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Eur J Cardiothorac Surg 2007;32:215-219. doi:10.1016/j.ejcts.2007.04.023
Copyright © 2007, European Association for Cardio-Thoracic Surgery. Published by Elsevier B.V. All rights reserved
a Department of Cardiothoracic Surgery, Thoraxcentre, Erasmus MC University Hospital, Rotterdam, The Netherlands
b Department of Cardiology, Thoraxcentre, Erasmus MC University Hospital, Rotterdam, The Netherlands
Received 16 January 2007; received in revised form 13 April 2007; accepted 16 April 2007.
* Corresponding author. Address: Department of Cardiothoracic Surgery, Thoraxcentre, Bd 569, Erasmus MC, P.O. Box 2040, 3000 CA Rotterdam, The Netherlands. Tel.: +31 10 4635411; fax: +31 10 4633993. (Email: g.bolraap{at}erasmusmc.nl).
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Abstract |
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 Top Abstract 1. Introduction2. Methods3. Results4. DiscussionReferences |
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Key Words: Ventricular septal defect • Surgical closure • Adult
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1. Introduction |
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TopAbstract1. Introduction2. Methods3. Results4. DiscussionReferences |
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In patients with a VSD, the clinical course depends on the size of the defect, the localisation of the defect and on concomitant anomalies [1]. In young patients the indication for surgical closure of a VSD is most often left-to-right shunting related symptoms [5]. Surgical closure abolishes the left-to-right shunt, prevents development of pulmonary vascular disease, decreases the risk of endocarditis, may prevent aortic valve regurgitation, improves functional classification and increases long-term survival [6].
If spontaneous closure of a VSD did not occur in childhood or adolescence, the chance that it will close in adult life is small. In adults spontaneous VSD closure is reported to occur in only 10% of the patients [1,7,8]. With advancing age, symptoms are related to secondary effects of a shunt, and persistent defects may predispose to endocarditis, aortic regurgitation and in selected cases to heart failure, arrhythmias and pulmonary hypertension [9]. Very few data exist on indication for VSD closure at adult age, on postoperative course and there are almost no data on long-term follow-up of patients with a VSD operated upon at adult age [5].
Quality of life of patients undergoing surgical closure during childhood is comparable to that of the normal population [3]. For those operated in adulthood this has not been studied. Therefore, we describe the follow-up, including quality of life of patients undergoing surgical closure of a VSD in adulthood.
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2. Methods |
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TopAbstract1. Introduction2. Methods3. Results4. DiscussionReferences |
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Diagnosis and hemodynamic assessment was performed by echocardiography. In the early part of the series, before ultrasound was available for clinical use, diagnosis was confirmed with cardiac catheterisation in 14 patients.
All patients were operated with cardiopulmonary bypass. The mean extracorporeal circulation time was 76 min (range 37–124 min) and the mean aortic cross-clamp time 43 min (range 20–87 min). In seven patients one or more concomitant anomalies were treated. In one patient the aortic valve was replaced by a mechanical valve, in two patients a secundum atrial septal defect (ASD) was primarily closed, in one a persistent arterial duct was ligated and in one patient both a secundum ASD was closed and a persistent arterial duct was ligated. In one patient a stenosis of the left pulmonary artery was treated with patch augmentation with autologous pericardium and in one a discrete subaortic stenosis was enucleated.
A right atrial approach to the VSD was used in 27 patients. An additional right ventriculotomy and an additional aortotomy were performed in two patients, and an additional incision in the pulmonary artery in one patient. An exclusively right ventriculotomy was used in only one patient. To improve the exposition of the VSD the septal leaflet of the tricuspid valve was temporarily detached in two patients [10,11].
Closure of the VSD was performed either with a Gore-Tex® patch (n = 13, 46%), a Dacron® patch (n = 8, 29%), a pericardial patch (n = 5, 18%) or primarily (n = 2, 7%).
One patient went abroad 11 years postoperatively in good clinical health and was included as lost to follow-up from that time on. Two patients, who were in good clinical health, refused to undergo echocardiography and ECG. They agreed in being interviewed by telephone. All 25 remaining patients were seen at our outpatient clinic with a mean follow-up time of 13 years. Patients underwent echocardiography, ECG and quality of life assessment. One cardiologist analysed echocardiographic data. Echocardiographic data were available for 25 patients, and included chamber dimensions, valve regurgitations, pulmonary systolic pressure, and flow through a residual VSD. Quantification of the chamber dimensions was performed according to the ASE guidelines [12]. Regurgitant severity of the aortic, pulmonary, and tricuspid valve was graded as none/trivial to severe (0 to 4+) on the basis of colour flow imaging [13]. Pulmonary systolic pressures were calculated on the basis of tricuspid regurgitant jet velocities and estimated right atrial pressures using inferior vena caval dimension [12,13]. A pulmonary systolic pressure >35 mmHg was considered pulmonary hypertension.
The electro-cardio graphic data were also analysed by one cardiologist.
Health related quality of life was determined with the TAAQOL-questionnaire [14]. It consists of 45 questions divided into 12 domains. Each domain contains two to four questions (the actual number per domain is given in parentheses): gross motor functioning (4), fine motor functioning (4), pain (4), sleeping (4), cognitive functioning (4), social functioning (4), daily activities (4), sexual activity (2), vitality (4), happiness (4), depressive moods (4), and aggressiveness (3). Scores of each subscale are normalised to a scale ranging from 0 to 100, with higher scores indicating better quality of life. For each item, the frequency of occurrence of a health status problem is assessed. If such a problem is reported, the emotional reaction to this problem is also determined [14,15]. TAAQOL data of the Dutch general population are available [14].
Categorical variables, summarised in terms of relative frequencies, were analysed using the chi-square test with the Yates correction or using Fisher's exact test. Continuous variables, described as the mean and range, were analysed using Student's t-test. P-values <0.05 were considered significant. The participants’ health related quality of life (as measured by the TAAQOL) was compared with that of a random sample of the general Dutch population in 4410 persons using analysis of variance with adjustment for age and sex. After comparison, the data were summarised as mean values with standard error.
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3. Results |
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TopAbstract1. Introduction2. Methods3. Results4. DiscussionReferences |
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Seven of the 12 patients with a substantial left-to-right shunt presented relatively late and were in NYHA class II. Of these, two patients (2/7) came from developing countries and in two others the parents of the patients had declined diagnostic procedures at paediatric age. The remaining three patients came from referring centres and were presented late; one patient with pulmonary hypertension, one had Down's syndrome and the third was not operated upon in the past because of presumed high operative risk.
The indication for surgical closure was endocarditis in eight patients (29%). In none of these patients the VSD was haemodynamicilly important. In four patients it concerned an endocarditis lenta and in four the VSD was closed after successful medical treatment of a second episode of endocarditis. Identified organisms were Streptococcus Viridans (2), Streptococcus Mutans (1), Enterococcus Faecalis (1), Streptococcus species with undetermined isotype (3) and Staphylococcus Aureus (1). Endocarditis was more prevalent in males than in females (7 vs 1).
Progressive aortic valve regurgitation was the indication for closing a perimembraneous ventricular septal defect in eight other patients (29%). In one patient the aortic valve was replaced, in two patients a fenestration in the right coronary cusp was closed, and in five patients a mild aortic regurgitation was left untreated.
At preoperative presentation 17 patients (61%) were in NYHA class I and II (39%) in NYHA class II. It seems that in the later years of the study, more patients were in NYHA class II (see Fig. 1 ).
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Early postoperative complications were mediastinal exploration for persistent blood loss in three patients and adult respiratory distress syndrome in one patient.
Follow-up was complete in 27 patients with a mean duration of 13 years (range 1–23 years). No patient died. One patient was reoperated because of a residual VSD 2 years after the initial procedure, in which the indication was aortic regurgitation. No recurrent episodes of endocarditis were reported.
At last follow-up, 26 of the 27 patients were in NYHA class I. One patient, preoperatively in NYHA class II with pulmonary hypertension, was in NYHA class III still suffering from pulmonary hypertension. In addition she was in atrial fibrillation.
Echocardiographic findings at follow-up are listed in Table 1 . Tricuspid regurgitation grade 1+ was present in 8 (32%) patients and grade 2+ in 4 (16%) patients, including the patient with pulmonary hypertension. No tricuspid regurgitation was present in the two patients with temporary tricuspid valve detachment. The aortic valve was normal in 15 (60%) patients, grade 1+ aortic regurgitation was present in 9 (36%) and grade 2+ in one (4%) patient. Six of the eight patients in whom the indication for closure of the VSD was aortic regurgitation had grade 1+ aortic insufficiency at follow-up, the other two patients had no aortic insufficiency. Left ventricular dilatation at the end of follow-up, as defined as left ventricular enddiastolic diameter >55 mm, was present in 7 (28%) patients, the indication for closure of the VSD was a left-to right shunt in three patients, endocarditis in three patients and aortic regurgitation in one patient. It seems that more patients with an enlarged left ventricle at the end of follow-up were operated in earlier years (see Fig. 2 ).
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In seven patients (26%) the ECG showed a right bundle branch block. One patient with a right bundle branch block was also in atrial fibrillation. None of the patients had signs of sinus node dysfunction, none had a complete A-V block.
Fig. 3 gives the average TAAQOL scores of the patients compared to the general population sample. The patients had significantly lower health related quality of life in cognitive functioning (score 73.8 ± 5.9 vs 82.7 ± 0.3 in controls, P < 0.045) and sleep (60.0 ± 6.0 vs 73.9 ± 0.4 in controls P < 0.006).
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4. Discussion |
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TopAbstract1. Introduction2. Methods3. Results4. DiscussionReferences |
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More important is the finding that a substantial portion of patients operated upon at adult age did not show a normalisation of intra-cardiac dimensions after VSD closure, which is hardly ever seen after surgical closure of a VSD in childhood. The long-term follow-up after surgical closure of a VSD in infancy is documented extensively [2–4] in contrast to surgically VSD closure at adult age. This suggests that these patients probably would have benefited from an earlier operation.
In addition to closure of the VSD, one patient had aortic valve replacement 18 years after initial indication for closure at the age of 12 years, when the parents refused the operation. In none of the remaining seven patients, for whom indication for surgical closure was a subaortic, perimembranous localisation of the VSD in combination with development of aortic regurgitation, aortic regurgitation progressed to a degree beyond mild. In the natural history of these defects development of moderate or even severe aortic regurgitation is reported to be frequent [7]. These results show once more that surgery without mortality may be a preferable option for these patients.
The prevalence of arrhythmia in this study – less than 10% of the study population – is in line with that of other reports [7]. Theoretically, atrial fibrillation can be expected in those patients with long-standing left atrial dilatation due to substantial left-to-right shunting. Arrhythmias, ascribed to intracardiac surgery performed through a right atrial incision, due to a ‘macro’ re-entry tachycardia around the surgical scar, presenting as atrial flutter are rare. This might indicate that the theoretically pro-arrhythmogenic surgical right atrial scar does not lead to clinically significant arrhythmias up to 24 years after surgery. It is evident that longer follow-up is necessary in this matter.
Quality of life of this operated adult VSD group is comparable with general population. In 10 out of twelve domains they had an equal score. In two domains, cognitive functioning and sleep, our population differed from the general population with regard to the quality of life. This can be interpreted as the quality of life of our patient group being practically equal to the normal population.
Looking at the very good results in terms of mortality and complication rate of surgical closure of VSDs at adult age, realizing that patients operated upon because of left-to-right shunting might had benefited from earlier surgery and knowing the cumulative risk of endocarditis in this population of over 10% [7] with a mortality rate of 15–25% [7,16,17] even in the modern era of sophisticated antibiotic therapy, one might consider closure of all VSDs that have not closed spontaneously in the first two decades of life.
Apart from these considerations, the results of this study may become part of a benchmark for the emerging experience of device closure of a VSD [18].
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References |
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TopAbstract1. Introduction2. Methods3. Results4. DiscussionReferences |
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  L. M. Putman, M. van Gameren, F. J. Meijboom, P. L. de Jong, J. W. Roos-Hesselink, M. Witsenburg, J. J.M. Takkenberg, and A. J.J.C. Bogers Seventeen years of adult congenital heart surgery: a single centre experience Eur. J. Cardiothorac. Surg., July 1, 2009; 36(1): 96 - 104. [Abstract] [Full Text] [PDF]
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 A. Haussler and R. Pretre Surgical closure of a perimembranous ventricular septum defect with a running suture MMCTS, May 23, 2008; 2008(0523): 2410. [Abstract] [Full Text] [PDF]
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