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Interact CardioVasc Thorac Surg 2009;9:83-87. doi:10.1510/icvts.2008.195180
© 2009 European Association of Cardio-Thoracic Surgery
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Institutional report - Congenital

Outcome after reoperation for atrioventricular septal defect repair{star}

Özcan Birima,*, Menno van Gamerena, Peter L. de Jonga, Maarten Witsenburgb,c, Lennie van Osch-Geversb and Ad J.J.C. Bogersa

a Department of Cardio-Thoracic Surgery, Erasmus MC Rotterdam, Dr. Molewaterplein 40, 3015 GD, Rotterdam, The Netherlands
b Department of Cardiology, Erasmus MC Rotterdam, Dr. Molewaterplein 40, 3015 GD, Rotterdam, The Netherlands
c Department of Pediatric Cardiology, Erasmus MC Rotterdam, Dr. Molewaterplein 40, 3015 GD, Rotterdam, The Netherlands

Received 25 September 2008; received in revised form 19 March 2009; accepted 24 March 2009

{star} Presented at the 22nd Annual Meeting of the European Association for Cardio-thoracic Surgery, Lisbon, Portugal, September 15, 2008.

*Corresponding author. Department of Cardio-Thoracic Surgery, Room BD 575, Erasmus MC, P.O. Box 2040, 3000 CA Rotterdam, The Netherlands. Tel.: +31-10-7035412; fax: +31-10-7033993.

E-mail address: o.birim{at}erasmusmc.nl (Ö. Birim).


   Abstract
Top
 Abstract
1. Introduction
 2. Material and methods
 3. Results
 4. Discussion
 References
 
Results of surgical repair of atrioventricular septal defect (AVSD), both partial (PAVSD) and complete (CAVSD), have improved. However, reoperation is not uncommon. This report describes our experience in 59 patients who underwent reoperation after AVSD repair, between 1977 and 2008. Thirty-one patients had a PAVSD, 28 had a CAVSD. Mean interval between initial repair and reoperation was 10±11 years (PAVSD vs. CAVSD: 13±12 vs. 6±9 years, P=0.063). Reoperations were required for left atrioventricular valve regurgitation (LAVVR) in 53 patients (combined with right atrioventricular valve regurgitation in 10, atrial septal defect (ASD) in 11, ventricular septal defect (VSD) in 7, left ventricular outflow tract (LVOT) obstruction in 1, and aortic valve stenosis in 1), ASD in 3, and LVOT obstruction in 3. Valve repair was performed in 45 patients and replacement in 8. Repair techniques of the left-sided atrioventricular valve (LAVV) included cleft closure in 44 patients, commissuroplasty in 19, and annuloplasty in 1. Freedom from additional reoperation was 85%, and 80% at 5 and 15 years. Hospital mortality was 3%. Overall survival was 91%, and 86% after 5 and 15 years. The most common indication to undergo reoperation is LAVVR. Reoperation is safe and in the majority of cases, a durable repair of the LAVV can still be achieved.

Key Words: Atrioventricular septal defect; Reoperation; Congenital


   1. Introduction
Top
 Abstract
 1. Introduction
2. Material and methods
 3. Results
 4. Discussion
 References
 
Results of primary surgical repair of atrioventricular septal defect (AVSD), both partial (PAVSD) and complete (CAVSD), have improved over the last decades. Despite improved survival, reoperation is not uncommon, mostly due to severe regurgitation of the left-sided atrioventricular valve (LAVV) [1, 2], but residual atrial septal defect (ASD), residual ventricular septal defect (VSD), and left ventricular outflow tract (LVOT) obstruction may play a role as well. Management of left atrioventricular valve regurgitation (LAVVR) after AVSD repair presents challenges for the surgeon. Mechanical LAVV replacement does not provide adequate size increase in growing children and may be followed by problems of thromboembolism, prosthetic valve infection, bleeding, and paravalvular leakage. Therefore, a reparative approach is preferable, despite the risk that repair in a reoperation may require subsequent reoperations.

This report describes our experience with reoperation after primary correction of an AVSD with special emphasis on LAVVR.


   2. Material and methods
Top
 Abstract
 1. Introduction
 2. Material and methods
3. Results
 4. Discussion
 References
 
2.1. Patients

The medical records of 59 consecutive patients who underwent reoperation after primary repair of a PAVSD or CAVSD were reviewed. All reoperations were performed at the Erasmus MC Rotterdam between 1977 and 2008. Our center is a tertiary referral center; therefore, the information on the initial cohort of patients is incomplete. Four (7%) patients had their initial repair at outside institutions. Data were collected from hospital records, operative reports, and intraoperative epicardial or transesophageal echocardiographic studies. Follow-up was completed through July 2008. Median follow-up time was 7.5±6 years. Patient characteristics are outlined in Table 1. Thirty-one patients (53%) had a PAVSD, and 28 (47%) patients had a CAVSD. The atrioventricular valve morphology within patients with a CAVSD consisted of Rastelli type A in 9 (32%) patients, type B in 6 (21%), and type C in 10 (36%) patients, all with a free-floating anterior bridging leaflet [3]. Rastelli type was unknown in 3 (11%) patients. Of the 59 patients, 26 (44%) were men and 33 (56%) women. Sixteen patients (27%) had Down's syndrome, of which 15 (94%) had a CAVSD. Associated congenital cardiac anomalies at initial diagnosis, other than secundum ASD and patent ductus arteriosus, were present in 7 (12%) patients. Four patients had an obstruction of the right ventricular outflow tract, two had an obstruction of the LVOT, and one had a left isomerism with persistent superior vena cava at the left side. Mean age at time of initial repair was 9±15 years (median 3 years; range 1 month–65 years). Mean age at time of initial repair for patients with a PAVSD was 15±18 years (median 8 years; range 4 months–65 years). This was 3±5 years (median 0.5 years; range 1 month–24 years) for patients with a CAVSD (P<0.0001).


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  		Table 1 Baseline characteristics of the study population

 
2.2. Surgical techniques and results

Initial repair was performed using standard techniques specific for the type of defect present. Surgical closure of the ASD in patients with a PAVSD was done with an autologous pericardial patch. Additional repair of the LAVV had been performed in 19 (61%) patients (Table 1). The left-sided cleft had been closed in 18 patients, commissuroplasty had been done in two patients, and annuloplasty in two patients. The surgical reconstruction in patients with a CAVSD consisted of a two-patch technique, as described previously [4]. The VSD had been closed by a Gore-Tex patch. Additional plasty on the LAVV had been performed in 23 (82%) patients. The left-sided cleft had been closed in 21 patients. Commissuroplasty had been done in nine patients, and annuloplasty in one patient. Cleft closure of the LAVV had been performed more often in CAVSD patients (75%) than in PAVSD patients (58%). Additional repair of the right-sided atrioventricular valve (RAVV) had been performed in 10 (36%) patients. The right-sided cleft had been closed in 10 patients, and commissuroplasty was done in two patients. The ASD had been closed by an autologous pericardial patch.

After initial repair, LAVVR was absent or limited in 50 (85%) patients, moderate in 6 (10%), and severe in 3 (5%). Right-sided atrioventricular valve regurgitation (RAVVR) was absent or limited in all patients. Ten (17%) patients had a limited residual VSD, while no patient had a residual ASD.

2.3. Statistical analysis

Discrete variables are displayed as proportions, continuous variables as means±S.D. The {chi}2 (whenever n>5 in all groups) or Fisher exact test was used to analyze the categorical data. Differences between continuous variables were analyzed using the Mann–Whitney test. Differences in survival were compared using the log-rank test. A P-value<0.05 was considered significant. Statistical analyses were performed with SPSS 15.0 for Windows (SPSS, Chicago, IL).


   3. Results
Top
 Abstract
 1. Introduction
 2. Material and methods
 3. Results
4. Discussion
 References
 
Mean age at time of first reoperation was 19±20 years (median 7 years; range 1 month–71 years). Mean age for patients with a PAVSD was 27±21 years (median 26 years; range 4 months–71 years) and for CAVSD 9±14 years (median 3 years; range 1 month–53 years) (P=0.0005). Mean interval between initial repair and reoperation was 10±11 years (median 4 years; range 1 week–37 years). There was an approximately significant difference (P=0.063) in mean interval between initial repair and reoperation between patients with a PAVSD (13±12 years; median 11 years; range 1 week–37 years) and a CAVSD (6±9 years; median 2 years; range 2 days–30 years).

Reoperations were required for LAVVR (all severe) in 53 (90%) patients, residual ASD in 3 (5%), and postoperative LVOT obstruction in 3 (5%) (Table 2). Additional indications requiring correction at reoperation (all in patients with LAVVR) were RAVVR (all severe) in 10 patients, residual ASD in 11, residual VSD in 7, LVOT obstruction in 1, and aortic valve stenosis in 1. Remarkably, in the PAVSD patients, a small residual VSD that had not been noted in the first operation was closed in two patients (Tables 2 and 3).


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  		Table 2 Indications for reoperation

 

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  		Table 3 First reoperations performed in the total study population

 
At reoperation, the LAVV was carefully inspected to assess feasibility of valve repair. Valve re-repair was successfully performed in 45 patients with central leakage of the valve, and mechanical valve replacement (MVR) was necessary in eight patients due to fibrotic valve leaflets in seven and a failed attempt to secondary repair in one (Table 3). All valve replacement patients received a tilting, bileaflet mechanical prosthesis (St Jude Medical). Prosthesis size ranged from 27 to 33 mm. Four of the eight patients receiving a mechanical valve received a LAVV repair at initial operation. Repair techniques of the LAVV included cleft closure in 44 patients. At initial operation, the cleft was left open in 14 of these patients, partly closed in five, and completely closed in 25. Commissuroplasty at re-repair was done in 19 patients, in whom commissuroplasty had been performed at initial operation in six, and annuloplasty occurred in two patients. Mean interval between initial repair and LAVV repair or MVR was 9±10 years (median 2 years; range 1 week–27 years) and 21±11 years (median 25 years; range 5–35 years), respectively (P=0.002). Other reoperations consisted of residual ASD repair in three patients, and enucleation of a LVOT obstruction in three. Additional corrections at reoperation consisted of RAVV repair in 10 patients, residual ASD repair in 11, residual VSD repair in seven, enucleation of a LVOT obstruction in one, and aortic valve replacement in one.

3.1. Additional reoperations

Freedom from an additional reoperation (excluding deaths without additional reoperation) was 93% at 1 year, 85% at 5 years, and 80% at 15 years (Fig. 1). After the first reoperation, 14 reoperations were necessary in 11 (19%) patients. Two patients received a third reoperation and one required a fourth reoperation. Of these 11 patients, five had a CAVSD and six a PAVSD.


Figure 1
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  		Fig. 1. Freedom from additional reoperation (excluding deaths without second reoperation). 95% CI at 15 years: AVSD 0.64–0.90, PAVSD 0.52–0.94, CAVSD 0.55–0.90. AVSD, atrioventricular septal defect; PAVSD, partial atrioventricular septal defect; CAVSD, complete atrioventricular septal defect.

 
Mean interval between first reoperation and second reoperation was 6±8 years (median 3 years; range 1 day–26 years). For the second reoperation, LAVVR was the indication in seven patients, LAVV stenosis due to fibrotic valve leaflets in one, a combined LAVVR and LAVV stenosis due to fibrotic valve leaflets in three, and LVOT obstruction in three. Six patients received a second repair of the LAVV. In five patients, the LAVV morphology was not amenable to a second repair. Of these patients, four received an MVR, and one died during the procedure. Three patients received an enucleation of the LVOT obstruction.

Indication for the third reoperation was LAVVR in both patients. One patient received an MVR one month after second reoperation and in the other patient a third repair of the LAVV was performed 3.5 years after second reoperation. This patient received an MVR five years after third reoperation because of severe LAVVR.

3.2. Survival

During follow-up, 7 (12%) patients died. There were two early deaths (3%) after the first reoperation and five deaths during further follow-up (one died 10 days after the first reoperation during second reoperation because repair of the LAVV was not possible and MVR was not feasible due to a small annulus, the other four died of progressive cardiac failure 1 year, 4 years, 11 years, and 16 years after first reoperation). One early postoperative death occurred in a patient who developed a bacterial sepsis, which was the cause of death 1.5 months after reoperation. The other early postoperative death occurred in a patient who developed progressive cardiac failure and a pneumonia, which were the cause of death four months after reoperation.

Overall survival after reoperation, including early postoperative deaths, was 95% after 1 year, 91% after 5 years, and 86% after 15 years (Fig. 2). One-, five-, and 15-year survival of patients with a PAVSD and CAVSD was 97, 93, and 93, and 93, 89, and 78%, respectively (P=0.50).


Figure 2
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  		Fig. 2. Overall survival (including early deaths). 95% CI at 15 years: AVSD 0.69–0.94, PAVSD 0.74–0.98, CAVSD 0.45–0.93. AVSD, atrioventricular septal defect; PAVSD, partial atrioventricular septal defect; CAVSD, complete atrioventricular septal defect.

 
Event-free survival (including death or additional reoperation) was 90, 78, and 69% after 1, 5, and 15 years, respectively (Fig. 3). No difference was seen between patients with a CAVSD and a PAVSD (P=0.39).


Figure 3
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  		Fig. 3. Event-free survival (events included death or additional reoperation). 95% CI at 15 years: AVSD 0.50–0.82, PAVSD 0.50–0.91, CAVSD 0.33–0.79. AVSD, atrioventricular septal defect; PAVSD, partial atrioventricular septal defect; CAVSD, complete atrioventricular septal defect.

 
At the latest follow-up, in the 38 patients with a durable repair, LAVVR was absent or limited in 31 patients, moderate in six, and severe in one. Patients who had moderate or severe LAVVR were asymptomatic and stable on serial echocardiograms. In all patients RAVVR was absent or limited. No patient had a residual VSD, a residual ASD or LVOT obstruction.

All patients with a durable MVR were free of major bleeding, thromboembolic complications, or other complications.

Three patients, of whom two received an MVR, needed pacemaker implantation because of a complete heart block 4 months, 1 year, and 18 years after reoperation.


   4. Discussion
Top
 Abstract
 1. Introduction
 2. Material and methods
 3. Results
 4. Discussion
References
 
Complete reports of reoperations after initial AVSD repair are rare. Some reports are published only on one indication for reoperation, such as LAVVR [5]. Our report on all reoperations over a 30-year period provides insight into the reoperative events after initial repair of AVSD.

Outcome of surgical management of AVSD has improved with a decrease in postoperative mortality. The currently achieved results are extremely good, even with smaller and sicker patients being routinely operated upon. This is in part due to improved surgical skills, better myocardial protection, increased experience with intraoperative transesophageal echocardiography, and improved understanding of anatomy and physiology.

Trends in the current era include earlier age at repair [6, 7], which has decreased the incidence of pulmonary hypertensive crisis [8, 9]. Despite the decrease in postoperative mortality and decrease in the incidence of pulmonary vascular disease, postoperative morbidity remains significant. Reoperation due to severe LAVVR remains the primary contributor to reoperations. Rhodes and colleagues [10] demonstrated that severity of LAVVR increased by at least one grade over a 3-year period in 41% of patients after AVSD repair. This is possibly related to leaving the left cleft unsutured at initial repair [4, 11], as originally recommended by Carpentier [12]. In the last decades, the left cleft is sutured, which should result in less LAVVR. Other risk factors for postoperative LAVVR that have been described are preoperative LAVVR [13, 14], and severe LAVV anomalies [11].

Successful correction of LAVVR occurred in 90% (53 patients) of our study population. Of these patients, cleft closure at initial operation was performed in 34 (64%) patients (partial closure in six patients, complete in 28). Management of recurrent LAVVR in the pediatric population is especially problematic as the possibility of MVR and its attendant complications must be weighted judiciously. MVR, especially in young children, is accompanied by a high mortality risk, and up to 37% may show the development of complete heart block [15]. The need for lifelong anticoagulation remains a disadvantage with a risk of anticoagulation-related complications. Nevertheless, remarkably often the LAVV can be additionally repaired, 85% in our series, as was earlier described in other reports [7]. MVR was necessary in 15% (8 patients) of the first reoperations and in seven patients receiving subsequent reoperations, with a 14% (2/14 patients) incidence of complete heart block in operative survivors necessating pacemaker implantation and no incidence of anticoagulation-related complications.

The number of second reoperations at 19% was reasonably low. It is gratifying that the early mortality was low (9%, 1/11 patients) and that there was no mortality for subsequent reoperations.

We conclude that a significant number of patients have to undergo reoperation after AVSD repair for a variety of reasons. This article provides a guide in advising parents and patients what to expect with regard to further operations after initial repair. The most common indication for reoperation is LAVVR. Reoperation after initial repair is safe and can be performed with a low operative mortality rate. In the majority of cases, a durable repair of the LAVV can be achieved. Even in cases of recurrent LAVVR, MVR can be deferred until the patient is older, potentially deceasing the requirement for future replacements. It is encouraging that only 19% of our study population required a second reoperation with low mortality rates.


   References
Top
 Abstract
 1. Introduction
 2. Material and methods
 3. Results
 4. Discussion
 References
 

  1. Ten Harkel AD, Cromme-Dijkhuis AH, Heinerman BC, Hop WC, Bogers AJ. Development of left atrioventricular valve regurgitation after correction of atrioventricular septal defect. Ann Thorac Surg 2005;79:607–612.[Abstract/Free Full Text]
  2. Murashita T, Kubota T, Oba J, Aoki T, Matano J, Yasuda K. Left atrioventricular valve regurgitation after repair of incomplete atrioventricular septal defect. Ann Thorac Surg 2004;77:2157–2162.[Abstract/Free Full Text]
  3. Rastelli G, Kirklin JW, Titus JL. Anatomic observations on complete form of persistent common atrioventricular canal with special reference to atrioventricular valves. Mayo Clin Proc 1966;41:296–308.[Medline]
  4. Bogers AJ, Akkersdijk GP, de Jong PL, Henrich AH, Takkenberg JJ, van Domburg RT, Witsenburg M. Results of primary two-patch repair of complete atrioventricular septal defect. Eur J Cardiothorac Surg 2000;18:473–479.[Abstract/Free Full Text]
  5. Malhotra SP, Lacour-Gayet F, Mitchell MB, Clarke DR, Dines ML, Campbell DN. Reoperation for left atrioventricular valve regurgitation after atrioventricular septal defect repair. Ann Thorac Surg 2008;86:147–151; discussion 151–152.[Abstract/Free Full Text]
  6. Lacour-Gayet F, Campbell DN, Mitchell M, Malhotra S, Anderson RH. Surgical repair of atrioventricular septal defect with common atrioventricular valve in early infancy. Cardiol Young 2006;16(Suppl_3):52–58.[CrossRef][Medline]
  7. Reddy VM, McElhinney DB, Brook MM, Parry AJ, Hanley FL. Atrioventricular valve function after single patch repair of complete atrioventricular septal defect in infancy: how early should repair be attempted? J Thorac Cardiovasc Surg 1998;115:1032–1040.[Abstract/Free Full Text]
  8. Bando K, Turrentine MW, Sun K, Sharp TG, Ensing GJ, Miller AP, Kesler KA, Binford RS, Carlos GN, Hurwitz RA, Caldwell RL, Darragh RK, Hubbard J, Cordes TM, Girod DA, King H, Brown JW. Surgical management of complete atrioventricular septal defects. A twenty-year experience. J Thorac Cardiovasc Surg 1995;110:1543–1552; discussion 1552–1554.[Abstract/Free Full Text]
  9. Wetter J, Sinzobahamvya N, Blaschczok C, Brecher AM, Gravinghoff LM, Schmaltz AA, Urban AE. Closure of the zone of apposition at correction of complete atrioventricular septal defect improves outcome. Eur J Cardiothorac Surg 2000;17:146–153.[Abstract/Free Full Text]
  10. Rhodes J, Warner KG, Fulton DR, Romero BA, Schmid CH, Marx GR. Fate of mitral regurgitation following repair of atrioventricular septal defect. Am J Cardiol 1997;80:1194–1197.[CrossRef][Medline]
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  14. Suzuki K, Tatsuno K, Kikuchi T, Mimori S. Predisposing factors of valve regurgitation in complete atrioventricular septal defect. J Am Coll Cardiol 1998;32:1449–1453.[Abstract/Free Full Text]
  15. Moran AM, Daebritz S, Keane JF, Mayer JE. Surgical management of mitral regurgitation after repair of endocardial cushion defects: early and mid-term results. Circulation 2000;102(19 Suppl 3):III160–III165.[Medline]

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