some part of the right coronary leaflet with prolapse of that leaflet. Most of the remainder were below the right commissure or the anterior part of the noncoronary leaflet with prolapse of one or both adjacent leaflets. Associated structural defects, usuaUy including some fusion at a commissure, were present in 18 of the 70 patients and occurred more often with a ventricular septal defect in or below the commissure between the right and noncoronary leaflets (p < 0.001). FoUow-up ranged from 1.9 to 19.6 (mean 9.8) years. There were no early deaths or cases of atrioventricular block, but there were two late deaths. Patient survival rate was 96 % at 10 years. Freedom from valvuloplasty failure and freedom from reoperation were 76% and 85%, respectively, at 10 years. The major predictor for failure by multivariate analysis was the presence of an associated structural defect (p < 0.01). Age at repair and position of the ventricular septal defect were not significant risk factors. We conclude that aortic valvuloplasty produces good palliation in most children. The few failures occurred early and chiefly in patients with associated structural valve defects that occurred more frequently in children who had a ventricular septal defect in the right commissure, where both the right and noncoronary leaflets may be affected. (J THoRAc CARDIOVASC SURG 1992;103:276-81)
George A. Trusler, MD, William G. Williams, MD, Jeffrey F. Smallhorn, MD, and Robert M. Freedom, MD, Toronto, Ontario, Canada
Our experience with repair of the aortic insufficiency (AI) that sometimes occurs in association with ventricular septal defect (VSD) dates back to 1968.1, 2 Intermediateresults were reported in 1983.3 Recent interest in the malformation and the results of aortic valvuloplasty prompted this review. Patients and methods From 1968 to the end of 1988, 70 patients had repair of the combined defect, VSD and AI. Ages at operation ranged from 1.96 to 35.9 (mean 10.1) years. Eleven patients were less than 5 years of age and four were more than 18years old. There were 46 male and 24 female patients. Pulmonary-systemicflow ratios ranged from 1:1 to 3.4:1 (mean 1.7:1).Preoperative assessment From the Divisions of Cardiovascular Surgery and Cardiology, The Hospital for SickChildren, and the Departments of Surgery and Pediatrics, University of Toronto, Toronto, Ontario, Canada. Received for publication May 7,1991. Accepted for publication Sept.24, 1991. Address for reprints: George A.Trusler, MD,Suite 1525, 555 UniversityAve., Toronto, Ontario, CanadaM5G 1X8. 12/1/34006
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included symptomatology, cardiothoracic ratio, aortic diastolic murmur, and arterial pulse pressure. The degree of AI was established by cardiac catheterization and angiography in all but one patient and was assessed as mild, moderate, or severe. Follow-up ranged from 1.9 to 19.6 (mean 9.8 ± 5.3) years. Postoperatively,the amount of AI was assessed by the cardiologist from clinical parameters, and in 54 patients this was verifiedby angiography or Dopplercolorflowechocardiography,or both, and graded as described by Hunt and associates." Valvuloplastyfailure was deemed to have occurred if the AI was the same as or worse than preoperatively or if the AI was moderate or severe in degree even if there had been some improvement. The LIFETEST procedure in the Statistical Analysis System (SAS) was used to produce specific KaplanMeier curves related to survival time, time to reoperation, and time to valvuloplastyfaflure.' The PHGLM procedure in SAS was used to identify important predictors of failure," The procedure fits the Cox? proportional hazards linear regression model to one dependent variable by means of the Breslow'' modificationfor tied data.P The dependent variable was failure time from the first surgical date. The possible prognostic or independent variables considered were age at valvuloplasty, whether or not a structural defect was present, the position of the VSD relative to the aortic valve,and whether the VSD was subpulmonary or supracristal. The cutoff point of significance was considered to be a probability value of 0.05.
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VSD-AI complex
Number 2 February 1992
subpulmonary Subcristal
1
1
16 11
2 17 14
5
2
Fig. 1. Position of the VSD relative to the aortic valve. Operation. Details of the operation included a median sternotomy incision, cardiopulmonary bypass, a left ventricular vent, moderate hypothermia, and, since January 1976, cardioplegia. The aortic valvuloplasty was performed first, by means of the technique described previously.? The prolapsed leaflet, usually the right, was inspected and measured with particular regard to the length of the free margin and its shape, thickness, and potential competence. If the prolapse was severe and the free margin greatly elongated, the valve leaflet was piicated at both of its commissures. If the elongation was less extensive the valve was plicated at one commissure, usually where the leaflet was shallow or thin and weak, and in such a way that the weak area was eliminated in the plication and the sinus deepened. This usually allowed the plicating suture to be placed in reasonably strong tissue, although in a few cases the sutures had to be placed in a relatively weak part of the valve and this may have resulted in some valvuloplasty failures. After the part of the valve to be plicated had been identified, small stay sutures were placed in the corpora arantii of the two normal valveleaflets and at the future center point of the prolapsed cusp. The length of the free margin of the prolapsed cusp was adjusted to equal the adjacent margins of the two normal leaflets and tacked with a fine silk suture to the adjacent leaflet atoneor both commissures to maintain the correct length. The excess leaflet tissue was folded into the sinus at the commissure and then sutured to the aortic wall with two or three mattress sutures. These sutures were reinforced by Teflon felt pledgets, one within the aortic sinus and one on the outside of the aortic wall, for security. The pledget within the sinus extended over the free margin of the folded valve tissue to prevent blood from dissecting the repair from the aortic wall. Occasionally, pericardium was used as a pledget material, particularly if only a small amount of valve was to be plicated. Finally, the commissure was reinforced with a tiny, 2 by 3 mm thin Dacron patch held with a mattress suture. After one or two additional whipping bites for security, both ends of this suture were passed through the aortic wall and tied over the external pledgets. The aortotomy incision was then closed, the aortic root perfused, and the right ventricle (or right atrium, or pulmonary artery) opened. Through the VSD the aortic valve was examined for competence. The VSD was closed with a Dacron patch in 63 patients and by direct suture in seven. Originally, the VSD
277
Fig. 2. VSDs in or below the right commissure frequently were associated with a combination of structural defects, including 5 to 8 mm fusion of the right and noncoronary leaflets, some deficiency of commissure attachment, and prolapse of both adjacent leaflets. was closed during a second period of aortic crossclamping, but lately it has been closed during the initial crossclamp period while additional cardioplegic solution was infused. The right ventricular approach to the VSD has the advantage of allowing inspection of the valvuloplasty result to estimate the degree of residual regurgitation. Because of increasing confidence in the valve repair, many VSDs are now closed through either the right atrium or the pulmonary artery and some through the aorta. In a few patients the method described by Spencer and colleagues? was used as part of the valvuloplasty. This simple technique uses an over-and-over running suture along the free edge of the redundant portion of the leaflet. Tying the two ends of the suture plicates the leaflet. We have used this only where it was necessary to shorten a leaflet margin by I or 2 rnrn, such as when the initial plication was judged insufficient. Both ends of the suture were passed through the aortic wall and tied outside to anchor the repair to the aortic wall. Operative findings. There was apparent counterclockwise rotation of the aortic root in the majority of patients. The right coronary leaflet was affected in 44 of the 70 patients, with elongation of its free margin and prolapse. The noncoronary leaflet was prolapsed in 16 patients and the left coronary leaflet in one. In six patients both the right and noncoronary leaflets appeared to be affected. In three patients the large cusp of a bicuspid valve was prolapsing: in two the combined right and left leaflets and in one the combined right and noncoronary leaflets. The VSD was subpulmonary (supracristal, doubly committed, subarterial) in 20 patients and subcristal (perimembranous) in the other 50. The size varied from 1.5 to 20 (mean 10) mm true diameter as measured from the right ventricular aspect. The subcristal VSD was often up in the crista, anterior to the commissure between the septal and anterior leaflets of the tricuspid valve rather than in a true perimembranous position. Twenty-one patients had sufficient infundibular stenosis to warrant excision, including one with absent pulmonary valve syndrome. From the aortic aspect, the VSD was most commonly (47 cases) immediately below some part of the right coronary leaflet of the aortic valve (Fig. I). One was directly below the anterior commissure between the right and left coronary leaflets, and 21 were below the anatomic right commissure or below the
The Journal of Thoracic and Cardiovascular Surgery
Trusler et al.
27 8
100
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76%
%
Results
50
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1'\=
o
38
28
11
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-'--_ _---'
5
20
10 15 Time after Valvuloplasty (Years)
Fig. 3. Kaplan-Meier curve showing prevalence of valvuloplasty failure.
Table I. Results in 70 patients Regurgitation
Noneup to trivial Mild Moderate Severe Repeat repair Aortic valve replacement Death
right commissure were generally smaller than the othersmean diameter 7.1 mm compared with 10.9 mm (p = 0.001). One other patient had an associated sinus of Valsalva aneurysm and one a perforation of the right aortic cusp.
Preop.
Early
Late
o
46
29 27
47
18 5
11
I
12
5 3 4 4 2
Atrioventricular block, 0; subacute bacterial endocarditis, 3; persistent YSD, 9.
anterior portion of the noncoronary leaflet. In one child the positionof the tiny VSD was not recorded,Almost invariablythe affected leaflet was that immediately adjacent to the defect. VSDs under the midportion of the right leafletor further to the left were usually subpulmonary and those to the right were usually subcristal. In 18 children the valveabnormality was more complexthan simpleprolapsein that there was an additional structural defect, typically involving the attachment of one or more leaflets at a commissure. The VSD was subpulmonary in three and subcristal in IS. The aortic valvewas bicuspidin three patients, In two children all the aortic leafletswere thin and fenestrated near the commissural attachments, and in another two children one leafletwas unusuallyshallowwith a lowattachment at the right commissure. The remaining II structural defects had the following in common: a generally small VSD immediately below the anatomic right commissure (with some extension under an adjacent leafletin three cases), 5 to 8 mm fusionof the right and noncoronaryleafletsat the commissure,and frequently some deficiencyof attachment and a shallow appearance as if there was a loss of leaflet substance at the commissure (Fig. 2). We refer to this combination as a right commissure defect. In six of II both the right and noncoronary leaflets appeared elongated and prolapsed. Fourteen of the 70 patients had VSDs directly in or belowthe anatomic right commissure and 10 of these had associated structural valve defects, compared with only eight structural defects in the other 55 patients (p < 0.00 I). The VSDs in the
The repair was considered to have failed when the AI was unimproved over the preoperative status or if there was either moderate or severe AI. Immediately after repair, the condition of 63 patients was improved (Table I). Seven patients had valvuloplasty failure. One of the seven had a second repair with persistent improvement and another had an early aortic valve replacement. There were no early deaths or instances of atrioventricular block. Length of follow-up varied from 1.9 to 19.6 (mean 9.8) years. Fifty-six patients were considered to have good late results with no regurgitation to mild regurgitation on examination, although two of these had required a second repair and their valvuloplasties were considered to have failed. In total, valvuloplasty failure occurred in 16 of 70 patients; at times, failure occurred up to 5 years after operation, with no failures beyond that time. The probability of remaining free of valvuloplasty failure was 76% at 5 years (95% CL,* 65% to 86%) (Fig. 3). There were seven failures in 41 children who were younger than 10 years of age at repair and two failures in nine patients older than 15 years of age. The degree of preoperative AI was identical in patients who had successful or unsuccessful repair. . The original valve deformity in seven of the 16 patients whose valvuloplasties failed was simple prolapse of a valve leaflet, whereas in the other nine there were other structural defects as well. By multivariate analysis, the presence of a structural defect was associated with an increased risk of valvuloplasty failure (p < 0.01 ). Age at first operation, the position of the VSD, whether subpulmonary or subcristal, and its position in relation to the aortic valve were not significant predictors of valvuloplasty failure. Plicating a leaflet at both commissures rather than one did not decrease the likelihood of valve failure significantly. In two children subacute bacterial endocarditis was an apparent cause of failure, and in a third the endocarditis occurred later. Nine patients required reoperation. Four had a second repair and one had repair of a false aneurysm of the ascending aorta. Five children required aortic valve replacement, including the child who had the aortic aneurysm. Freedom from reoperation was 85% at 15 years (95% CL, 75% t094%) (Fig.4). Six patients with valvuloplasty failure have had no further operations. Five *CL = Confidence limits.
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Number 2 February 1992
have only moderate AI, insufficient to warrant valve replacement, and the other one, although free of symptoms, is reported to have severe AI and will likely require valve replacement in the future. There were two late deaths, 4 years and 6 years after the original repair. Patient survival rate at 6 years and beyond was 96% (95% ci, 90% to 100%) (Fig. 5). A review of the 18 patients with structural defects revealed that two of the three children with bicuspid valves maintained improvement and only one had valvuloplasty failure. Neither of the two children with thin fenestrated leaflets and one of the two with low shallow cusps were in improved condition on late follow-up, whereas six of the 11 children with right commissure defects continued to be in improved condition. Small recurrent VSDs were identified in nine patients. Two were closed subsequently at reoperations for repair or aortic valve replacement.
100 "'"'-... J
T
279
,
~I~--~~~---------- 85%
%
50
Tl= 0
46 5
0
33
11
10 15 Time after Valvuloplasty (Years)
20
Fig. 4. Freedom from reoperation.
Discussion The earliest attempts at valvuloplasty for the VSD-AI complex were by Garamella.!'' Starr, II and their associates.Plauth and his colleagues 12 and Frater I 3 emphasized the importance of and methods for accurate measurement of 'the free margin of the prolapsed aortic cusp. Security of the repair seemed to be a key requirement, and leafletplication at one or both commissures with pledgetsupported sutures produced acceptable results.? Tatsuno and coworkers!" reviewed their extensive experience in 1973, demonstrating the relationship between the positionof the VSD and the affected portion ofaortic cusp and indicating how this could be identified angiographically. Recently, Momma and colleagues.P from the same institution, updated this series and described the natural history of the subarterial (subpulmonary) VSD with reference to aortic valve prolapse. An intriguing feature of the anatomy of the VSD-AI complex was the occurrence of some degree of dextro-rotationof the aortic root in many patients. 16-18 The counterclockwise rotation was such that the anatomic right commissure between the right and noncoronary aortic leaflets was oriented at 12:30 rather than at 2:30 o'clock. Additionally, the most common subcristal VSD that we identified in this complex was not in the usual perimembranousposition but actually up in the lower border of the crista and anterior to the anteroseptal commissure of the tricuspid valve, with a narrow bar of muscle separating theVSD and the tricuspid valve, as if the VSD had rotated withthe aortic root. This was described by Van Praagh and Mclvamara!? and by Barratt-Boyes.P Because aortic valve prolapse is relatively common in patientswith subpulmonary (doubly committed subarte-
%
50
Ol.-
o
'1=
37
51 .L..-
5
13
.L..-
10
.L..-_ _---.J
15
20
Time after Valvuloplasty (Years)
Fig. 5. Kaplan-Meier curve of patient survival.
rial) VSDs, children with such VSDs should have serial echocardiograms to detect aortic valve prolapse. The presence of prolapse warrants early repair of the VSD to prevent AI. We have seen only one instance in which AI developed later, after the VSD was repaired. In reality, there are very few patients with small subpulmonary VSDs who are treated medically for a long period. Most ofthe VSDs are either large or associated with valve prolapse and therefore are closed surgically. In the presence of AI, if more than trivial, the valve is repaired when the VSD is closed. It makes sense to repair the valve early before excess deformity, stretching, or degenerative changes occur in the valve leaflet. We say this despite the fact that some patients with longstanding AI have excellent results after valvuloplasty and it is easier to identify prolapse and accurately tailor the plication in a larger patient. A more conservative approach was reported by Boone and colleagues.I' who found that closing the VSDalone, without valvuloplasty, frequently resulted in reduction of the associated AI. Although our initial results were encouraging, at late
28 0
Trusler et al.
follow-up 2 to 19 years later there had been 16 valvuloplasty failures, all occurring within 5 years of repair. The actuarial freedom from valvuloplasty failure at 5 and 15 years was 76% and the freedom from reoperation at 10 years was 85%. Results presented by a number of other groups22-25 are similar but in general indicate some continuing late failures. In the 38 patients we have followed up beyond 5 years there was no further late failure, which suggests that the problem was an inadequate or weak repair that became evident early. Those patients with a secure and accurate repair remained well, which suggests that the repair itself, with pledgets within the aortic sinus, did not create an ongoing late problem, at least over a two-decade period. We believe that all details of the repair, accurate measurement, plication with pledgetsupported sutures, and reinforcement of the commissure with a tiny Dacron patch, may be necessary to produce optimum results. On multivariate analysis, the only significant risk factor for failure of the valvuloplasty was the presence of a structural abnormality of one or more valve leaflets (p < 0.01), usually at a commissure and in association with prolapse of one or two leaflets. The problem of defective commissure development was described by Van Praagh and McNamaral'' and has been noted by others. 2,25 It was present in 18 of our 70 patients, and nine had late valvuloplasty failure. VSDs in the right commissure between the right coronary and the noncoronary leaflets were associated with structural defects more often than VSDs in other positions (p < 0.001); however, a VSD in the right commissure position was not a significant predictor of valvuloplasty failure. The structural defects included a variety of problems including bicuspid valves, thin, fenestrated leaflets, and deficient leaflets with an abnormally low, shallow commissural attachment. Eleven children had an abnormality that we have termed a right commissure defect. In six, both the right and noncoronary leaflets were considered to be elongated and prolapsed, whereas in the other five only one leaflet was thought to be so affected. Results were generally better when both leaflets were plicated, which suggests that possibly both leaflets (right and noncoronary) were affected more often than was recognized. This should be suspected whenever the VSD is situated in or below the right commissure or in a position to affect both leaflets. Others have reported the need to plicate two leaflets.P Accurate measurement and repair is more difficult when two leaflets are prolapsed. When the fused right commissure is poorly formed or weak and the leaflets are relatively normal at the other commissures, the main plication should be done at the right commissure, both leaf-
The Journal of Thoracic and Cardiovascular Surgery
lets being shortened until one is competent. The still incompetent leaflet is then plicated at its other commissure with reference to the length of the adjacent portion of the normal cusp. Recently several groups have reported experience with alternative techniques of aortic valve repair. Chauvaud and colleagues-" and Carpentier'" described a triangular resection of the free border of the prolapsed leaflet in combination with a circumferential annuloplasty. In a series of adults, Cosgrove and coworkersf used a similar triangular resection but in association with a commissural mattress suture that was first applied by Duran's group'? and was somewhat reminiscent of the commissure-deepening suture described by Ankeney, Tzeng, and Liebman.l" In some cases, particularly in patients with a bicuspid aortic valve, this technique has some appeal because the central portion of the cusp is often sufficiently thick to hold sutures and the valve deformity seems mainly central as well. The same may hold true for some patients with uncomplicated VSD-AI complex, but many such anomalies have an appearance that suggests that the abnormal part of the leaflet is adjacent to the commissure where it appears thin and attenuated. This is the area that we have attempted to exclude by plication to strengthen the leaflet and, at the same time, deepen its attachment at the commissure. It is quite possible, however, that in concentrating on the commissural parts of the leaflets, we may have missed instances in which the major defect was in the center. Continued careful examination of the leaflet shape and structure in each patient should lead to improved selection of valvuloplasty techniques. In patients with longstanding Alone would expect some dilation of the aortic root, and annuloplasty as part of the repair might be worthwhile. Although we have not done annuloplasties, some of our older patients have had surprisingly good early and late results with valvuloplasty alone. Age was not a significant predictor for valvuloplasty failure, and we have not encountered patients in whom the prolapsed valve was fixed in position and could not be plicated with benefit unless there was associated structural deformity. On the other hand, there is room for improvement in results, and a judicious annuloplasty or deepening of the commissure may be helpful in some patients, particularly those with longstanding AI. In general, the results of aortic valvuloplasty have been good. Fifty-six of the 70 children remain in improved condition (two after a second repair) as long as 19 years after repair, and 28 of these have no AI or trivial AI. Valvuloplasty failure occurred in 16, but all within the first 5 years, which suggests a basic inadequacy of repair rather than a late problem. Failures were significantly more frequent in the children with associated structural
Volume 103 Number 2 February 1992
defects at the commissures. Recognition and appropriate repair of the special problems associated with these defects should improve results. We expressour appreciation to Valerie Daniels for assistance in preparation of the manuscript and to Henriette Sherret, PhD, for help with the statistical review. REFERENCES 1. Trusler GA, Discussion of Treasure RL, Hopeman AR, Jahnke EJ, Green DC, Czarnecki SW. Ventricular septal defect with aortic insufficiency. Ann Thorac Surg 1971; 12:411. 2. Trusler GA, Moos CAF, Kidd BSL. Repair of ventricular septal defect with aortic insufficiency. J THORAC CARDIO. VASC SURa 1973;66:394-403. 3. Trusler GA, Williams WG, Wood AE. Aortic valvuloplasty. In: Cohn L, ed. Modern technics in surgery. New York: Futura, 1983:561-8. 4. Hunt D, Baxley WA, Kennedy JW, Judge TP, Williams JE, Dodge HT. Quantitative evaluation of cine-aortography in the assessment of aortic regurgitation. Am J Cardi01 1973;31:696-700. 5. SAS Institute Inc. The LIFETEST procedure. In: SAS Users Guide: Statistics, version 5 edition. Cary, North Carolina: 1985:529-57. 6. SAS Institute Inc. The PHGLM procedure. In: SUGI supplementary library users guide, version 5 edition. Cary, North Carolina: 1986:437-66. 7. Cox DR. Regression models and life tables. J R Stat Soc B 1972;34:187-220. 8. BreslowNE. Covariance analysis of censored survival data. Biometrics 1974;30:89-99. 9. Spencer Fe, Doyle EF, Danilowicz DA, Bahnson HT, Weldon CS. Long-term evaluation of aortic valvuloplasty for aortic insufficiency and ventricular septal defect. J THORAC CARDlOVASC SURa 1973;65:15-31. 10. Garamella JJ, Cruz AB, Heupel WH, Dahl JC, Jensen NK, Berman R. Ventricular septal defect with aortic incompetence:successfulsurgical correction of both defects by the transaortic approach. Am J Cardiol 1960;5:26672. 11. Starr A, Menasche V, Dotter C. Surgical correction of aortic insufficiency associated with ventricular septal defect. Surg Gynecol Obstet 1960;3:71-6. 12. Plauth WH, Braunwald E, Rockoff SD, Mason DT, Morrow AG. Ventricular septal defect and aortic regurgitation. Am J Med 1965;39:552-67. 13. Frater RWM. The prolapsing aortic cusp. Ann Thorac Surg 1967;3:63-7. 14. Tatsuno K, Konno S, Ando M, Sakakibara S. Pathogenic mechanisms of prolapsing aortic valve and aortic regurgitation associated with ventricular septal defect. Circulation 1973;48:1028-37.
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15. Momma K, Toyama K, Takao A, et al. Natural history of subarterial infundibular ventricular septal defect. Am Heart J 1984;108:1312-17. 16. Keck EWO, Ongley PA, Kincaid OW, Swan HJC. Ventricular septal defect with aortic insufficiency. Circulation 1963;27:203-18. 17. Nadas AS, Thilenius OG, La Farge CG, Hauck AJ. Ventricular septal defect with aortic regurgitation. Circulation 1964;29:862-73. 18. Somerville J, Brandao A, Ross DN. Aortic regurgitation with ventricular septal defect. Circulation 1970;41:317-30. 19. Van Praagh R, McNamara 11. Anatomic types of ventricular septal defect with aortic insufficiency. Am Heart J 1968;75:604-19. 20. Barratt-Boyes BG. Ventricular septal defect. In: Kirklin JW, Barratt-Boyes BG, eds. Cardiac surgery. New York: John Wiley, 1986:599-664. 21. Boone JW, Vincent RN, Dooley KJ, Williams WHo Ventricular septal defect closure without aortic valve plication in patients with aortic valve prolapse. Am J Cardiol 1990; 65:1271-3. 22. Okita Y, Miki S, Kusuhara K, et al. Long-term results of aortic valvuloplasty for aortic regurgitation associated with ventricular septal defect. J THORAC CARDIOVASC SURa 1988;96:769-74. 23. Maehara T, Blackstone EH, Kirklin JW, Kirklin JK, Pacifico AD, Colvin E. The results of the Trusler operation for ventricular septal defect and aortic valvar incompetence. In: Crupi G, Parenzan L, Anderson RH, eds. Perspectives in pediatric cardiology. vol. 2, part 1. New York: Futura, 1989:66-70. 24. Leung MP, Beerman LB, Siewers RD, Bahnson HT, Zuberbuhler JR. Long-term follow-up after aortic valvuloplasty and defect closure in ventricular septal defect with aortic regurgitation. Am J Cardiol 1987;60:890-4. 25. Rhodes LA, Keane JF, Keane JP, et al. Long follow-up (to 43 years) of ventricular septal defect with audible aortic regurgitation. Am J CardioI1990;66:340-5. 26. Chauvaud S, Serraf A, Mihaileanu S, et al. Ventricular septal defect associated with aortic valve incompetence: results of two surgical managements. Ann Thorac Surg 1990;49:875-80. 27. Carpentier A. Cardiac valve surgery-the "French correction." J THORAC CARDIOVASC SURa 1983;86:323-37. 28. Cosgrove DM, Rosenkranz ER, Hendren WG, BartlettJC, Stewart WJ. Valvuloplasty for aortic insufficiency. J THORAC CARDIOVASC SURa [In press]. 29. Duran CMG, Alonso J, Gaite L, et al. Long-term results of conservative repair of rheumatic aortic valve insufficiency. Eur J Cardiothoracic Surg 1988;2:217-23. 30. Ankeney JL, Tzeng TS, Liebman J. Surgical therapy for congenital aortic valvular stenosis.J THORAC CARDIOVASC SURa 1983;85:41-8.
George A. Trusler, MD, William G. Williams, MD, Jeffrey F. Smallhorn, MD, and Robert M. Freedom, MD, Toronto, Ontario, Canada
Our experience with repair of the aortic insufficiency (AI) that sometimes occurs in association with ventricular septal defect (VSD) dates back to 1968.1, 2 Intermediateresults were reported in 1983.3 Recent interest in the malformation and the results of aortic valvuloplasty prompted this review. Patients and methods From 1968 to the end of 1988, 70 patients had repair of the combined defect, VSD and AI. Ages at operation ranged from 1.96 to 35.9 (mean 10.1) years. Eleven patients were less than 5 years of age and four were more than 18years old. There were 46 male and 24 female patients. Pulmonary-systemicflow ratios ranged from 1:1 to 3.4:1 (mean 1.7:1).Preoperative assessment From the Divisions of Cardiovascular Surgery and Cardiology, The Hospital for SickChildren, and the Departments of Surgery and Pediatrics, University of Toronto, Toronto, Ontario, Canada. Received for publication May 7,1991. Accepted for publication Sept.24, 1991. Address for reprints: George A.Trusler, MD,Suite 1525, 555 UniversityAve., Toronto, Ontario, CanadaM5G 1X8. 12/1/34006
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included symptomatology, cardiothoracic ratio, aortic diastolic murmur, and arterial pulse pressure. The degree of AI was established by cardiac catheterization and angiography in all but one patient and was assessed as mild, moderate, or severe. Follow-up ranged from 1.9 to 19.6 (mean 9.8 ± 5.3) years. Postoperatively,the amount of AI was assessed by the cardiologist from clinical parameters, and in 54 patients this was verifiedby angiography or Dopplercolorflowechocardiography,or both, and graded as described by Hunt and associates." Valvuloplastyfailure was deemed to have occurred if the AI was the same as or worse than preoperatively or if the AI was moderate or severe in degree even if there had been some improvement. The LIFETEST procedure in the Statistical Analysis System (SAS) was used to produce specific KaplanMeier curves related to survival time, time to reoperation, and time to valvuloplastyfaflure.' The PHGLM procedure in SAS was used to identify important predictors of failure," The procedure fits the Cox? proportional hazards linear regression model to one dependent variable by means of the Breslow'' modificationfor tied data.P The dependent variable was failure time from the first surgical date. The possible prognostic or independent variables considered were age at valvuloplasty, whether or not a structural defect was present, the position of the VSD relative to the aortic valve,and whether the VSD was subpulmonary or supracristal. The cutoff point of significance was considered to be a probability value of 0.05.
Volume 103
VSD-AI complex
Number 2 February 1992
subpulmonary Subcristal
1
1
16 11
2 17 14
5
2
Fig. 1. Position of the VSD relative to the aortic valve. Operation. Details of the operation included a median sternotomy incision, cardiopulmonary bypass, a left ventricular vent, moderate hypothermia, and, since January 1976, cardioplegia. The aortic valvuloplasty was performed first, by means of the technique described previously.? The prolapsed leaflet, usually the right, was inspected and measured with particular regard to the length of the free margin and its shape, thickness, and potential competence. If the prolapse was severe and the free margin greatly elongated, the valve leaflet was piicated at both of its commissures. If the elongation was less extensive the valve was plicated at one commissure, usually where the leaflet was shallow or thin and weak, and in such a way that the weak area was eliminated in the plication and the sinus deepened. This usually allowed the plicating suture to be placed in reasonably strong tissue, although in a few cases the sutures had to be placed in a relatively weak part of the valve and this may have resulted in some valvuloplasty failures. After the part of the valve to be plicated had been identified, small stay sutures were placed in the corpora arantii of the two normal valveleaflets and at the future center point of the prolapsed cusp. The length of the free margin of the prolapsed cusp was adjusted to equal the adjacent margins of the two normal leaflets and tacked with a fine silk suture to the adjacent leaflet atoneor both commissures to maintain the correct length. The excess leaflet tissue was folded into the sinus at the commissure and then sutured to the aortic wall with two or three mattress sutures. These sutures were reinforced by Teflon felt pledgets, one within the aortic sinus and one on the outside of the aortic wall, for security. The pledget within the sinus extended over the free margin of the folded valve tissue to prevent blood from dissecting the repair from the aortic wall. Occasionally, pericardium was used as a pledget material, particularly if only a small amount of valve was to be plicated. Finally, the commissure was reinforced with a tiny, 2 by 3 mm thin Dacron patch held with a mattress suture. After one or two additional whipping bites for security, both ends of this suture were passed through the aortic wall and tied over the external pledgets. The aortotomy incision was then closed, the aortic root perfused, and the right ventricle (or right atrium, or pulmonary artery) opened. Through the VSD the aortic valve was examined for competence. The VSD was closed with a Dacron patch in 63 patients and by direct suture in seven. Originally, the VSD
277
Fig. 2. VSDs in or below the right commissure frequently were associated with a combination of structural defects, including 5 to 8 mm fusion of the right and noncoronary leaflets, some deficiency of commissure attachment, and prolapse of both adjacent leaflets. was closed during a second period of aortic crossclamping, but lately it has been closed during the initial crossclamp period while additional cardioplegic solution was infused. The right ventricular approach to the VSD has the advantage of allowing inspection of the valvuloplasty result to estimate the degree of residual regurgitation. Because of increasing confidence in the valve repair, many VSDs are now closed through either the right atrium or the pulmonary artery and some through the aorta. In a few patients the method described by Spencer and colleagues? was used as part of the valvuloplasty. This simple technique uses an over-and-over running suture along the free edge of the redundant portion of the leaflet. Tying the two ends of the suture plicates the leaflet. We have used this only where it was necessary to shorten a leaflet margin by I or 2 rnrn, such as when the initial plication was judged insufficient. Both ends of the suture were passed through the aortic wall and tied outside to anchor the repair to the aortic wall. Operative findings. There was apparent counterclockwise rotation of the aortic root in the majority of patients. The right coronary leaflet was affected in 44 of the 70 patients, with elongation of its free margin and prolapse. The noncoronary leaflet was prolapsed in 16 patients and the left coronary leaflet in one. In six patients both the right and noncoronary leaflets appeared to be affected. In three patients the large cusp of a bicuspid valve was prolapsing: in two the combined right and left leaflets and in one the combined right and noncoronary leaflets. The VSD was subpulmonary (supracristal, doubly committed, subarterial) in 20 patients and subcristal (perimembranous) in the other 50. The size varied from 1.5 to 20 (mean 10) mm true diameter as measured from the right ventricular aspect. The subcristal VSD was often up in the crista, anterior to the commissure between the septal and anterior leaflets of the tricuspid valve rather than in a true perimembranous position. Twenty-one patients had sufficient infundibular stenosis to warrant excision, including one with absent pulmonary valve syndrome. From the aortic aspect, the VSD was most commonly (47 cases) immediately below some part of the right coronary leaflet of the aortic valve (Fig. I). One was directly below the anterior commissure between the right and left coronary leaflets, and 21 were below the anatomic right commissure or below the
The Journal of Thoracic and Cardiovascular Surgery
Trusler et al.
27 8
100
r------------------.., I
76%
%
Results
50
OL-
1'\=
o
38
28
11
J....-
J....-
-'--_ _---'
5
20
10 15 Time after Valvuloplasty (Years)
Fig. 3. Kaplan-Meier curve showing prevalence of valvuloplasty failure.
Table I. Results in 70 patients Regurgitation
Noneup to trivial Mild Moderate Severe Repeat repair Aortic valve replacement Death
right commissure were generally smaller than the othersmean diameter 7.1 mm compared with 10.9 mm (p = 0.001). One other patient had an associated sinus of Valsalva aneurysm and one a perforation of the right aortic cusp.
Preop.
Early
Late
o
46
29 27
47
18 5
11
I
12
5 3 4 4 2
Atrioventricular block, 0; subacute bacterial endocarditis, 3; persistent YSD, 9.
anterior portion of the noncoronary leaflet. In one child the positionof the tiny VSD was not recorded,Almost invariablythe affected leaflet was that immediately adjacent to the defect. VSDs under the midportion of the right leafletor further to the left were usually subpulmonary and those to the right were usually subcristal. In 18 children the valveabnormality was more complexthan simpleprolapsein that there was an additional structural defect, typically involving the attachment of one or more leaflets at a commissure. The VSD was subpulmonary in three and subcristal in IS. The aortic valvewas bicuspidin three patients, In two children all the aortic leafletswere thin and fenestrated near the commissural attachments, and in another two children one leafletwas unusuallyshallowwith a lowattachment at the right commissure. The remaining II structural defects had the following in common: a generally small VSD immediately below the anatomic right commissure (with some extension under an adjacent leafletin three cases), 5 to 8 mm fusionof the right and noncoronaryleafletsat the commissure,and frequently some deficiencyof attachment and a shallow appearance as if there was a loss of leaflet substance at the commissure (Fig. 2). We refer to this combination as a right commissure defect. In six of II both the right and noncoronary leaflets appeared elongated and prolapsed. Fourteen of the 70 patients had VSDs directly in or belowthe anatomic right commissure and 10 of these had associated structural valve defects, compared with only eight structural defects in the other 55 patients (p < 0.00 I). The VSDs in the
The repair was considered to have failed when the AI was unimproved over the preoperative status or if there was either moderate or severe AI. Immediately after repair, the condition of 63 patients was improved (Table I). Seven patients had valvuloplasty failure. One of the seven had a second repair with persistent improvement and another had an early aortic valve replacement. There were no early deaths or instances of atrioventricular block. Length of follow-up varied from 1.9 to 19.6 (mean 9.8) years. Fifty-six patients were considered to have good late results with no regurgitation to mild regurgitation on examination, although two of these had required a second repair and their valvuloplasties were considered to have failed. In total, valvuloplasty failure occurred in 16 of 70 patients; at times, failure occurred up to 5 years after operation, with no failures beyond that time. The probability of remaining free of valvuloplasty failure was 76% at 5 years (95% CL,* 65% to 86%) (Fig. 3). There were seven failures in 41 children who were younger than 10 years of age at repair and two failures in nine patients older than 15 years of age. The degree of preoperative AI was identical in patients who had successful or unsuccessful repair. . The original valve deformity in seven of the 16 patients whose valvuloplasties failed was simple prolapse of a valve leaflet, whereas in the other nine there were other structural defects as well. By multivariate analysis, the presence of a structural defect was associated with an increased risk of valvuloplasty failure (p < 0.01 ). Age at first operation, the position of the VSD, whether subpulmonary or subcristal, and its position in relation to the aortic valve were not significant predictors of valvuloplasty failure. Plicating a leaflet at both commissures rather than one did not decrease the likelihood of valve failure significantly. In two children subacute bacterial endocarditis was an apparent cause of failure, and in a third the endocarditis occurred later. Nine patients required reoperation. Four had a second repair and one had repair of a false aneurysm of the ascending aorta. Five children required aortic valve replacement, including the child who had the aortic aneurysm. Freedom from reoperation was 85% at 15 years (95% CL, 75% t094%) (Fig.4). Six patients with valvuloplasty failure have had no further operations. Five *CL = Confidence limits.
Volume 103
VSD-AI complex
Number 2 February 1992
have only moderate AI, insufficient to warrant valve replacement, and the other one, although free of symptoms, is reported to have severe AI and will likely require valve replacement in the future. There were two late deaths, 4 years and 6 years after the original repair. Patient survival rate at 6 years and beyond was 96% (95% ci, 90% to 100%) (Fig. 5). A review of the 18 patients with structural defects revealed that two of the three children with bicuspid valves maintained improvement and only one had valvuloplasty failure. Neither of the two children with thin fenestrated leaflets and one of the two with low shallow cusps were in improved condition on late follow-up, whereas six of the 11 children with right commissure defects continued to be in improved condition. Small recurrent VSDs were identified in nine patients. Two were closed subsequently at reoperations for repair or aortic valve replacement.
100 "'"'-... J
T
279
,
~I~--~~~---------- 85%
%
50
Tl= 0
46 5
0
33
11
10 15 Time after Valvuloplasty (Years)
20
Fig. 4. Freedom from reoperation.
Discussion The earliest attempts at valvuloplasty for the VSD-AI complex were by Garamella.!'' Starr, II and their associates.Plauth and his colleagues 12 and Frater I 3 emphasized the importance of and methods for accurate measurement of 'the free margin of the prolapsed aortic cusp. Security of the repair seemed to be a key requirement, and leafletplication at one or both commissures with pledgetsupported sutures produced acceptable results.? Tatsuno and coworkers!" reviewed their extensive experience in 1973, demonstrating the relationship between the positionof the VSD and the affected portion ofaortic cusp and indicating how this could be identified angiographically. Recently, Momma and colleagues.P from the same institution, updated this series and described the natural history of the subarterial (subpulmonary) VSD with reference to aortic valve prolapse. An intriguing feature of the anatomy of the VSD-AI complex was the occurrence of some degree of dextro-rotationof the aortic root in many patients. 16-18 The counterclockwise rotation was such that the anatomic right commissure between the right and noncoronary aortic leaflets was oriented at 12:30 rather than at 2:30 o'clock. Additionally, the most common subcristal VSD that we identified in this complex was not in the usual perimembranousposition but actually up in the lower border of the crista and anterior to the anteroseptal commissure of the tricuspid valve, with a narrow bar of muscle separating theVSD and the tricuspid valve, as if the VSD had rotated withthe aortic root. This was described by Van Praagh and Mclvamara!? and by Barratt-Boyes.P Because aortic valve prolapse is relatively common in patientswith subpulmonary (doubly committed subarte-
%
50
Ol.-
o
'1=
37
51 .L..-
5
13
.L..-
10
.L..-_ _---.J
15
20
Time after Valvuloplasty (Years)
Fig. 5. Kaplan-Meier curve of patient survival.
rial) VSDs, children with such VSDs should have serial echocardiograms to detect aortic valve prolapse. The presence of prolapse warrants early repair of the VSD to prevent AI. We have seen only one instance in which AI developed later, after the VSD was repaired. In reality, there are very few patients with small subpulmonary VSDs who are treated medically for a long period. Most ofthe VSDs are either large or associated with valve prolapse and therefore are closed surgically. In the presence of AI, if more than trivial, the valve is repaired when the VSD is closed. It makes sense to repair the valve early before excess deformity, stretching, or degenerative changes occur in the valve leaflet. We say this despite the fact that some patients with longstanding AI have excellent results after valvuloplasty and it is easier to identify prolapse and accurately tailor the plication in a larger patient. A more conservative approach was reported by Boone and colleagues.I' who found that closing the VSDalone, without valvuloplasty, frequently resulted in reduction of the associated AI. Although our initial results were encouraging, at late
28 0
Trusler et al.
follow-up 2 to 19 years later there had been 16 valvuloplasty failures, all occurring within 5 years of repair. The actuarial freedom from valvuloplasty failure at 5 and 15 years was 76% and the freedom from reoperation at 10 years was 85%. Results presented by a number of other groups22-25 are similar but in general indicate some continuing late failures. In the 38 patients we have followed up beyond 5 years there was no further late failure, which suggests that the problem was an inadequate or weak repair that became evident early. Those patients with a secure and accurate repair remained well, which suggests that the repair itself, with pledgets within the aortic sinus, did not create an ongoing late problem, at least over a two-decade period. We believe that all details of the repair, accurate measurement, plication with pledgetsupported sutures, and reinforcement of the commissure with a tiny Dacron patch, may be necessary to produce optimum results. On multivariate analysis, the only significant risk factor for failure of the valvuloplasty was the presence of a structural abnormality of one or more valve leaflets (p < 0.01), usually at a commissure and in association with prolapse of one or two leaflets. The problem of defective commissure development was described by Van Praagh and McNamaral'' and has been noted by others. 2,25 It was present in 18 of our 70 patients, and nine had late valvuloplasty failure. VSDs in the right commissure between the right coronary and the noncoronary leaflets were associated with structural defects more often than VSDs in other positions (p < 0.001); however, a VSD in the right commissure position was not a significant predictor of valvuloplasty failure. The structural defects included a variety of problems including bicuspid valves, thin, fenestrated leaflets, and deficient leaflets with an abnormally low, shallow commissural attachment. Eleven children had an abnormality that we have termed a right commissure defect. In six, both the right and noncoronary leaflets were considered to be elongated and prolapsed, whereas in the other five only one leaflet was thought to be so affected. Results were generally better when both leaflets were plicated, which suggests that possibly both leaflets (right and noncoronary) were affected more often than was recognized. This should be suspected whenever the VSD is situated in or below the right commissure or in a position to affect both leaflets. Others have reported the need to plicate two leaflets.P Accurate measurement and repair is more difficult when two leaflets are prolapsed. When the fused right commissure is poorly formed or weak and the leaflets are relatively normal at the other commissures, the main plication should be done at the right commissure, both leaf-
The Journal of Thoracic and Cardiovascular Surgery
lets being shortened until one is competent. The still incompetent leaflet is then plicated at its other commissure with reference to the length of the adjacent portion of the normal cusp. Recently several groups have reported experience with alternative techniques of aortic valve repair. Chauvaud and colleagues-" and Carpentier'" described a triangular resection of the free border of the prolapsed leaflet in combination with a circumferential annuloplasty. In a series of adults, Cosgrove and coworkersf used a similar triangular resection but in association with a commissural mattress suture that was first applied by Duran's group'? and was somewhat reminiscent of the commissure-deepening suture described by Ankeney, Tzeng, and Liebman.l" In some cases, particularly in patients with a bicuspid aortic valve, this technique has some appeal because the central portion of the cusp is often sufficiently thick to hold sutures and the valve deformity seems mainly central as well. The same may hold true for some patients with uncomplicated VSD-AI complex, but many such anomalies have an appearance that suggests that the abnormal part of the leaflet is adjacent to the commissure where it appears thin and attenuated. This is the area that we have attempted to exclude by plication to strengthen the leaflet and, at the same time, deepen its attachment at the commissure. It is quite possible, however, that in concentrating on the commissural parts of the leaflets, we may have missed instances in which the major defect was in the center. Continued careful examination of the leaflet shape and structure in each patient should lead to improved selection of valvuloplasty techniques. In patients with longstanding Alone would expect some dilation of the aortic root, and annuloplasty as part of the repair might be worthwhile. Although we have not done annuloplasties, some of our older patients have had surprisingly good early and late results with valvuloplasty alone. Age was not a significant predictor for valvuloplasty failure, and we have not encountered patients in whom the prolapsed valve was fixed in position and could not be plicated with benefit unless there was associated structural deformity. On the other hand, there is room for improvement in results, and a judicious annuloplasty or deepening of the commissure may be helpful in some patients, particularly those with longstanding AI. In general, the results of aortic valvuloplasty have been good. Fifty-six of the 70 children remain in improved condition (two after a second repair) as long as 19 years after repair, and 28 of these have no AI or trivial AI. Valvuloplasty failure occurred in 16, but all within the first 5 years, which suggests a basic inadequacy of repair rather than a late problem. Failures were significantly more frequent in the children with associated structural
Volume 103 Number 2 February 1992
defects at the commissures. Recognition and appropriate repair of the special problems associated with these defects should improve results. We expressour appreciation to Valerie Daniels for assistance in preparation of the manuscript and to Henriette Sherret, PhD, for help with the statistical review. REFERENCES 1. Trusler GA, Discussion of Treasure RL, Hopeman AR, Jahnke EJ, Green DC, Czarnecki SW. Ventricular septal defect with aortic insufficiency. Ann Thorac Surg 1971; 12:411. 2. Trusler GA, Moos CAF, Kidd BSL. Repair of ventricular septal defect with aortic insufficiency. J THORAC CARDIO. VASC SURa 1973;66:394-403. 3. Trusler GA, Williams WG, Wood AE. Aortic valvuloplasty. In: Cohn L, ed. Modern technics in surgery. New York: Futura, 1983:561-8. 4. Hunt D, Baxley WA, Kennedy JW, Judge TP, Williams JE, Dodge HT. Quantitative evaluation of cine-aortography in the assessment of aortic regurgitation. Am J Cardi01 1973;31:696-700. 5. SAS Institute Inc. The LIFETEST procedure. In: SAS Users Guide: Statistics, version 5 edition. Cary, North Carolina: 1985:529-57. 6. SAS Institute Inc. The PHGLM procedure. In: SUGI supplementary library users guide, version 5 edition. Cary, North Carolina: 1986:437-66. 7. Cox DR. Regression models and life tables. J R Stat Soc B 1972;34:187-220. 8. BreslowNE. Covariance analysis of censored survival data. Biometrics 1974;30:89-99. 9. Spencer Fe, Doyle EF, Danilowicz DA, Bahnson HT, Weldon CS. Long-term evaluation of aortic valvuloplasty for aortic insufficiency and ventricular septal defect. J THORAC CARDlOVASC SURa 1973;65:15-31. 10. Garamella JJ, Cruz AB, Heupel WH, Dahl JC, Jensen NK, Berman R. Ventricular septal defect with aortic incompetence:successfulsurgical correction of both defects by the transaortic approach. Am J Cardiol 1960;5:26672. 11. Starr A, Menasche V, Dotter C. Surgical correction of aortic insufficiency associated with ventricular septal defect. Surg Gynecol Obstet 1960;3:71-6. 12. Plauth WH, Braunwald E, Rockoff SD, Mason DT, Morrow AG. Ventricular septal defect and aortic regurgitation. Am J Med 1965;39:552-67. 13. Frater RWM. The prolapsing aortic cusp. Ann Thorac Surg 1967;3:63-7. 14. Tatsuno K, Konno S, Ando M, Sakakibara S. Pathogenic mechanisms of prolapsing aortic valve and aortic regurgitation associated with ventricular septal defect. Circulation 1973;48:1028-37.
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15. Momma K, Toyama K, Takao A, et al. Natural history of subarterial infundibular ventricular septal defect. Am Heart J 1984;108:1312-17. 16. Keck EWO, Ongley PA, Kincaid OW, Swan HJC. Ventricular septal defect with aortic insufficiency. Circulation 1963;27:203-18. 17. Nadas AS, Thilenius OG, La Farge CG, Hauck AJ. Ventricular septal defect with aortic regurgitation. Circulation 1964;29:862-73. 18. Somerville J, Brandao A, Ross DN. Aortic regurgitation with ventricular septal defect. Circulation 1970;41:317-30. 19. Van Praagh R, McNamara 11. Anatomic types of ventricular septal defect with aortic insufficiency. Am Heart J 1968;75:604-19. 20. Barratt-Boyes BG. Ventricular septal defect. In: Kirklin JW, Barratt-Boyes BG, eds. Cardiac surgery. New York: John Wiley, 1986:599-664. 21. Boone JW, Vincent RN, Dooley KJ, Williams WHo Ventricular septal defect closure without aortic valve plication in patients with aortic valve prolapse. Am J Cardiol 1990; 65:1271-3. 22. Okita Y, Miki S, Kusuhara K, et al. Long-term results of aortic valvuloplasty for aortic regurgitation associated with ventricular septal defect. J THORAC CARDIOVASC SURa 1988;96:769-74. 23. Maehara T, Blackstone EH, Kirklin JW, Kirklin JK, Pacifico AD, Colvin E. The results of the Trusler operation for ventricular septal defect and aortic valvar incompetence. In: Crupi G, Parenzan L, Anderson RH, eds. Perspectives in pediatric cardiology. vol. 2, part 1. New York: Futura, 1989:66-70. 24. Leung MP, Beerman LB, Siewers RD, Bahnson HT, Zuberbuhler JR. Long-term follow-up after aortic valvuloplasty and defect closure in ventricular septal defect with aortic regurgitation. Am J Cardiol 1987;60:890-4. 25. Rhodes LA, Keane JF, Keane JP, et al. Long follow-up (to 43 years) of ventricular septal defect with audible aortic regurgitation. Am J CardioI1990;66:340-5. 26. Chauvaud S, Serraf A, Mihaileanu S, et al. Ventricular septal defect associated with aortic valve incompetence: results of two surgical managements. Ann Thorac Surg 1990;49:875-80. 27. Carpentier A. Cardiac valve surgery-the "French correction." J THORAC CARDIOVASC SURa 1983;86:323-37. 28. Cosgrove DM, Rosenkranz ER, Hendren WG, BartlettJC, Stewart WJ. Valvuloplasty for aortic insufficiency. J THORAC CARDIOVASC SURa [In press]. 29. Duran CMG, Alonso J, Gaite L, et al. Long-term results of conservative repair of rheumatic aortic valve insufficiency. Eur J Cardiothoracic Surg 1988;2:217-23. 30. Ankeney JL, Tzeng TS, Liebman J. Surgical therapy for congenital aortic valvular stenosis.J THORAC CARDIOVASC SURa 1983;85:41-8.