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Eur J Cardiothorac Surg 2000;18:62-67
© 2000 Elsevier Science NL

Outcome following single-stage repair of coarctation with ventricular septal defect

Divisions of Pediatric Cardiothoracic Surgery and Pediatric Cardiology, The Cardiac Center, The Children's Hospital of Philadelphia, 34th Street and Civic Center Blvd., Suite 8527, Philadelphia, PA 19104, USA

Received 7 September 1999; received in revised form 28 February 2000; accepted 29 March 2000.

Corresponding author. Tel.: +1-215-590-2708; fax: +1-215-590-2715
e-mail: gaynor{at}email.chop.edu

	Abstract

Top Abstract 1. Introduction 2. Materials and methods... 3. Results 4. Discussion Appendix A. Conference... References

 
Objective: A recent multi-institutional study suggested improved survival (97%) after staged repair of coarctation with ventricular septal defect (VSD) compared with single-stage repair. The current study was undertaken to determine outcome and need for reintervention following single-stage repair of coarctation and VSD at our institution. Methods: Retrospective review of patients undergoing single-stage repair of coarctation with VSD between October 1, 1994 and August 15, 1999. Results: Single-stage repair of coarctation with VSD was performed in 25 infants (12 males, 13 females) at a median age of 12 days (range 1–87 days) and median weight of 3.3 kg (range 1.3–4.4 kg). The VSD was conoventricular in ten patients, malalignment type with posterior deviation of the infundibular septum in ten, muscular in four and conal septal hypoplasia in one. Arch hypoplasia was present in all patients and bicuspid aortic valve in 13. At least moderate subaortic narrowing was present in six patients (secondary to prolapse of tricuspid valve tissue across the VSD in four). Repairs were performed via a median sternotomy with a mean circulatory arrest time of 38±12 min. Overall patient survival was 96% with one operative death and no late deaths at a median follow-up of 16 months (range 1–50 months). Reinterventions included balloon dilatation of recurrent coarctation (five), closure of residual VSD (one) and Ross–Konno procedure (two). Actuarial freedom from reintervention for the hospital survivors was 81% (95% confidence limit (CL) 61%, 92%) at 6 months, 71% (95% CL 47%, 87%) at 1 year and 59% (95% CL 31%, 82%) at 2 years following surgery. Conclusion: Single-stage repair of coarctation with VSD can be performed with low operative mortality and good midterm survival equivalent to reported results for staged repair.

Key Words: Ventricular septal defect • Coarctation • Cardiac surgery

	1. Introduction

Top Abstract 1. Introduction 2. Materials and methods... 3. Results 4. Discussion Appendix A. Conference... References

 
The optimal management for patients with aortic coarctation and a ventricular septal defect (VSD) remains controversial [1–5]. A variety of therapeutic strategies have been proposed including coarctation repair alone; coarctation repair with pulmonary artery banding and delayed VSD closure; and single-stage repair of both defects. A recent multi-institutional study suggested improved survival with initial coarctation repair in conjunction with pulmonary artery banding and later VSD closure [1]. Single-stage repair, however, has the advantages of correcting all of the anatomic and physiologic abnormalities at a single operation and possibly decreasing the need for subsequent operations. We have adopted an institutional policy of single-stage repair of the coarctation and VSD in infants with moderate or large VSDs. The current study was undertaken to determine the outcome and need for reintervention following single-stage repair of coarctation with VSD at our institution.

	2. Materials and methods

Top Abstract 1. Introduction 2. Materials and methods... 3. Results 4. Discussion Appendix A. Conference... References

 
Review of the Cardiology and Cardiac Surgery Databases at The Children's Hospital of Philadelphia identified 25 patients who underwent single-stage repair of coarctation and VSD between October 1, 1994 and August 15, 1999. Patients with interrupted aortic arch were excluded, as were patients with transposition of the great arteries with VSD and coarctation. A review of each patient's medical record was performed. Follow-up data was obtained from medical records and via correspondence from the patient's cardiologist.

During the study period, 65 additional patients underwent coarctation repair via a left thoracotomy. VSDs which were thought to be restrictive were present in seven of these patients (10.8%). Pulmonary artery banding was not performed in any of these patients and only one of the seven patients has subsequently required closure of the VSD. One additional patient underwent a primary Ross–Konno procedure for coarctation with VSD and severe subaortic stenosis. No patient underwent a Damus–Kaye–Stansel procedure with VSD closure and arch reconstruction.

2.1. Operative technique
All patients underwent single-stage repair via median sternotomy. The aortic cannula was placed in the ascending aorta and either single venous or bicaval cannulation utilized at the surgeon's preference. Deep hypothermic circulatory arrest (DHCA) was utilized for the repair of the aortic arch in all patients and for closure of the VSD in 21 of 25 patients. The aortic arch was reconstructed by either resection of the coarctation with primary anastomosis to the ascending aorta or by homograft patch augmentation of the arch. The VSD was closed either with a dacron patch using a continuous suture technique or by direct suture. Additional VSDs, if present, were closed during the same operation.

2.2. Statistical analysis
All data are presented as either mean±standard deviation or median and range as appropriate. Actuarial survival and actuarial freedom from reintervention with 95% confidence levels were calculated using the Kaplan–Meier method. For the survival analysis, birth was taken as time zero. Freedom from reintervention for recoarctation and reintervention were calculated for the hospital survivors using time of surgery as time zero. Data analysis was performed using the NCSS (Kaysville, UT) statistical software.

	3. Results

Top Abstract 1. Introduction 2. Materials and methods... 3. Results 4. Discussion Appendix A. Conference... References

 
Twenty-five patients underwent single-stage repair of aortic coarctation with VSD (12 males, 13 females) at a median age of 12 days (range 1–87 days) and a median weight of 3.3 kg (range 1.3–4.4 kg). A discrete coarctation was present in all patients. Hypoplasia of the proximal aortic arch (proximal to the left carotid artery) as assessed qualitatively by either preoperative echocardiography or by intraoperative inspection was present in all patients. A bicuspid aortic valve was present in 13 patients and subaortic narrowing in six. In four patients, the subaortic obstruction was secondary to prolapse of tricuspid tissue across the VSD (Fig. 1) . The mitral valve was anatomically abnormal in two patients, including one patient with a non-obstructive double-orifice mitral valve. The VSD was of the malalignment type with posterior deviation of the infundibular septum in ten patients, conoventricular in ten, muscular in four, and conal septal hypoplasia type in one patient. Additional muscular VSDs were present in three patients.

View larger version (129K): [in this window] [in a new window]   Fig. 1. Parasternal long-axis view of the left ventricle. Preoperative echocardiogram demonstrating subaortic obstruction secondary to prolapse of tricuspid valve tissue (arrow) across VSD.

Overall actuarial survival was 96% (95% confidence limit (CL) 60%, 99%) at 3 years following surgical repair (Fig. 2) . Ten patients have been followed for at least 2 years following surgical repair and seven patients for longer than 3 years. Eight reinterventions were performed in seven patients (29%); four underwent balloon dilatation for recurrent arch obstruction; two underwent Ross–Konno procedures for complex left ventricle outflow tract obstruction, and one underwent both balloon dilatation of the arch and closure of a residual VSD. Progressive subaortic obstruction has developed in two additional patients who may require Ross–Konno procedures. There was no residual subaortic narrowing either in the immediate postoperative period or at last follow-up in any of the four patients with subaortic obstruction secondary to tricuspid valve tissue. Both patients with preoperative subaortic narrowing not due to tricuspid valve tissue have developed progressive subaortic obstruction.

View larger version (7K): [in this window] [in a new window]   Fig. 2. Actuarial survival for all patients undergoing single-stage repair of coarctation with VSD (n=25).

View larger version (9K): [in this window] [in a new window]   Fig. 3. Freedom from reintervention for recurrent coarctation for hospital survivors (n=24).

View larger version (9K): [in this window] [in a new window]   Fig. 4. Freedom from all reinterventions for hospital survivors (n=24).

	4. Discussion

Top Abstract 1. Introduction 2. Materials and methods... 3. Results 4. Discussion Appendix A. Conference... References

Advocates of coarctation repair alone note that the VSDs may close spontaneously and thus a second operation can be avoided in many infants. However, a significant number of infants with moderate or large VSDs have persistent congestive heart failure following coarctation repair and many remain ventilator dependent. Closure of the VSD is often necessary as a second operation during the same hospitalization. In addition, certain VSDs (malalignment type with posterior deviation of the infundibular septum and conal septal hypoplasia types) never undergo spontaneous closure, thus patients with these defects would be committed to a second operation. Some centers have advocated coarctation repair in conjunction with pulmonary artery banding to prevent congestive heart failure secondary to the VSD. Such a strategy requires that all infants undergo a second operation for VSD closure and pulmonary artery debanding or debanding alone if the VSD closes spontaneously. Other centers have advocated single-stage repair of both defects via median sternotomy. This strategy has the advantages of correcting all hemodynamic abnormalities at the initial procedure with no obligate need for reoperation. Proximal arch hypoplasia is frequently present and repair via a median sternotomy allows augmentation of the entire aortic arch. However, some studies have suggested that this approach is associated with an increased early mortality.

We have adopted an institutional policy of single-stage repair of both coarctation and VSD in infants with a moderate or large VSD and in patients with proximal arch hypoplasia which mandates repair via a median sternotomy using cardiopulmonary bypass and DHCA. In patients with a restrictive VSD and discrete coarctation, we perform coarctation repair via a left thoracotomy. In patients with proximal arch hypoplasia and a restrictive VSD, the repair is performed via a median sternotomy and the VSD closed at the same operation. Early and midterm survival for single-stage repair has been excellent with only one operative death in a 1600-g neonate and an actuarial survival of 96% at 3 years. Seven patients in our series (29% of survivors) have required reintervention. Five patients have undergone balloon dilatation for recurrent coarctation and one has also required reoperation for a residual VSD. In addition, two patients have developed significant subaortic obstruction and have undergone Ross–Konno procedures. The majority of the reinterventions have been transcatheter treatments and only three patients have required reoperation. However, the patients remain at risk for development of additional levels of left ventricular outflow tract obstruction. It is difficult to assess adequacy the subaortic region in the presence of distal obstruction and an unrepaired VSD. As with patients with interrupted aortic arch; repair of the arch, VSD closure, and ligation of the ductus arteriosis may ‘unmask’ significant subaortic obstruction as the entire cardiac output must traverse the left ventricle outflow tract. In our series, four patients presented initially with significant subaortic obstruction secondary to prolapse of tricuspid valve tissue across the VSD. At the time of repair, the tricuspid valve tissue was removed from the subaortic area in the VSD repair. None of these patients has developed evidence of recurrent subaortic obstruction at early or later follow-up echocardiographic evaluation. In children with posterior deviation of the infundibular septum which could potentially produce subaortic narrowing, our current policy is to close the VSD without resection of infundibular muscle. If subaortic obstruction develops, we perform a Ross–Konno procedure. In children with definite subaortic obstruction, we perform a primary Ross–Konno procedure at the time of arch augmentation.

A recent multi-institutional study evaluated outcome in 326 symptomatic neonates with coarctation with or without an associated VSD [1]. A VSD was present in 157 patients and in 105 patients, the VSD was thought to be moderate or large. Single-stage repair was associated with the lowest non-risk adjusted survival. Survival at 2 years was greatest (97%) in patients who underwent repair of coarctation in conjunction with pulmonary artery banding. Only 19% of the VSDs which were not repaired at the initial operation significantly decreased in size within 24 months. Among the 47 patients with moderate to large VSDs who underwent isolated coarctation repair, five died prior to VSD closure, four underwent pulmonary artery banding with two deaths, and 17 underwent VSD closure with three deaths. Among the 34 patients who underwent coarctation repair and pulmonary artery banding, 13 had undergone VSD closure and/or debanding with no deaths, three had died without any additional procedures, and 18 were awaiting additional surgery.

Brouwer and colleagues reported outcome of 80 infants less than 3 months of age with coarctation and VSD [2]. Sixty-four infants underwent simple coarctation repair with banding of the pulmonary artery in ten. Eventually, 34 of these 64 patients (53.1%) required surgical treatment of the VSD. Sixteen infants underwent a single-stage repair via median sternotomy. Hospital mortality was 4.7% (three of 64) in the multi-stage group and 18.8% (three of 16) for single-stage repair. This difference was not statistically significant. Freedom from recoarctation at 5 years was 91.3% for the multi-stage group compared with 60% in the single-stage group. However, freedom from secondary reoperation for treatment of the VSD in the multi-stage group was only 40.7%.

Conte and colleagues evaluated outcome of 307 consecutive neonates undergoing coarctation repair between 1983 and 1994 [3]. The majority of patients with coarctation and an associated ventricular septal defect underwent isolated coarctation repair (97 of 102). Spontaneous VSD closure was observed in 39% of these patients; however, they do not state how many subsequently required a second operation for VSD closure. Park and colleagues evaluated outcome in 31 infants with coarctation and a VSD following surgical repair [4]. Coarctation repair alone was performed in 23 infants. Nine needed no additional surgical therapy, six required early VSD closure, and eight underwent late closure of the defect. Seven infants underwent coarctation repair and simultaneous pulmonary banding with one early death. One patient underwent VSD closure alone. Additional operations for treatment of the VSD were required in 20 (65%) of the patients. The overall mortality rate at a mean follow-up of 5.7 years was 10.3%.

There are several limitations to the current study. It is a retrospective analysis and the data are limited by information available in the medical record. In particular, the assessment of proximal arch hypoplasia and subaortic obstruction are qualitative rather than quantitative. Also, there is potential for patient selection bias; however, because of our institutional policy, only one patient underwent coarctation repair with secondary VSD closure and only one underwent a primary Ross–Konno procedure with arch reconstruction during the study.

Proponents of staged repair have argued that VSDs in patients with coarctation and VSD frequently close spontaneously. Previous reports, however, suggest that most infants will need early or delayed closure of the VSD following coarctation repair alone with associated mortality and morbidity [1,2,4]. If pulmonary artery banding is performed in conjunction with coarctation repair, reoperation is always necessary. The major disadvantage of single-stage repair has been a reported increased risk of mortality [1,2]. The current study demonstrates that single-stage repair can be accomplished with low mortality equivalent to reported results for staged repair. The incidence of reoperation is also decreased. These patients are at risk for recurrent coarctation after repair; however, with the success of transcatheter techniques for management of recurrent coarctation, these patients can be managed safely and effectively without reoperation. Regardless of the treatment strategy employed, this group of children appears to be at risk for multiple operations, reinterventions and the late development of left ventricular outflow tract obstruction at multiple levels. We believe that primary single-stage repair in the neonate results in a normal series circulation in all patients and reduces the overall long-term risk for morbidity and mortality with potential psychosocial, logistical and financial advantages.

Finally, we wish to point out that since the submission of this paper, one child with complete absence of the thymus and severe immunodeficiency died of sepsis while awaiting thymic transplantation. There was no evidence of recoarctation.

	Acknowledgments

 
Supported in part by grants from the Ethel Brown Foerderer Fund for Excellence and the Daniel M. Tabas Endowed Chair in Cardiothoracic Surgery.

	Footnotes

 
Presented at the 13th Annual Meeting of the European Association for Cardio-thoracic Surgery, Glasgow, Scotland, UK, September 5–8, 1999.

	Appendix A. Conference discussion

Top Abstract 1. Introduction 2. Materials and methods... 3. Results 4. Discussion Appendix A. Conference... References

 
Dr L. Parenzan (Bergamo, Italy): We described in a known surgical journal, ‘Surgery in Italy’, in 1974, the combined treatment of VSD through left ventriculotomy and closure of the VSD through sternotomy. Now, this is historical background, but what about the six cases in which you have been doing direct suture of the VSD?

Dr Gaynor: The group of patients who underwent direct suture of the VSD included patients with severe proximal arch hypoplasia, which was thought not to be amenable to repair through a left thoracotomy. In addition, two patients with left ventricular outflow tract obstruction secondary to prolapse of the tricuspid valve tissue had small VSDs which could be closed just by direct suture. So in these patients there were other indications for arch reconstruction via a median sternotomy, and the VSD was closed during the same procedure.

Dr A. Corno (Lausanne, Switzerland): If now you are always having good results with primary repair, are you still considering a staged approach?

Dr Gaynor: The CHSS studies suffer from the problems of multi-institutional studies, and the results varied between the institutions. There was some question even in the manuscript concerning whether the difference between the survival for single-stage versus staged repair represented a true difference. We had good experience at our institution with single-stage repair, so we continued that approach. We currently do not use the staged approach in any child who is thought to have either proximal arch hypoplasia or moderate to large VSD. We would undertake single-stage repair in all of those children. We think the advantages of the single-stage repair are that it will relieve all of the hemodynamic abnormalities at the first operation. Many children, if you perform isolated coarctation repair, will remain in heart failure and ventilator-dependent with associated morbidity and mortality. If you place a pulmonary artery band, you are committing them to a second operation. We think that overall the morbidity and mortality can be lessened by a single-stage approach.

Mr D. Anderson (London, UK): I noticed that the arch repairs were almost exclusively done using homograft tissue. My own personal observation has been that homograft tissue is rarely ever required in the neonate with severe arch hypoplasia and that the descending aorta will easily come up to the ascending aorta underneath the innominate artery. And finally, you pointed out that six cases had left ventricular outflow tract obstruction due to prolapsing tricuspid valve tissue. I was always told by my cardiologist that this was the ideal substrate for spontaneous closure of the ventricular septal defect. Do you think in retrospect that it is necessary to close the VSD in a child where the tricuspid tissue is tending to come through the ventricular septal defect?

Dr Gaynor: In regards to the last question, the tricuspid valve tissue was not just occluding the VSD, but was actually prolapsing into the left ventricular outflow tract causing subaortic obstruction, so we thought it was necessary to remove that tissue. As I showed on the echocardiogram, this was not just tricuspid valve tissue abutting against and into the VSD but actually completely across into the LV outflow tract, creating significant subaortic obstruction. Interestingly, none of these children had any subaortic narrowing present after the repair, either early or on late follow-up. We are concerned both in interrupted arches and in coarctation with VSD; if we do a primary anastomosis, there may be compression of the left main stem bronchus. By using the homograft patch we can decrease tension on the repair and prevent bronchial compression. Increased tension may also result in increased bleeding with a high possibility of anastomotic stenosis. So for interrupted arch and for coarctation with VSD, we generally tend to use a homograft patch augmentation and avoid pulling the descending aorta up to the ascending aorta.

Dr G. Stellin (Padova, Italy): Dr Haas’ group had two patients who died from severe residual subaortic stenosis. What would be the prospect of a more radical operation in these patients rather than trying to resect the subaortic obstruction, let's say, a Konno–Ross operation?

Dr Gaynor: Just one comment along those lines. As I mentioned, we had one patient with severe subaortic stenosis during this time period who underwent a primary Ross–Konno procedure with arch augmentation. After the end of the study period, another child who underwent a primary repair with VSD closure and arch augmentation could not be separated from bypass because of severe subaortic stenosis and was converted to a Ross–Konno at that point and has done well. It is very difficult in many of these patients to assess the severity of the subaortic stenosis, but in the group that have very small subaortic regions, a primary repair with a Ross–Konno type procedure and arch augmentation may result in a better outcome than other types of repair.

	References

Top Abstract 1. Introduction 2. Materials and methods... 3. Results 4. Discussion Appendix A. Conference... References

 

1. Quaegebeur J.M., Jonas R.A., Weinberg A.D., Blackstone E.H., Kirklin J.W. Outcomes in seriously ill neonates with coarctation of the aorta. J Thorac Cardiovasc Surg 1994;108:841-854.[Abstract/Free Full Text]
2. Brouwer R.M.H.J., Cromme-Dijkhuis A.H., Erasmus M.E., Contant C. Decision making for the surgical management of aortic coarctation associated with ventricular septal defect. J Thorac Cardiovasc Surg 1996;111:168-175.[Abstract/Free Full Text]
3. Conte S., Lacour-Gayet F., Serraf A., Sousa-Uva M., Bruniaux J., Touchot A., Planché C. Surgical management of neonatal coarctation. J Thorac Cardiovasc Surg 1995;109:663-675.[Abstract/Free Full Text]
4. Park J.K., Dell R.B., Ellis K., Gersony W.M. Surgical management of the infant with coarctation of the aorta and ventricular septal defect. J Am Coll Cardiol 1992;20:176-180.[Abstract]
5. DeLeon S.Y., Downey F.X., Baumgartner N.E., Ow E.P., Quinones J.A., Torres L., Ilbawi M.N., Pifarré R. Transternal repair of coarctation and associated cardiac defects. Ann Thorac Surg 1994;58:179-184.[Abstract]

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This Article Abstract Full Text (PDF) Alert me when this article is cited Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to Personal Folders Download to citation manager Author home page(s): William M. DeCampli Thomas L. Spray Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Gaynor, J. W. Articles by Spray, T. L. Search for Related Content PubMed PubMed Citation Articles by Gaynor, J. W. Articles by Spray, T. L.