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Eur J Cardiothorac Surg 2007;31:649-653. doi:10.1016/j.ejcts.2006.12.038
Copyright © 2007, European Association for Cardio-Thoracic Surgery. Published by Elsevier B.V. All rights reserved

Subclavian flap aortoplasty: still a safe, reproducible, and effective treatment for infant coarctation

Christopher J. Barreiro, Trevor A. Ellison, Jason A. Williams, Megan L. Durr, Duke E. Cameron, Luca A. Vricella*

Division of Cardiac Surgery, The Johns Hopkins Medical Institutions, Baltimore, MD, USA

Received 21 August 2006; received in revised form 4 December 2006; accepted 11 December 2006.

* Corresponding author. Address: Division of Cardiac Surgery, The Johns Hopkins Medical Institutions, 600 North Wolfe Street, Blalock 618, Baltimore, MD 21287, USA. Tel.: +1 410 287 1262; fax: +1 410 955 3809. (Email: lvricella{at}jhmi.edu).


   Abstract
Top
 Abstract
1. Introduction
 2. Materials and methods
 3. Results
 4. Discussion
 References
 
Objective: Subclavian flap repair of infant coarctation has been criticized and in many centers abandoned in favor of resection with end-to-end anastomosis. The goal of this study was to examine intermediate and long-term results of infant subclavian flap aortoplasty, which has been the preferred technique at our institution over the last two decades. Methods: Our patient database identified all infants (age < 1 year) who underwent repair of isthmic coarctation via thoracotomy between January 1984 and December 2004. Procedure details and late results were collected by retrospective review of hospital and clinic data. Follow-up was 95.8% complete at a mean of 6.7 years. Results: Between January 1984 and December 2004, 119 infants underwent isolated subclavian flap repair of coarctation. Mean age and weight at operation were 35 ± 52 days (range 1–269 days) and 3.5 ± 1.3 kg (range 0.7–9.3 kg), respectively. Concomitant pulmonary artery banding was performed in 22% (26/119). In-hospital mortality was 4% (5/119) and cumulative late mortality was 6% (7/114) of patients with long-term follow-up. Actuarial survival at 1, 5, and 10 years was 91, 85, and 85%, respectively. Overall re-intervention rate for re-stenosis was 11% (12/114); 10 patients (9%) underwent balloon angioplasty while 3 patients (3%) required operative revision. All re-stenoses occurred in the descending aorta, and all occurred in patients who had undergone neonatal repair. At late follow-up, there were no significant neurologic events (left recurrent laryngeal nerve injury, stellate ganglion dysfunction, or paraplegia), no clinically significant ischemic arm complications, and no flap aneurysms. Conclusions: Subclavian flap aortoplasty remains our procedure of choice for isthmic coarctation, as it is a simple, technically straightforward technique with a low incidence of re-stenosis and serious early and late morbidity. Furthermore, subclavian flap re-stenoses are easily treated with percutaneous intervention and seldom require surgical re-intervention via thoracotomy.

Key Words: Aortic coarctation • Congenital heart disease • Subclavian artery


   1. Introduction
Top
 Abstract
 1. Introduction
2. Materials and methods
 3. Results
 4. Discussion
 References
 
Surgical correction of aortic coarctation can be a challenging undertaking in neonates, either in isolation or in association with complex congenital cardiac anomalies. Successful resection with end-to-end anastomosis (EEA) was first described by Crafoord and Nylin in 1945 [1]. Since that initial report, variations in surgical technique have included subclavian flap aortoplasty (SFA), synthetic patch aortoplasty, interposition grafting and extended EEA as alternative treatment options for some of the more complex anatomical variants of this anomaly [2]. Treatment modalities now include catheter-based techniques, such as balloon angioplasty or insertion of expandable endovascular stents [3,4]. However, surgical repair should still be considered as the preferred initial treatment modality for significant coarctation in infancy, since previous studies have demonstrated a higher recurrence rate with angioplasty as compared to surgery [5]. No consensus has yet been reached as to the best operative treatment option for these patients. Subclavian flap repair of infant coarctation has been criticized because of reportedly higher re-coarctation rates, and in many centers has been abandoned in favor of resection with EEA [6,7]. The goal of this study was to examine the intermediate and long-term results of infant subclavian flap aortoplasty, which has been the preferred operative technique utilized at our institution over the last two decades.


   2. Materials and methods
Top
 Abstract
 1. Introduction
 2. Materials and methods
3. Results
 4. Discussion
 References
 
2.1 Study design
Retrospective review of our cardiac surgery database identified 119 patients who underwent SFA for aortic coarctation between January 1984 and December 2004. This cohort included all patients undergoing isolated isthmic coarctation repair with or without concomitant palliative closed procedures via a left thoracotomy. Patients undergoing intracardiac repair of coexisting anomalies were excluded. Institutional Review Board approval was obtained prior to study inclusion and all patients were less than 1 year of age at the time of surgical intervention. Data collection from hospital and office records included pre-operative patient characteristics, procedure details, post-operative complications, and mortality data. Clinical follow-up was 96% (114/119) complete for a total of 740 patient-years and a mean of 6.7 years.

2.2 Statistical analysis
Statistical analysis was performed with SPSS© 12.0 software (SPSS Inc., Chicago, Illinois, USA). All statistics are expressed as mean + standard deviation unless otherwise reported. Non-parametric comparisons were made using Wilcoxon's signed rank test. The freedom from restenosis and survival figures were created using the Kaplan-Meier method. Statistical significance was defined as a two-tailed p value < 0.05.


   3. Results
Top
 Abstract
 1. Introduction
 2. Materials and methods
 3. Results
4. Discussion
 References
 
3.1 Patient population
The patient cohort consisted of 119 infants (age < 1 year) undergoing subclavian flap aortoplasty, with a mean age of 35 days (range 1–269 days) and mean weight of 3.5 kg (range 0.7–9.3) at operation. Clinical characteristics of the 80 (67%) male and 39 (33%) female patients are summarized in Table 1 . Isolated isthmic aortic coarctation was present in 50 (42%) of our patients. Concomitant cardiovascular malformations were present in 69 patients (58%). The mean preoperative systolic blood pressure gradient across the site of aortic coarctation was 31.0 ± 27.8 mmHg.


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  		Table 1 Patient demographics and concomitant cardiac malformations
 
3.2 Operative data
Mean aortic cross-clamp time was 23 ± 10.6 min. Operative status was defined as urgent or emergent in 92 of patients (77%). Sixty-four percent (77/119) of patients were on a prostaglandin E1 (PGE1) infusion at the time of operation. Another 19% (23/119) of patients were on inotropic support in the preoperative phase (dopamine or dobutamine infusion).

Absorbable suture (polyglyconate [Maxon®, Sherwood-Davis & Geck, St. Louis, MO] or polydioxanone [PDSTM, Ethicon, Somerville, NJ]) was utilized to perform the SFA in 57% of patients (69/119). Thirty-one percent (37/119) of patients underwent repair with non-absorbable polypropylene (ProleneTM, Ethicon, Somerville, NJ) monofilament suture. Medical records did not clearly indicate the type of suture used in the remaining 12% (14/119) of cases.

3.3 Postoperative data
There were no intraoperative deaths. At most recent follow-up, mean systolic blood pressure gradient was 6.1 mmHg (n = 109, range: 0–50 mmHg), as compared to 31.0 mmHg (n = 105; range: 0–152 mmHg) preoperatively (p < 0.05). Postoperative complications were few, with 4 of 114 patients (3%) developing a superficial wound infection requiring antibiotic therapy. One patient required early operative ligation of the thoracic duct for chylothorax. Three patients (3%) described a subjective perception of very mild left limb length discrepancy at follow-up without any functional impairment, and in no case was an acute or chronic ischemic complication observed. No patients were identified (by history or specific questioning at late follow-up) to have had a left recurrent laryngeal/phrenic nerve injury or Horner's syndrome. In addition, no flap aneurysms were identified in any of our patients.

Twelve out of 114 hospital survivors (10.5%) required either surgical or percutaneous intervention for clinically significant anastomotic stenosis at a mean post-operative interval of 8.4 ± 12.4 months. All re-stenoses occurred in the area of the aortic isthmus involving the anastomotic repair site. In addition, all occurred in patients who had undergone repair in the neonatal period (<30 days old), with a mean age at operation of 11 days (range: 2–30 days). Nine of the 12 patients requiring re-intervention for re-stenosis underwent a single revision via catheterization and balloon dilatation. One patient required two subsequent balloon dilatations at 2 and 6 months postoperatively. Only three of 114 patients (3%) required operative revision of their subclavian flap. Two of these patients underwent Gortex patch aortoplasty at 4 and 8 months postoperatively without interim percutaneous dilatations. The third patient underwent balloon dilatation 14 months postoperatively followed by Gortex patch aortoplasty 3 months later. Fig. 1 represents the overall freedom from re-stenosis requiring reintervention, which was 89, 87, and 87% at 1, 5, and 10 years, respectively. In Fig. 2 , patients are separated into those undergoing SFA only versus SFA with late intracardiac repair. Those undergoing SFA alone had a 1, 5, and 10 year freedom from re-stenosis of 89% at all three time points. Similarly, those undergoing SFA with late intracardiac repair had a 1, 5, and 10 year freedom from re-stenosis of 88, 80, and 80%, respectively (p = 0.50).


Figure 1
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  		Fig. 1. Freedom from re-stenosis for the entire cohort.

 

Figure 2
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  		Fig. 2. Freedom from re-stenosis based on the need for late repair of intracardiac defects (SFA: subclavian flap aortoplasty).

 
With regard to the two different suture materials utilized, re-stenosis occurred in 10 of the 68 patients (15%) who underwent repair with absorbable suture, while only 2 of the 37 patients (5%) repaired with non-absorbable suture developed re-stenosis. Although this difference between groups was not statistically significant (p = 0.20), ten of the 12 patients (83%) requiring surgical or percutaneous reintervention for re-stenosis had their SFA performed with absorbable monofilament suture.

3.4 Mortality
The causes of death and associated cardiac anomalies are summarized in Table 2 . In-hospital mortality following coarctation repair was 5/119 (4%). Additional late mortality was 7/114 (6%), for a total cumulative mortality of 10% (12/114). Four of the five in-hospital mortalities occurred in patients with hypoplastic LV, with three patients dying from sepsis. The other two patients died from persistent postoperative hemodynamic instability. Of the seven late mortalities, four patients died following repair of an associated intracardiac malformation. One patient died from bacterial endocarditis, and another from septic shock. The cause of death was unknown in one patient. The flow diagram in Fig. 3 stratifies mortality according to the initial procedure performed (SFA vs SFA + pulmonary artery (PA) band) and need for subsequent repair of intracardiac defects. Although not statistically significant (given the relatively small size of the patient cohort), late repair of intracardiac defects was associated with a higher mortality rate. Using the Kaplan-Meier method, overall patient survival at 1, 5, and 10 years was found to be 91, 85, and 85%, respectively (Fig. 4 ). Patients were separated into those undergoing SFA only versus SFA with late intracardiac repair. Those undergoing SFA only had a 1, 5, and 10 year survival of 95, 92, and 92%, respectively. Conversely, those undergoing SFA with late intracardiac repair had a 1, 5, and 10 year actuarial survival of 81, 71, and 71%, respectively (p < 0.01; Fig. 5 ).


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  		Table 2 Cause of death
 

Figure 3
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  		Fig. 3. Mortality based on initial and subsequent procedures performed (PA: pulmonary artery, SFA: subclavian flap aortoplasty).

 

Figure 4
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  		Fig. 4. Kaplan-Meier survival of the entire cohort.

 

Figure 5
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  		Fig. 5. Kaplan-Meier survival based on need for later repair of intracardiac defects (SFA: subclavian flap aortoplasty).

 

   4. Discussion
Top
 Abstract
 1. Introduction
 2. Materials and methods
 3. Results
 4. Discussion
References
 
The two main surgical techniques currently utilized in neonates and infants with isolated coarctation of the aorta include subclavian flap aortoplasty and resection with end-to-end anastomosis. Subclavian flap aortoplasty has the advantage of using autologous tissue with potential for growth. It also provides a tension-free repair with limited need for extensive dissection. However, ductal tissue is often left in situ with this technique, and has been implicated in re-coarctation. In fact, a recent publication by Burkhart et al. demonstrated that excision of ductal and coarctation tissue is associated with lower aortic arch obstruction rates in patients undergoing the Norwood procedure [8]. Conversely, resection with end-to-end anastomosis has the advantage of removing all ductal tissue at the expense of a circumferential suture line under variable tension [9]. We have been quite satisfied with the SFA results in our neonatal patient population, and are currently using this surgical technique as our procedure of choice.

In a previous study from our institution of 179 children < 1 year of age undergoing coarctation repair between 1962 and 1991, 85 patients underwent SFA while 65 underwent EEA. Although not statistically significant, there was a trend towards a lower recoarctation rate in the SFA group (11%) versus the EEA group (23%, p < 0.1) [10]. In another report by Conte and colleagues on 307 consecutive patients undergoing EEA between 1983 and 1994, actuarial survival of patients with isolated coarctation at 10 years was 98%, and only 60% for those with other major intracardiac defects [11]. Several studies in the literature have also compared freedom from re-stenosis requiring intervention in patients undergoing SFA versus EEA, and have found similar outcomes at 5 years. A freedom from re-stenosis in SFA patients of 87–89% at 5 years versus 86–95% in the EEA patients has been reported [12,13].

Among the specific morbidities reported following SFA, ischemic left arm injury has been regarded as one of the major drawbacks. Although 3% of patients subjectively described a very mild limb length discrepancy at follow-up, all patients reported no functional deficits and sustained no acute or chronic neurologic/vascular sequelae. In addition, none of the patients in our cohort sustained recurrent laryngeal nerve injury, phrenic nerve injury, or paraplegia.

The patients in our cohort experienced an acceptable rate of re-stenosis with 12/114 (11%) requiring a revision at a mean post-operative interval of 8 months. All re-stenoses occurred in the descending aorta, a location favorable for catheter or operative re-intervention. Seventy-five percent of these patients were treated successfully with percutaneous balloon angioplasty. The remaining 25% required operative revision with Gortex patch aortoplasty. These operative revisions were performed in the late 1980s and early 1990s. However, with the improving results of catheter-based interventions over the past decade, these cases would perhaps have undergone revision with less invasive techniques.

While more patients with absorbable suture than non-absorbable suture developed re-stenosis (15% vs 5%), this difference was not statistically significant. Experimental models have demonstrated a higher propensity for re-stenosis of vascular anastomoses when using non-absorbable polypropylene suture on growing vessels [14,15]. However, a paucity of clinical evidence exists to support these experimental findings. Nonetheless, due to the theoretical concerns about anastomotic stricture in growing vessels, we currently prefer to use second generation PDS or Maxon to prevent this specific complication. Less tissue reaction has been observed in comparison to older generation absorbable products, and several recent studies have confirmed the strength and durability of anastomoses using these materials [16–18].

We recognize the limitations inherent to any retrospective review. In addition, our study had follow-up that was 96% complete with 5 patients lost to follow-up. With our mean follow-up of 6.7 years, our morbidity data is also limited to those patients who have reached clinically significant re-coarctation during that time period. We did not present a group of matched cases undergoing EEA, and made comparisons to historical data from the literature. Nevertheless, the actuarial survival and freedom from restenosis in our SFA patient cohort has been shown to be similar to comparable EEA groups in the literature.

Although SFA has been criticized and in many centers abandoned in favor of resection with EEA, we currently maintain SFA as the procedure of choice at our institution for infants undergoing repair of isolated isthmic coarctation. SFA remains a simple, technically straightforward technique with a low incidence of re-stenosis and limited early and late morbidity. Furthermore, subclavian flap re-stenoses are easily treated with catheter-based interventions and rarely require repeat surgical intervention via thoracotomy.


   Acknowledgments
 
This study was supported by the Dana and Albert Broccoli Center for Aortic Diseases, and the Mildred and Carmont Blitz Cardiac Research Fund. Dr Christopher Barreiro is a Hugh R. Sharp Jr Research Fellow, and Dr Jason Williams is an Irene Piccinini Investigator in the Division of Cardiac Surgery. The authors would also like to acknowledge: Raafeh A. Waseem for assistance with data collection, and Nishant D. Patel and Dr Martin Makary for statistical analysis of the data.


   Footnotes
 
\#9734; Presented at the Annual Meeting of the Southern Thoracic Surgical Association, Orlando, Florida, USA, November 10–12, 2005.


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

  1. Crafoord C, Nylin G. Congenital coarctation of the aorta and its surgical treatment. J Thorac Surg 1945;14:347-361.
  2. Waldhausen JA, Nahrwold DL. Repair of coarctation of the aorta with a subclavian flap. J Thorac Cardiovasc Surg 1966;51:532-533.[Medline]
  3. Singer MI, Rowen M, Dorsy TJ. Transluminal aortic balloon angioplasty for coarctation of the aorta in the newborn. Am Heart J 1982;103:131-132.[CrossRef][Medline]
  4. Suarez de Lezo J, Pan M, Romero M, Medina A, Segura J, Pavlovic D, Martinez C, Tejero I, Perez Navero J, Torres F. Balloon-expandable stent repair of severe coarctationof the aorta. Am Heart J 1995;129:1002-1008.[CrossRef][Medline]
  5. Shaddy RE, Boucek MM, Sturtevant JE, Ruttenberg HD, Jaffe RB, Tani LY, Judd VE, Veasy LG, McCough EC, Orsmond GS. Comparison of angioplasty and surgery for unoperated coarctation of the aorta. Circulation 1993;87:793-799.[Abstract/Free Full Text]
  6. Clarkson PM, Nicholson MR, Barratt-Boyes BG, Neutze JM, Whitlock RM. Results after repair of coarctation of the aorta beyond infancy: a 10 to 28 year follow-up with particular reference to late systemic hypertension. Am J Cardiol 1983;51:1481-1488.[CrossRef][Medline]
  7. Presbitero P, Demarie D, Villani M, Perinetto EA, Riva G, Orzan F, Bobbio M, Morea M, Brusca A. Long-term results (15 to 30 years) of surgical repair of aortic coarctation. Br Heart J 1987;57:462-467.[Abstract/Free Full Text]
  8. Burkhart HM, Ashburn DA, Konstantinov IE, De Oliveira NC, Benson L, Williams WG, Van Arsdell GS. Interdigitating arch reconstruction eliminates recurrent arch coarctation after the Norwood procedure. J Thorac Cardiovasc Surg 2005;130:61-65.[Abstract/Free Full Text]
  9. Massey R, Shore DF. Surgery for complex coarctation of the aorta. Intl J Card 2004;97:67-73.[CrossRef]
  10. Zehr KJ, Gillinov AM, Redmond JM, Greene PS, Kan JS, Gardner TJ, Reitz BA, Cameron DE. Repair of coarctation of the aorta in neonates and infants: a thirty year experience. Ann Thorac Surg 1995;59:33-41.[Abstract/Free Full Text]
  11. Conte S, Lacour-Gayet F, Serraf A, Sousa-Uva M, Bruniaux J, Touchot A, Planche C. Surgical management of neonatal coarctation. J Thorac Cardiovasc Surg 1995;109:663-674.[Abstract/Free Full Text]
  12. van Son JA, Daniels O, Vincent JG, van Lier HJ, Lacquet LK. Appraisal of resection and end-to-end anastomosis for repair of coarctation of the aorta in infancy: preference for resection. Ann Thorac Surg 1989;48:496-502.[Abstract]
  13. Trinquet F, Vouhe PR, Vernant F, Touati G, Roux PM, Pome G, Leca F, Neveux JY. Coarctation of the aorta in infants: which operation?. Ann Thorac Surgery 1988;45:186-191.[Abstract]
  14. Soejima Y, Yanaga K, Nishizaki T, Yoshizumi T, Uchiyama H, Sugimachi K. The fate of non-absorbable suture in growing vascular anastamoses in liver transplantation. Hepatogastroenterology 1997;44(13):227-230.[Medline]
  15. Chiu I, Hung C, Chao S, Huang SH, How SW. Growth of the aortic anastomosis in pigs. Comparison of continuous absorbable suture with nonabsorbable suture. J Thorac Cardiovasc Surg 1988;95(1):112-118.[Abstract]
  16. Haluck R, Richenbacher W, Myers J, Miller CA, Abt AB, Waldhausen JA. Results of aortic anastamoses made under tension using polydioxanone suture. Ann Thorac Surg 1990;50(3):392-395.[Abstract]
  17. von Segesser LK, Lachat M, Duewell S, Gianom D, Turina MI. Improved growth with bioabsorbable sutures in both high- and low-pressure zones. Ann Thorac Surg 1996;62:1045-1049.[Abstract/Free Full Text]
  18. Chang S, Weng Z, Yang A, Lai S. Absorbable PDS-II suture and nonabsorbable polypropylene suture in aortic anastamoses in growing piglets. J Formos Med Assoc 1998;97(3):165-169.[Medline]



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