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Ann Thorac Surg 1999;67:736-738
© 1999 The Society of Thoracic Surgeons


Original Articles

Outcome of surgical closure of doubly committed subarterial ventricular septal defect

Eugene K.W. Sim, FRCSa, Robert T. Grignani, IB Dipa, May-Ling Wong, M Med(Paed)a, Swee Chye Quek, M Med(Paed)a, Julia C.L. Wong, M Med(Paed)a, William C.L. Yip, FRCPb, Chuen Neng Lee, FRACSb

a Cardiac Department, National University Hospital, Singapore
b Gleneagles Medical Centre, Singapore

Accepted for publication July 20, 1998.

Address reprint requests to Dr Sim, Cardiac Department, National University Hospital, 5 Lower Kent Ridge Rd, Singapore 119074


    Abstract
 Top
 Abstract
 Introduction
 Material and methods
 Results
 Comment
 References
 
Background. From 1986 to March 1997, 128 patients diagnosed to have doubly committed subarterial ventricular septal defects (VSD) were reviewed. Patients with aortic regurgitation (AR), and aortic valve (AV) deformity or a large left-to-right shunt across the VSD were offered operation. Forty-five patients (27 men, 18 women) agreed to surgical closure of their VSDs.

Methods. Thirty-eight patients had VSD closure alone, and 7 had an additional AV repair. Other associated defects corrected at operation were closure of atrial septal defects, closure of other ventricular septal defects, ligation of patent ductus arteriosus, and repair of ruptured sinus Valsalva aneurysm.

Results. There was no mortality nor major morbidity associated with operation. In the 26 patients with AR and AV deformity preoperatively, valve repair was performed in 6 patients. The condition of AR improved in 4, and remained unchanged in 22 patients. In the 10 patients with a deformity of the AV and no AR preoperatively, the condition remained unchanged in 5 patients, from whom 1 had valve operation, but progressed in 5 patients postoperatively at a mean follow-up of 6.4 years. In 9 patients with no deformity of the AV and no AR preoperatively, there was no postoperative AR and no progress of valve deformity.

Conclusions. Excellent results were obtained with VSD closure and AV repair. Surgical closure of VSD, if performed before the onset of AV deformity, may prevent progressive AR. If AV repair is performed after the onset of AV deformity, progressive AR may not always be prevented.


    Introduction
 Top
 Abstract
 Introduction
 Material and methods
 Results
 Comment
 References
 
Doubly committed subarterial ventricular septal defects (DCSA VSD) differ from other ventricular septal defects. Spontaneous closure is a rarity and prolapse of an aortic cusp is a common feature, which progresses to aortic regurgitation (AR). The latter may necessitate aortic valve (AV) replacement. They are less common in the Occidental population as compared to the Oriental population [1, 2]. Up to 30% of ventricular septal defects in the Asian patients are DCSA VSDs [36].The natural history is not clearly defined and the timing of surgical closure has to be carefully planned to optimize benefit to the patient [3, 7]. We review all patients with surgical closure of the DCSA VSD to assess the relation of timing of operation with AV prolapse and AR.


    Material and methods
 Top
 Abstract
 Introduction
 Material and methods
 Results
 Comment
 References
 
Patient population
From January 1986 to March 1997, 128 patients diagnosed with DCSA VSD were seen at the National University Hospital, Singapore and Gleneagles Medical Centre, Singapore. Forty-five of them underwent surgical closure of their VSD in one of the two institutions. The diagnosis was made primarily on cross-sectional echocardiography with Doppler color flow. Cardiac catheterization was performed on 39 patients. The mean VSD size was 5.8 mm (2.0 to 11.0 mm), and with a mean pulmonary-to-systemic blood flow ratio (Qp:Qs) of 1.7:1 (1.1 to >4). Most patients had a left-to-right shunt across their VSD, 4 of whom had a bidirectional shunt. The AV was carefully studied to determine whether there were any abnormalities, and to grade the condition of AR if any. The AR was graded mild, moderate, or severe. There were 27 men and 18 women, and mean age at operation was 5.8 years (1 month to 21 years). The patients were classified into three groups. Group I (26 patients) were those with AR and deformity of the AV. Group II (10 patients) were those with AV deformity but without AR, and group III (9 patients) were those with neither AR nor deformities of the AV (Table 1).


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Table 1. Comparison of Ventricular Septal Defect Size and Shunt Size in Patients With Doubly Committed Subarterial Defects

 
Associated defects
Associated defects were present in 26 patients: patent ductus arteriosus in 8, atrial septal defects in 7, other VSDs in 5, patent foramen ovale in 5, ruptured sinus of Valsalva in 3 patients. Pulmonary hypertension defined as systolic pulmonary pressure more than 30 mm Hg was present in 16 patients, whereas 11 patients suffered from congestive heart failure.

Surgery
Patients with DCSA VSDs with large shunt (Qp:Qs >2), congestive heart failure or with AR or AV prolapse were offered operation. Forty-five patients underwent VSD closure, of whom 7 had VSD closure and AV repair. All patients were echocardiographically followed up for a mean of 4.9 years (1 to 17 years).

Operative technique
The VSD was closed with a median sternotomy on cardiopulmonary bypass. The VSD was approached through the pulmonary artery in 10 patients, through the aorta in 2, and through the right atrium in 33 patients. The approach to closure was individualized according to the surgeon’s preference, although in later years the pulmonary approach was favored. The VSD was closed with a Dacron patch in 41 patients. In 4 patients, the VSD was found to be small with a firm rim and the surgeon chose to close the defect directly with one to two pledgetted polypropylene sutures. In 1 of these 4 patients late follow-up revealed a residual VSD leak and it is not our practice now to close these defects directly. When there was significant (more than trivial) AR or severe deformity of the AV, the aorta was inspected by way of an aortotomy at the time of operation (10 patients). Valve repair was performed on 7 patients with cusp resuspension, as described by Trusler and colleagues [8] performed in 5 patients, and subcommissural annuloplasty in 2 where pledgetted mattress sutures were placed at the top of the commissures through the aortic wall to reduce cusp prolapse [9]. When the aortic valve was deformed but the aortic regurgitation trivial or less, the VSD was closed without repair except in 1 patient as it was believed that closing the VSD would support the valve and eliminate the Venturi effect.

Associated defects were corrected during operation, such as ligation of patent ductus arteriosus (8 patients), repair of ruptured sinus of Valsalva aneurysm (3 patients), direct closure of arterial septal defects (7 patients), patch closures of other VSDs (5 patients). Mean cardiopulmonary bypass time was 62.8 minutes (39 to 142 minutes) and mean cross-clamp time was 35.1 minutes (12 to 67 minutes).


    Results
 Top
 Abstract
 Introduction
 Material and methods
 Results
 Comment
 References
 
There was no operative mortality. Complications included chest infection in 3 patients (7%) and pericardial effusion in 7 patients (16%), all of which were treated conservatively. After surgical closure of the VSD, postoperative echocardiography revealed satisfactory patch closure in all the patients with no significant patch leak. However, late follow-up show a recurrent VSD in 1 patient who had direct closure of the VSD. Patients were successfully discharged after a mean hospital stay of 10.5 days (4 to 17 days).

Group I patients
In 20 patients where AR was trivial or mild, the general tendency was not to repair the aortic valve as it was believed that closing the VSD would reduce the Venturi effect and prevent progress of AR. The degree of AR remained unchanged in 19 patients on a mean postoperative follow-up of 4.3 years (1 to 7 years). One patient diagnosed with progressive AR is awaiting an AV repair or replacement. In 6 patients with moderate to severe AR, valve repair was deemed necessary. After the repair of the aortic valve, AR improved from moderate AR to mild in 4 of the patients on mean follow-up of 5.8 years (5 to 7 years), but in 2 in whom AR was severe preoperatively, the AR condition remained the same on mean follow-up of 5 years (4 to 6 years).

Group II patients
In 9 patients no valve repair was performed because of mildly prolapsing aortic cusp without AR present. AR did not appear in 4 patients on a mean follow-up of 3.8 years (1 to 7 years). However, it progressed in 5 patients on a mean follow-up of 9.8 years (5 to 17 years). One patient with a moderate prolapse of an aortic cusp preoperatively had surgical repair of his AV although there was no AR. Postoperatively there was no AR on a follow-up of 5 years.

Group III patients
In 9 patients who had a large VSD with mean size of 8.2 mm (4.3 to 11.0 mm) and mean Qp:Qs of 2.9 but without AR preoperatively, closure of the VSD was performed with no valve repair. AR did not progress on a mean follow-up of 3.6 years (1 to 5 years).

Hence 9 of 9 patients in whom VSD was closed before the onset of AV deformity or AR, closure appeared to prevent the progression of AV deformity and AR on a mean follow-up of 3.6 years. However, when closure of VSD was performed after the onset of AV deformity, AR progressed in 5 of 9 patients on mean follow-up of 9.8 years (5 to 17 years).


    Comment
 Top
 Abstract
 Introduction
 Material and methods
 Results
 Comment
 References
 
Doubly committed subarterial VSD occurs more commonly in Asians, with prolapse of an aortic cusp a common feature. Yip and colleagues [10] from Singapore reported an incidence of 9.1% of DCSA VSD in a series of 464 patients diagnosed. In a series of 2,085 VSD closures, Xu and colleagues [11] from China reported an incidence of DCSA VSD of 21%, and Anzai and colleagues [3] from Japan reported incidence of 30%. In our experience, DCSA VSD makes up 32% of all surgically closed VSDs. Where spontaneous closure of other types of VSD is estimated to be between 25% and 50% in patients followed up from birth [10, 1214], spontaneous closure of DCSA VSD is extremely rare and in our experience occurred in 1 of 128 patients.

The optimal timing of operation for DCSA VSD is controversial [11,1517]. As in any type of open heart operation there always will be a small but real risk of mortality. However, this has to be weighted against the benefits of preventing AR [11] and the necessity of valve replacement later on.

When AR is present in patients with DCSA VSD most clinicians will agree that closure of the VSD with or without AV repair should be performed as soon as possible [1, 3, 18]. However, once AR has occurred, despite closure of VSD, AR may progress and ultimately the patient may require an AV repair or replacement. Chauvaud and colleagues [19] reported a need for valve replacement in 16% of his patients, with de Leval and colleagues [4] reporting a need for valve replacement in 14%. Our experience is that once there is an onset of AR surgical closure without valve repair may not always suffice to prevent progressive AR, as 1 patient needed an AVR to correct his progressive AR.

Early closure of VSD has been proposed to prevent the onset of AR. De Leval [4] and Ishikawa [18] and their colleagues believe that closure before the onset of AR should be performed. However, in our experience, surgical closure may not always prevent progressive AR once an AV deformity exists even with no AR. Repair of the prolapse valve should be performed. Various techniques of repair of the aortic valve have been described. Repair of the aortic valve as described by Cabrol and associates [20] and popularized by Trusler and colleagues [8] is the most common procedure performed. Annuloplasty and ridge enhancement have also been described. More recently Chauvaud and colleagues [20] compared two types of repair in two groups of patients. One group had plication of the prolapsed leaflets and folding of the free edges, and the other group had triangular resection of the prolapsed cusp, annuloplasty, and reinforcement of the aortic wall. There was a higher success rate in group 2, with only 4% requiring reoperation, unlike the first group in which a reoperation was required in 43%. Other methods include aortic valvuloplasty as performed by Hitchcock and colleagues [21], with 19% requiring reoperation.

In our experience AR did not occur before the age of 3 years, except in 6 patients in whom it appeared in infancy. Its incidence then increased with age thereafter. Xu and colleagues [11] reported similar findings with AR not appearing before the age of 4 years. Therefore, AR is a progressive disease. It is advisable that surgical closure be performed early in patients with DCSA VSD before the onset of AR or any AV deformity. However, after the onset of AV deformity, surgical closure without valve repair may not suffice to prevent progressive AR, and once there is an onset of AR, valve repair seems to improve and control any progressive AR. Closure of DCSA VSD should, therefore, be performed early, possibly before the onset of AV deformity and AR.


    References
 Top
 Abstract
 Introduction
 Material and methods
 Results
 Comment
 References
 

  1. Griffin M.L., Sullivan I.D., Anderson R.H., Macartney F.J. Doubly committed subarterial ventricular septal defect: new morphological criteria with echocardiographic and angiographic correlation. Br Heart J 1988;59:474-479.[Abstract/Free Full Text]
  2. Rhodes L.A., Keane J.F., Fellows K.E., Jonas R.A., Castaneda A.R., Nadas A.S. Long follow-up (to 43 years) ventricular septal defect with audible aortic regurgitation. Am J Cardiol 1990;66:340-345.[Medline]
  3. Anzai T., Iijima T., Yoshida I., Sakata Y., Obayashi T., Ishikawa S. The natural history and timing of radical surgical operation for subpulmonic ventricular septal defects. Jpn J Surg 1991;21:487-493.[Medline]
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