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Eur J Cardiothorac Surg 1998;14:19-26
© 1998 Elsevier Science NL

Surgery for coarctation of the aorta in infants younger than 3 months: end-to-end repair versus subclavian flap angioplasty: is either operation better?1

Adnan Cobanoglu, Ganeshakrishnan K. Thyagarajan, Jeri L. Dobbs

Division of Cardiopulmonary Surgery, Oregon Health Sciences University, 3181 SW Sam Jackson Park Road, Portland, OR 97201-3098, USA

Received 24 June 1997; received in revised form 14 April 1998; accepted 21 April 1998.

Corresponding author. Tel.: +1 503 4947820; fax: +1 503 4947829.


   Abstract
Top
 Abstract
Introduction
Materials and methods
 Results
Discussion
Appendix A. Conference...
 References
 
Objective: Recurrent coarctation is a complication which is seen at a consistent rate following all types of repair for coarctation of the aorta. Particularly disappointing late results are reported in younger infants, under 3 months of age. This retrospective analysis was undertaken to compare the outcomes on late follow-up between subclavian flap angioplasty and resection and end-to-end repair, in this age group. Methods: Over a 12-year period, between 1982 and 1994, 86 infants under 3 months of age underwent surgical repair of coarctation (39 resections and end-to-end repair, and 47 subclavian flap angioplasty procedures). Operative mortality was not significantly different (P=0.6) between resection and end-to-end repair (5.1%) and subclavian flap angioplasty (8.5%). All operative deaths (six patients) were in infants with associated ventricular septal defects. The mean follow-up for all patients was 7.95 years±4.10 (range 0–14.5 years). The 5-year survival for resection and end-to-end repair was 87±5%, compared to 75±7% for subclavian flap angioplasty (P=0.2). Results: Recurrent coarctation occurred in nine patients who needed reoperation. The reoperation-free rates at both 5 and 10 years for resection and end-to-end anastomosis, and subclavian flap repair were 86±6% and 90±5%, respectively. The recurrence in the resection and end-to-end anastomosis group were due to constrictive scarring at the anastomosis, whereas periductal tissue and growth of posterior aortic ridge caused recurrence in the subclavian flap angioplasty group. There were no deaths during reoperation for recurrence. Conclusions: Both procedures are extremely effective for coarctation repair in young infants and run a similar risk of recurrence, which are due to completely different mechanisms. The surgeon's expertise is the major determinant of outcome.

Key Words: Coarctation of the aorta • Neonatal coarctation • Recurrent coarctation


   Introduction
Top
 Abstract
 Introduction
Materials and methods
 Results
Discussion
Appendix A. Conference...
 References
 
The first successful surgical repair of coarctation of the aorta was reported by Crafoord and Nylin [1], and, 50 years later, controversy still exists as to the ideal management of this problem, especially in infants. The high recurrence rates with resection and end-to-end anastomosis ranging from 20% to 86% [2] [3] [4], have prompted the development of patch aortoplasty, a concept introduced by Vosschulte [5] in 1957, and subclavian flap angioplasty (SFA), first introduced by Waldhausen and Nahrwold in 1966 [6].

Considerable debate continues to focus on the issues of the ideal timing of operative treatment, and optimal techniques in different age groups [7]. Operative repair in the first year of life is associated with considerable risk of recoarctation, with neonates carrying the highest risk, irrespective of the operative technique used [8]. Good results have been reported following SFA as well as resection and end-to-end repair [9] [10]. Our earlier experience had shown unacceptably high recurrence rates following SFA compared to resection and end-to-end anastomosis in patients younger than 3 months, at a mean follow-up of 38±4 months [11]. The present study was intended to compare the long-term outcomes between resection and end-to-end anastomosis, and SFA, performed by one surgeon (A.C.) at the same institution, in order to bring uniformity in judgement, approach, and technique to this analysis.


   Materials and methods
Top
 Abstract
 Introduction
Materials and methods
Results
Discussion
Appendix A. Conference...
 References
 
Patients
Between 1982 and 1994, a total of 86 patients, of less than 3 months of age, underwent surgical repair for coarctation of the descending thoracic aorta. There were 48 males and 38 females. The ages of the patients at operation ranged from 1 day to 89 days (mean 24.20±22.10). The weight ranged from 1.7–5.6 kg (mean 3.56±0.76). The age and weight distribution of patients is shown in Table 1. Sixty patients (69.76%) were less than 1 month old. The presenting symptoms of the patients are summarized in Table 2. Sixty-six infants were intubated preoperatively, seventy-two had preoperative inotropic support and fifty-six of the patients had preoperative prostaglandin infusion.


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  		Table 1. Age and weight distribution among patients

 

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  		Table 2. Pre-operative symptoms and findings

 
Measurement of pressure gradients
The gradients were measured either by cardiac catheterization (72 patients), or were echocardiographically derived using modified Bernoulli formula: 4(V22-V12), where V1=flow velocity in the ascending aorta, and V2=flow velocity in the descending aorta. During postoperative follow-up, resting blood pressure, measured by the oscillometric method, in the right arm and right leg were compared. Normal values were derived from percentile charts established by the task force on blood-pressure control in children [12]. A blood-pressure gradient of 20 mm Hg or greater was considered significant. Recoarctation was defined as a systolic blood-pressure gradient of greater than 20 mm Hg between the right arm and right leg with upper-extremity hypertension [13]. A blood-pressure gradient greater than 30 mm Hg with arterial hypertension was considered an indication for reintervention. The preoperative gradients ranged from 0 to 68 mm Hg with a mean gradient of 34.00±18.00 mm Hg.

Associated cardiac defects
Only 18 patients (21%) had isolated coarctation. The remaining 68 patients had one or more associated congenital cardiac lesions, shown in Table 3. The three most commonly-associated lesions were ventricular septal defect (48%), patent ductus arteriosus (48%), and atrial septal defect (16%).


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  		Table 3. Associated cardiac defects

 
Types of surgical procedures and techniques
Forty-seven patients underwent subclavian flap angioplasty and 39 patients underwent resection with end-to-end anastomosis. The distribution of patients by age and type of operation is shown in Table 4. All operations were done through a posterolateral left thoracotomy – through the third intercostal space in patients of less than 1 month of age and through the fourth intercostal space in older patients. The type of procedure to be performed was chosen randomly in individual patients to give the thoracic residents and other trainees, in this major teaching hospital, adequate exposure to both techniques. For resection with primary anastomosis the aorta was mobilized with dissection well up onto the aortic arch and down onto the distal aorta. After resection of all coarctation tissue, anastomosis was carried out with 5–0 or 6–0 non-absorbable sutures, avoiding tension at the suture line. Silk was the suture material in most early cases, whereas polypropylene was used more recently. The anastomotic suture line had continuous polypropylene posteriorly and interrupted polypropylene in the anterior half.


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  		Table 4. Age distribution by type of procedure

 
When subclavian patch angioplasty was used the ductus, distal aorta, area of coarctation, left subclavian artery, and distal arch were dissected. The subclavian artery was tied distally at the thoracic outlet. After vascular clamps were placed, the subclavian artery was divided. An incision through the coarctation segment was carried onto the subclavian artery and distally, well beyond the coarctation zone onto the descending thoracic aorta. It was made sure that the flap tissue was symmetrically positioned across the coarctation segment. The posterior aortic ridge was resected. The subclavian artery patch was sewn on with running 6–0 or 7–0 polypropylene sutures.

No concomitant pulmonary artery banding was performed in patients with VSDs. Pulmonary-artery banding was performed in patients with single-ventricle physiology with pulmonary overcirculation.

Follow-up
All the operative survivors were followed up with a mean follow-up for all patients of 7.95 years±4.10 years (range 0–14.5 years). The follow-up was every 3 months for the first year and then on a yearly basis. The information was obtained from clinic records as well as direct contact with the patient or the local physician. During clinic visits, the symptoms were carefully recorded and the blood pressures were measured as described earlier.

Statistical methods
Statistical analyses were performed using the Statistical Package for the Social Sciences (SPSS) Windows version 6.1. Survival analyses were performed using the actuarial method, and comparisons were tested with the Wilcoxon (Gehan) statistic. Risk factors for operative mortality were tested with Fisher's exact two-tailed test. Independent group t-tests were used to compare clamp-time and age differences by the two operation types. All data are presented as mean±standard deviation, or as percentages. Statistical significance was assigned to differences with a P-value of less than 0.05.


   Results
Top
 Abstract
 Introduction
Materials and methods
 Results
Discussion
Appendix A. Conference...
 References
 
There was no statistically-significant difference in the age, weight distribution and the preoperative gradients between the patients that received subclavian flap angioplasty and those who received resection and end-to-end anastomosis. The mean aortic cross-clamp time for the whole group was 18.43±4.45 min. There was no significant difference between the mean aortic cross-clamp time (P=0.365) between the patients that received subclavian flap angioplasty (19.6±4.6) and those receiving resection and end-to-end anastomosis (16.9±3.7).

Operative mortality
All six operative deaths (6.97%) were in patients with associated ventricular septal defects. The operative mortality was not significantly different (P=0.6) between resection and end-to-end anastomosis (5.1%) and subclavian flap angioplasty (8.5%) patients. Among the risk factors that were analyzed, the presence of a ventricular septal defect was the only significant predictor (P=0.01) of operative mortality (Table 5 and Table 6).


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  		Table 5. Risk factors for operative mortality (six deaths)a

 

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  		Table 6. Associated cardiac defects and operative mortality

 
Survival data
The 80 operative survivors were followed up for a mean of 7.95±4.10 years (range 0–14.5 years). There were 13 late deaths and the overall survival was 79±4% at 5 years and 76±5% at 14 years ( Fig. 1 ). The difference in survival by the type of procedure performed was not statistically significant (P=0.094) between resection and end-to-end repair (87±5% at both 5 and 14 years) and subclavian flap angioplasty (72±7% and 68±7% at 5 and 14 years, respectively) and is shown in Fig. 2 . The age at operation had no adverse effect on long-term survival (P=0.283) as seen in Fig. 3 . The presence of a ventricular septal defect (P=0.001), and a univentricular heart (P=0.016) were the only significant predictors of adverse long-term outcome among the associated cardiac defects analyzed (Table 7). However, as seen in Fig. 4 , most of the mortality is within the first 2 years following coarctation repair. An increasing number of associated cardiac defects was another significant predictor of late mortality (P=0.001), as seen in Fig. 5 .



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  		Fig. 1. Actuarial survival curve for all patients who had surgery for coarctation.

 


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  		Fig. 2. Survival by the type of procedure performed.

 


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  		Fig. 3. Survival by the age at operation.

 

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  		Table 7. Associated cardiac defects and survival

 


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  		Fig. 4. Presence of associated ventricular septal defect at the time of repair and survival.

 


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  		Fig. 5. Number of associated cardiac defects and survival.

 
Reoperation
Recurrent coarctation requiring reoperation occurred in nine patients during follow-up. Five of these were in patients with previous resection and end-to-end repairs and four in patients with previous subclavian flap angioplasty. The mean time to reoperation was 252±157 days (range 106–602 days), since the first repair. All but one patient underwent reoperation within the first year since the initial repair. The reoperation-free rates for subclavian flap angioplasty were 90±5% at both 5 and 10 years, and 86±6% for resection and end-to-end anastomosis at the same points on follow-up ( Fig. 6 ), and this difference was not statistically significant (P=0.4). All those who required reoperation underwent Gortex or Dacron patch aortoplasty for recoarctation. There were no operative deaths during the surgery for reoperation. None of these patients have had recurrence of the coarctation or repeat surgery. Although our policy is to resect the posterior coarctation ridge at the time of subclavian flap surgery, this did not cause late problems with aneurysm, dissection, etc. There were no reoperations related to resection of the posterior ridge.



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  		Fig. 6. Reoperation free survival, following resection and end-to-end anastomosis and subclavian angioplasty techniques.

 
Symptoms on follow-up and growth
There were nine patients (two with previous subclavian flap procedure and seven with previous resection and end-to-end anastomosis) with gradients greater than 20 mm Hg but less than 30 mm Hg who are being followed closely, but have not required reoperation at the time this report was submitted. These gradients have not been associated with upper extremity hypertension, left ventricular hypertrophy by echocardiography and the patients are asymptomatic. The significance of the sole pressure gradients remain to be seen and these patients are followed closely. The most common residual symptom was the presence of a heart murmur in 36 patients. The height and weight growth percentiles were below the 5th percentile in 20% of the patients.


   Discussion
Top
 Abstract
 Introduction
Materials and methods
 Results
Discussion
Appendix A. Conference...
 References
 
The search for the ideal form of therapy for aortic coarctation in neonates continues as we move into the sixth decade since it was first surgically corrected, in 1944 [1]. The first 40 years of clinical management of this problem were essentially surgical. However, the situation has become more complex over the past decade with the introduction of balloon dilatation as a form of management. Despite the advancement of catheter interventions, surgical therapy remains the treatment of choice at many institutions for native aortic coarctations [14]. Primary balloon dilation of coarctation has been successful in relieving stenosis, but recent reports suggest a higher rate of restenosis after balloon dilatation than after operative treatment in neonates [14] and in infants [15]. While it has been shown that early repair positively affects the incidence of late postoperative hypertension [16] [17] [18], there continues to be an ongoing debate about the ideal operative technique to achieve the lowest rate of recoarctation, especially in neonates.

Of the three most commonly-performed procedures, namely subclavian flap angioplasty, resection with end-to-end anastomosis, and patch angioplasty, prosthetic patch angioplasty procedures have been uniformly reported to have the highest rates of recurrence when performed in infancy [8] [19] [20] [21] and also late aneurysm formation [22], leaving the issue to be settled between subclavian flap angioplasty and end-to-end repair. There were six operative deaths (6.9%) seen in this series, of infants of less than 3 months of age, and all these patients had an associated ventricular septal defect. This is in accordance with previous reports in which the operative risk increased with the presence of hemodynamically-significant associated cardiac defects, and also in a large multi-center series of seriously ill neonates the operative mortality was 3%, in which the neonates had undergone resection and end-to-end anastomosis [19] whereas there were no operative deaths in infants with no associated cardiac defects [23]. The currently-reported hospital mortality rate in neonates varies between 8% and 26.6% [24] [25] [26] [27], and the present series compares favorably with these published data. The trend towards improving operative mortality in more recent series [19] [23] probably represents improvements in surgical, anesthetic and postoperative care of these sick neonates. There was no significant difference (P=0.6) in mortality rates for the patients who received subclavian flap angioplasty (8.5%) and those who had resection and end-to-end anastomosis (5.1%), which is similar to that reported [27]. However, we did not find small body size to be a predictor of operative mortality as reported by Wu, and associates [27]. The presence of a ventricular septal defect had an adverse outcome both for operative mortality (P=0.01) and for long-term survival (P=0.001). The presence of associated cardiac anomalies, and their number, contributed significantly to long-term outcome, as has been reported [3] [28] [29] [30] [31] previously. Poor preoperative condition of the patient has been shown to significantly increase the risk of operative death, as shown by others [27] [32] and appropriate preoperative stabilization with use of prostaglandin and inotropic support may help in decreasing operative mortality and certainly does not increase the operative risk as seen in this study.

One of the problematic issues regarding coarctation repair has been residual or recurrent coarctation. Earlier reports had indicated rates ranging from 8% to 44% with resection and end-to-end anastomosis [33] [34] [35], which had led to the popularity of subclavian flap angioplasty which had lower recurrence rates [36] [37] [38]. Several studies have concluded that subclavian flap angioplasty should be the preferred procedure in infants [9] [30] [39]. Earlier reports from this center found the subclavian flap procedure to be advantageous only in infants more than 8 weeks [40], and also that end-to-end repair was distinctly advantageous in infants under 3 months of age [11]. The present study did not demonstrate a significant difference in the rates of recurrence between the two procedures on long-term follow-up. Almost all restenoses were seen in the first year following repair in our series, as well as in other reports [20] [23]. Our series does not show an increased risk of late restenosis, as suggested by Zehr and colleagues [20]. Also, our feeling is that a relatively-undergrown hypoplastic isthmus or transverse arch does not need to be treated with extensive resection or patch plasty and that the hypoplastic segment will have the potential to grow once coarctation is fixed. This approach has not given rise to late transverse arch repairs in this series.

While it has been shown that the risk of restenosis is greater with earlier age of operation [20], the mechanisms involved in recurrence between resection and end-to-end anastomosis and subclavian flap angioplasty are entirely different. Jonas [41] and Sanchez and associates [42] implicated the involution of residual circumferential ductal tissue as the cause of restenosis and this has been agreed upon by others [20]. Inadequate resection with residual ductal tissue and failure of the circumferential suture line to grow have been stated as the causes for recurrence following end-to-end anastomosis [20] [23]. These problems can be improved with the advent of newer suture materials and more meticulous intraoperative techniques [43] [44] leading to improved results. Each procedure, even in experienced hands, has its own unavoidable rate of recurrence. The recurrence rates are similar on long-term follow-up, as seen in this series, and result from totally different mechanisms.

In conclusion, the experience from this series of infants under 3 months of age shows that both subclavian flap angioplasty and end-to-end anastomosis are equally effective procedures in this age group and that they have similar rates of recoarctation during long-term follow-up. The recurrences are due to different mechanisms, and the surgeon's comfort and expertise in performing either of these procedures should dictate the procedure of choice, and are the major determinants of outcome.


   Footnotes
 
Presented in part at the 9th Annual Meeting of the European Association for Cardio-thoracic Surgery, Paris, France, September 24–27, 1995. Back


   Appendix A. Conference discussion
Top
 Abstract
 Introduction
Materials and methods
 Results
Discussion
Appendix A. Conference...
References
 
Dr J. Monro (Southampton, UK): Could you just clarify whether you banded children with VSDs or not?

Dr Cobanoglu: No, we have not banded children with VSDs Our policy has been to either close the VSD at the time of coarctation repair or to perform the coarctation repair first and then wait for a few days, and if there is any difficulty, to wean the patient off the respirator, then to close the VSD. So we have not performed a banding for a single large VSD. A couple of patients with Swiss cheese septa, multiple VSDs, have had PA bands.

Dr Monro: That is an important point to clarify.

Dr S. Conte (Copenhagen, Denmark): Ten years ago you reported in a good article in the Journal of Thoracic and Cardiovascular Surgery about the superiority of end-to-end repair over the subclavian flap highlighting the paramount importance of periductal tissue resection [1]. So, if you continue to believe that periductal tissue is active in the neonatal period, how can the surgeon's expertise influence the growth of unresected periductal tissue? Our extensive experience about coarctation repair in early infancy in Paris [2] strongly supports extended end-to-end repair. Your previous results were largely in favour of end-to-end repair because of the lower recurrency rate. Today, this rate seems be increased at the same level obtained with the subclavian flap procedure. Don't you think that the increased recoarctation rate could, in part, be due to a circular anastomosis of limited size, as you don't adopt any extended incision in your end-to-end technique?

Dr Cobanoglu: Our initial report in the early '80s covered patients going back to 1960, and most of the end-to-end repairs were done during an earlier time period and the subclavian flaps during the late '70s, early '80s, and there were multiple surgeons involved. So though it was from a single institution, the time frames for both patient groups were different. So there may have been some differences during late follow-up because of that. But in this report, in a prospective manner, doing both operations with no patient preselection for one or the other, these are our more recent results in a recent time frame, in the '80s and early '90s.

As far as your question related to recurrence, I agree with you, recurrence in an end-to-end repair is because of the failure of the anastomotic line to grow. But recurrences are seer in both patient groups. No matter what you do, you have recurrences in the end-to-end repair group of patients and also in the subclavian flap group of patients. What I'm saving is that if you have a reasonable volume of patients with coarctation of the aorta and if you feel that you know what you are doing at the time of surgery with resection of the posterior ridge in a subclavian flap procedure and also not constricting the anastomosis at the time of end-to-end repair that both patient groups will end up with a certain rate of recurrence and these recurrences can be dealt with easily, and there isn't much of a difference in between the two patient groups.

Dr J.A. Van Son (Leipzig, Germany): As an ardent proponent of resection and end-to-end anastomosis versus other techniques in the repair of coarctation I think that there are only two secrets to obtain an adequate repair, and those are, first, resection of all macroscopically visible ductal tissue (aided by loop magnification) and, second, extensive mobilization of the entire aortic arch, arch vessels, isthmus, and descending aorta from the level of the innominate artery all the way down to the diaphragm; this may take up to half an hour and is especially essential in neonates and young infants. After this preparatory maneuver it is virtually almost always possible to anastomose the descending aorta to the proximal aortic arch without undue tension; I prefer a 7-0 or 6-0 Maxon suture for the anastomosis. Whenever the aortic arch at the level of the innominate artery is about 5 mm or less in diameter it becomes very difficult to clamp the proximal arch without obstructing the innominate artery; in such case I would strongly suggest to perform a median sternotomy, which also allows concomitant repair of intracardiac anomalies. Following these principles, we thus far have had no mortality or restenosis after coarctation repair in early infancy at the Herzzentrum in Leipzig. Similarly, Dr. Hanley in San Francisco recently reported absence of recurrent stenosis in about 30 patients who underwent repair as neonates. I notice that the recurrent stenosis on the angiogram that you showed was very low, which suggests that the resection of ductal tissue was not adequate. What is your view on this? My second question relates to your strategy regarding repair of coarctation and ventricular septal defect. Most of your deaths were in this category. What is your policy there?

Dr Cobanoglu: Well, I guess there are some programs where the incision in the aorta is carried all the way to the base of the innominate artery, but I would say that you are in the minority. In most centers the standard end-to-end resection technique does not involve slicing the whole arch over to the base of the innominate artery. So this report is really on the more conventional and standard method of doing end-to-end anastomosis after resection.

Dr T. Ebels (Groningen, the Netherlands): Your study was not randomised, and I suspect from one of your slides that there was a large, at least probably a significant difference in the ages of the two groups. It seems to me that you used the subclavian flap angioplasty in younger children more than you did the end-to-end anastomosis. In that light, I think that your conclusion that these operative techniques are equal may not be valid.

Dr Cobanoglu: Well, unfortunately the numbers did not lend themselves to looking at each week of age separately to see whether there was a difference, because the number of patients undergoing coarctation repair under three months of age in most centers are not that great, and then when you start splitting them into weeks, the numbers become quite small. I realise that one of the defects of this study is that it was not a prospective randomised study in the true sense, but since I was the surgeon doing all these operations, there was really no bias towards one surgery or the other. Being at a teaching institution, we train residents in both procedures and we just do one of the two operations. If there is an undue difficulty in doing one versus the other, we would very quickly switch to the other technique based on this experience.

Dr Ebels: But was there a difference in the ages between the groups?

Dr Cobanoglu: In the very first week, again with a small number of patients overall there seemed to be more patients who had subclavian flap repair, but there was no statistical significance as far as outcomes are concerned because the numbers are still too small to lend themselves to significance.

Dr F. Fontan (Bordeaux, France): It is interesting to see that coarctation is still an attractive topic. Mr. Stark will remember that the first paper presented to this Association was a coarctation paper by Richard Hopkins on behalf of Great Ormond Street.


   References
Top
 Abstract
 Introduction
Materials and methods
 Results
Discussion
Appendix A. Conference...
 References
 

  1. Crafoord C., Nylin G. Congenital coarctation of the aorta and its surgical treatment. J Thorac Surg 1945;14:347-361.
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