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Ann Thorac Surg 1998;65:754-760
© 1998 The Society of Thoracic Surgeons

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Original Articles: Cardiovascular

Long-Term Results After Repair of Complete Atrioventricular Septal Defects: Analysis of Risk Factors

Department of Cardiovascular Surgery, German Heart Center, Munich, Germany

Dr Günther, Department of Cardiovascular Surgery, German Heart Center Munich, Lazarettstraße 36, 80636 Munich, Germany.

Presented at the Thirty-third Annual Meeting of The Society of Thoracic Surgeons, San Diego, CA, Feb 3–5, 1997.

	Abstract

Top Abstract Introduction Material and Methods Results Comment References

 
Background. We analyzed data from 320 patients to evaluate the impact of different preoperative, operative, and postoperative factors on the outcome after repair of complete atrioventricular septal defect.

Methods. Between October 1974 and December 1995, 320 patients with complete atrioventricular septal defect not associated with major cardiac anomalies were operated on. Two hundred seventy-four patients underwent total repair. Sixty-three patients (23%) were less than 6 months old. One hundred ninety-eight (72.2%) underwent primary repair. Seventy-six patients (27.7%) had a previous palliative operation.

Results. Operative mortality in patients who underwent primary repair decreased from 17.6% (1974 to 1979) to 5.0% (1990 to 1995) despite an increase in the number of patients younger than 6 months. In patients undergoing a two-stage procedure operative mortality was 3.9% (late mortality, 7.9%). Young age (<6 months) was an incremental risk factor (p = 0.008) for operative mortality in the early study period. Coarctation of the aorta (p = 0.02) and severe dysplastic left atrioventricular valve (p = 0.001) were associated with a higher risk for operative mortality. Freedom from reoperation at 10 years was 82.5% ± 3.8%.

Conclusions. In patients with complete atrioventricular septal defect, primary repair is the treatment of choice and can be accomplished with good results. In our experience over a period of more than 20 years, earlier date of operation, young age (<6 months), dysplastic left atrioventricular valve, and coexisting coarctation were incremental risk factors for hospital death. The presence of a previously placed pulmonary artery band did not alter the outcome of repair. The reconstructed atrioventricular valve shows a good and long-lasting performance.

	Introduction

Top Abstract Introduction Material and Methods Results Comment References

 
Many reports document improved surgical results after repair of complete atrioventricular septal defect (CAVSD) [1][2][3][4][5][6][7]. Some authors advocate primary repair at an age of 6 month or earlier [6][7]. Our retrospective analysis of 20 years’ experience examines various preoperative, operative, and postoperative risk factors that may have had an influence on the outcome after repair of CAVSD.

	Material and Methods

Top Abstract Introduction Material and Methods Results Comment References

 
We analyzed data from 320 patients with CAVSD operated on between October 1974 and December 1995 at the German Heart Center, Munich. Patients with major associated cardiovascular anomalies (tetralogy of Fallot, double-outlet right ventricle, transposition of the great arteries, total anomalous pulmonary venous drainage) were excluded. Two hundred seventy-four patients underwent repair of CAVSD: 167 (60.9%) were female and 107 (39.0%) male. One hundred eighty-four patients (67.1%) had Down’s syndrome. According to Rastelli’s classification [8], 67.9% (186 patients) presented type A, 3.6% (10 patients) type B, and 28.5% (78 patients) type C. Age at repair ranged between 1 month and 15.2 years (median, 0.9 years). Weight varied between 2.8 and 46.2 kg (median, 6.3 kg). Of the repairs, 56.6% (155 patients) were performed on infants less than 1 year old and 23% (63 patients) on infants less than 6 months old. Primary repair was our main target. As demonstrated in Fig 1, the number of patients who underwent primary repair at an age of less than 6 months increased constantly. One hundred ninety-eight patients (72.3%) had primary repair. One hundred forty-four (72.7%) of these were less than 1 year old. Seventy-six patients (27.7%) underwent a palliative operation 1 month to 11.9 years (median, 2.9 years) before repair, which included pulmonary artery banding in 51, pulmonary artery banding and patent ductus arteriosus ligation in 13, coarctation repair and pulmonary artery banding in 10, and patent ductus arteriosus ligation alone in 2. Thirty-five patients in this group (46%) had a previous palliative operation in another center. In our institution pulmonary artery banding was performed in patients with high pulmonary vascular resistance (>6 U/m²) not reactive to oxygen, hypoplastic left ventricle (left ventricular enddiastolic volume <50% of predicted normal), or coexisting coarctation. In a group of 46 patients a palliative operation was the only option because of severe pulmonary vascular disease. One hundred ninety-five patients presented additional minor cardiac anomalies (Table 1). Significant extracardial anomalies were observed in 9 patients, including Hirschsprungs’ disease, duodenal atresia/stenosis, congenital tracheal stenosis, and Apert’s syndrome. Thirteen patients presented mitral valve anomalies (double-orifice mitral valve, [n = 3], single left ventricular papillary muscle [n = 4], and dysplastic valve with extreme deficiency of mitral tissue [n = 6]).

View larger version (14K): [in this window] [in a new window]   The height of the columns indicates the percentage of patients older than 6 months (black columns) or younger than 6 months (white columns) undergoing primary repair in four consecutive time intervals, 1974 to 1995.

In 23 patients (8.4%) (1974 to 1978), repair was performed using the one-patch technique described by Rastelli and associates [10]. Since 1978, adapting Carpentiers concept of the "tricommissure valve," we have applied the two-patch technique in 251 patients (91.6%) [11]. The ventricular septal defect was closed with a continuous 4.0 or 5.0 Prolene (Ethicon, Somerville, NJ) suture to affix a semicircular Dacron patch, keeping the suture line more on the right side of the crest of the ventricular septum. Then on the upper edge of the patch the valve leaflets were fixed with interrupted sutures starting at the central meeting point of the anterior and posterior leaflets. Competence of the reconstructed AV valve was tested with an injection of cold saline solution, and the anterior commissure or cleft of the left AV valve was closed using single Prolene stitches. In 12 patients the cleft was left open to prevent left AV valve stenosis. Usually we assess left AV valve diameter with a Hegar dilator, applying the nomograms of Rowlatt and colleagues [12].

A separate Dacron patch was then sutured to the superior aspect of the ventricular patch with the ventricular septal defect suture to close the atrial defect. In cases of sparse tissue in front of the coronary sinus the suture line passed above the AV node, placing the coronary sinus on the left side; otherwise, superficial stitches along the valve ring served for anchoring the atrial patch. Additional surgical procedures included closure of patent ductus arteriosus in 40 patients, debanding in 74 (followed by patch enlargement of the pulmonary artery in 11 patients and pulmonary artery resection with end-to-end anastomosis in 2 patients), ligation of left superior caval vein in 6 patients, resection of subaortic stenosis in 3, and closure of additional muscular ventricular septal defects in 2.

Data Acquisition
Complete patient data were assured by reviewing operative lists, computerized patient data bases, hospital records, and cardiac catheterization and echocardiographic reports. The majority of the patients underwent routine follow-up in our Department of Pediatric Cardiology, including physical examination, electrocardiography, radiography, and echocardiography. In 37 patients follow-up information was obtained by a telephone call to the patient’s family or physician or both.

Statistical Analysis
Univariate analysis was carried out using a t test or contingency tables to determine predictors of early and late mortality. Significant factors were entered into a multivariate logistic regression model to assess the independent impact of potential risk factors on early mortality. Actuarial survival curves were constructed using the Kaplan-Meier method. The log-rank test was used to ascertain differences between groups. A two-tailed p value of less than 0.05 was considered statistically significant.

	Results

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Preoperative Patient Data
Patients with Down’s syndrome showed a higher rate of type B (4.9% versus 1.1%) and C (31% versus 20%) CAVSD compared with patients without Down’s syndrome. This difference, however, was not significant (p = 0.17). There was also no difference in the incidence of minor associated cardiac anomalies in patients with or without Down’s syndrome (61.9% versus 62.9%). Five patients presented with advanced cardiorespiratory instability and required preoperative ventilatory assistance.

Catheterization Data
Preoperative cardiac catheterization data of all patients who underwent CAVSD repair were analyzed. The pulmonary-to-systemic flow ratio ranged between 0.3 and 11.0 (mean, 2.8 ± 2.0). Thirty-six patients had a pulmonary-to-systemic flow ratio of 4 or more. Room air pulmonary-to-systemic resistance ratio ranged between 0.01 and 2.56 (mean, 0.37 ± 0.35). In 60 patients the pulmonary-to-systemic resistance ratio was greater than 0.4. The average peak pulmonary systolic pressure in patients undergoing primary repair was 70 ± 14.53 mm Hg compared with 40.8 ± 20.36 mm Hg in those patients with a previous palliative operation (p = 0.001). Mean pulmonary-to-systemic flow ratio before primary repair was 3.0 ± 1.95 compared with 1.75 ± 1.75 (p = 0.001). Generally patients who had a palliative operation showed a higher pulmonary vascular resistance index and pulmonary-to-systemic resistance ratio values compared with those who underwent repair (pulmonary vascular resistance index, 6.1 ± 3.95 versus 4.5 ± 3.39 U/m²; pulmonary-to-systemic resistance ratio, 0.48 ± 0.38 versus 0.37 ± 0.35).

Mortality Data
Operative mortality of 274 patients who underwent repair was 6.6% (18 patients) There were 34 late deaths (12.4%), mainly caused by progressive cardiac failure and late pulmonary infections. The causes of death are summarized in Table 3.

Incidence of Reoperation
Within an observation period of more than 20 years, 31 patients (11.3%) required 35 reoperations 7 days to 8.0 years (mean, 1.2 ± 2.0 years) after repair. Half of these patients underwent reoperation within 2 months after repair. Freedom from reoperation at 10 and 20 years was 82.5% ± 3.8% and 50% ± 16%, respectively (Fig 2). The indication for reoperation was significant left AV valve incompetence in 88.5% (31/35 reoperations). Refixation and repair of the left AV valve was possible in 87% (27/31 patients); it was not possible in 4 patients who required valve replacement. A mechanical prosthesis (size, 19 to 22 mm) was used in 3 patients; 1 patient received a biological prosthesis. Other indications for reoperation were subaortic stenosis in 4/35 (11.4%), recurrent ventricular septal defect in 13/35 (37.1%), and tricuspid valve incompetence in 5/35 (14.3%). Of the patients having reoperation, 64.5% (20/31) had undergone primary repair and 35.5% (11/31) had a two-stage procedure. Fourteen (45.1%) were less than 1 year old at the time of repair. Eight patients died (operative mortality, 16.1% [5/31]). Analysis of potential risk factors that may be associated with a higher reoperation rate showed no influence of weight less than 5 kg (p = 0.09) or age less than 6 months (p = 0.13). Rastelli type C CAVSD was more prevalent in patients having reoperation (42% versus 25.5%). This difference, however, was not significant (p = 0.29). Operative technique (one-patch versus two-patch technique) also showed no significant influence (p = 0.50). Incision of anterior or posterior bridging leaflets had no significant influence on reoperation rate (p = 0.77). Sixteen patients required a permanent pacemaker system, in 2 because of congenital heart block. Of the remaining 14 patients (5.1%), 10 needed a pacemaker system immediate postoperatively whereas 4 needed one 5.2 ± 3.73 years later.

View larger version (12K): [in this window] [in a new window]   Kaplan-Meier estimate of freedom from reoperation after repair of complete atrioventricular septal defect, with 70% confidence intervals. Freedom from reoperation at 10 and 20 years is 82.5% ± 3.8% and 50% ± 16%, respectively.

View larger version (11K): [in this window] [in a new window]   Kaplan-Meier estimate of survival function and 70% confidence intervals for 274 patients undergoing repair of complete atrioventricular septal defect. The actuarial survival at 20 years is 79% ± 2.8%.

	Comment

Top Abstract Introduction Material and Methods Results Comment References

Concerning the time and mode of operation, primary repair of CAVSD is the treatment of choice today [2][6][7]. Most authors advocate primary repair in infants less than 1 year or even 6 months of age [3][6][7][17][18][19]. Yasui and associates [7] analyzed 40 patients (23 within the first year of life) and reported a 2.5% hospital mortality rate. As in other studies, the number of patients in our study group who underwent repair at an age of less than 1 year constantly increased over the years, with a simultaneous reduction in mortality [1]. Timing of operation is an important factor, to ensure surgical repair can be carried out before development of irreversible pulmonary vascular changes [7][19][20][21]. Pulmonary vascular disease in CAVSD develops during the first year of life; intimal fibrosis of the pulmonary vasculature can be found already at the age of 6 months [20]. Another argument in favor of repair in early infancy is the possible increase in degenerative changes of the AV valve as age increases [7]. Studer and colleagues [5] stated that young age was an incremental risk factor in the early period of their study (1967 to 1976) but disappeared as a risk factor after 1976. Pozzi and associates [3] and Berger and coworkers [22] found no correlation between age and mortality. In our series, age less than 6 months correlated with operative mortality. When different time periods were analyzed, however, young age was of borderline significance (p = 0.05) after 1984. This indicates that young age was an important risk factor in the early study period. Left ventricular hypoplasia, additional AV valve anomalies (eg, double-orifice mitral valve or singular papillary muscle) are also known as risk factors for increased mortality [2][5][21][23]. In contrast to these studies, none of our patients with double-orifice mitral valve or singular left papillary muscle died. In our experience, the presence of a severe dysplastic left AV valve was associated with a higher risk of operative mortality.

Despite a policy toward primary repair in young infants, we have a subgroup of 76 patients who underwent a two-stage procedure, with pulmonary artery banding in average 3.5 years before repair. We consider a hypoplastic left ventricle, elevated pulmonary vascular resistance (>6 U/m² not reactive to oxygen test), severe dysplastic left AV valve, and coexisting coarctation as an indication for a palliative operation. Early experiences with pulmonary artery banding in CAVSD have demonstrated significant hemodynamic improvement. Thus Newfeld and associates [20] reported a reduction in pulmonary artery pressure, diminution of left-to-right-shunting, and prevention of progressive increases in pulmonary vascular resistance. Operative mortality, however, in collected series between 1961 and 1977 averaged 34% [22]. Comparing the operative mortality of our patients who underwent a two-stage or primary repair, we found no significant difference (p = 0.28). This coincides with the data published by Clapp and associates [21]. Tweddell and colleagues [1] report that none of their patients who underwent pulmonary artery banding died. In a subgroup of 46 patients we performed pulmonary artery banding only. Those patients generally came late, presenting advanced pulmonary vascular disease. Operative mortality in this group was 13%, with 11 late deaths (26%). The superiority of palliative operation followed by later repair versus primary repair as surgical therapy for these infants is not proven by this report. Valid arguments can still be made against a staged surgical approach. Survivors of banding still have an increased risk of death in the interval before subsequent repair. In our experience and also other centers, however, debanding does not represent a significant risk factor for correction [2][15][24]. We believe primary repair is the treatment of choice for patients with CAVSDs.

	References

Top Abstract Introduction Material and Methods Results Comment References

 

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