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Ann Thorac Surg 2005;80:1453-1459
© 2005 The Society of Thoracic Surgeons

Original article: Cardiovascular

Extended Resection and End-to-End Anastomosis for Aortic Coarctation in Infants: Results of a Tailored Surgical Approach

Gail E. Wright, MD a , Cheryl A. Nowak, RN b , Caren S. Goldberg, MD a , Richard G. Ohye, MD b , Edward L. Bove, MD b , Albert P. Rocchini, MD a , *

a Division of Pediatric Cardiology, Department of Pediatrics and Communicable Diseases, University of Michigan Medical School, Ann Arbor, Michigan
b Section of Cardiac Surgery, Department of Surgery, University of Michigan Medical School, Ann Arbor, Michigan

Accepted for publication April 1, 2005.

* Address reprint requests to Dr Rocchini, C. S. Mott Children's Hospital, University of Michigan Medical Center, Womens L1242, Box 0204, 1500 East Medical Center Dr, Ann Arbor, MI 48109-0204 (Email: rocchini{at}med.umich.edu).


   Abstract
Top
 Abstract
Introduction
 Material and Methods
 Results
 Comment
 References
 
BACKGROUND: The surgical repair of aortic coarctation in infants has evolved over time. This study evaluates our current approach utilizing extended end-to-end anastomosis without prosthetic material to enlarge all areas of aortic arch hypoplasia.

METHODS: The Michigan Congenital Heart Center database was reviewed for infants who underwent repair of isolated aortic coarctation from January 1, 1990, to January 1, 2000.

RESULTS: Eighty-three infants underwent surgical repair of isolated coarctation during this decade. Median age at repair was 21 days (range, 2 to 365). Repair was performed through thoracotomy in 72 patients. Because of severe transverse arch hypoplasia, the remaining 11 infants underwent median sternotomy with circulatory arrest. There were 2 deaths: 1 due to pulmonary hypertension in a patient with alveolar capillary dysplasia and 1 late death due to pneumonia in a patient with noncardiac anomalies. Neither patient had residual coarctation. Technique-related complications of bronchial compression, chylothorax, and vocal cord paralysis were noted in 4 patients. Follow-up data were available for 66 patients (80%) with mean follow-up duration of 4.5 years (SD ± 3.1). Reintervention was required in 4 patients (6%). One underwent reoperation after 1 month, and 3 underwent balloon angioplasty within 7 months of initial repair. The remaining 61 patients are asymptomatic, on no antihypertensive medications, and have aortic arch gradients less than 15 mm Hg. One developed subaortic stenosis necessitating resection.

CONCLUSIONS: Tailored surgical repair for aortic coarctation has a low rate (6%) of residual and recurrent coarctation even when performed in infants. Mortality and morbidity are low.


   Introduction
Top
 Abstract
 Introduction
Material and Methods
 Results
 Comment
 References
 
Surgical repair of aortic coarctation has been available since 1944, but the surgical approach has changed over time [1–5]. Although some changes have reflected refinements in technique, in large part they have come in response to high rates of residual or recurrent coarctation or other complications, particularly in infants [6–8]. Overall, there is low early operative mortality with all approaches, with late mortality affected most by the presence of associated intracardiac defects. Over time there has also been a move toward repair in infancy which has decreased the risk of long-term hypertension and its associated cardiovascular risks [9–11]. However, repair in early infancy has been plagued by higher recoarctation rates [12–14]. The incidence of arch hypoplasia is high in both simple and complex coarctation especially in neonates. Repair of coarctation with arch hypoplasia is technically more difficult.

Currently at our institution, the approach to isolated aortic coarctation in the infant is tailored to the patient's aortic arch anatomy. The repair is an extended resection with end-to-end anastomosis using a beveled anastomosis without prosthetic material. If the arch hypoplasia extends proximal to the left carotid artery and would require clamping of the innominate artery, the repair is performed through a median sternotomy. Otherwise, the repair is performed through a lateral thoracotomy. In the 1990s, we moved to this approach in an attempt to better address transverse arch hypoplasia and thereby lower the rates of residual or recurrent coarctation.

The purpose of this study was to evaluate the outcomes of this tailored surgical approach of extended resection and end-to-end anastomosis for isolated coarctation in infants during the past decade. We hypothesized that extended end-to-end repair in infants (1) effectively relieves the coarctation gradient acutely, (2) has a lower rate of residual coarctation, (3) has a lower longterm rate of recoarctation with lower reintervention rates, and (4) additionally relieves transverse arch hypoplasia, with no difference in early or late mortality.


   Material and Methods
Top
 Abstract
 Introduction
 Material and Methods
Results
 Comment
 References
 
Study Methods
Retrospective review of the University of Michigan Congenital Heart Center surgical database identified infants less than 1 year of age who underwent isolated repair of aortic coarctation from January 1, 1990, to January 1, 2000. Chart review was performed to extract demographics, echocardiograms, operative notes, perioperative hemodynamics, and complications. Follow-up data were obtained from referring cardiologists. Follow-up blood pressure gradients, echocardiographic gradients, antihypertensive medications, reinterventions, and other associated morbidity and mortality were recorded. First follow-up visit was defined as the 6-week follow-up visit or the closest visit thereafter.

Institutional waiver of consent for retrospective chart review was granted for patients followed up at the University of Michigan Congenital Heart Center. For patients followed up elsewhere, written informed consent from the patient's parent was obtained before contact with the referring cardiologist. This project was approved by the Institutional Review Board of the University of Michigan on August 23, 2001.

Statistical Analysis
Mortality (early and late), actuarial survival rates, and freedom from reintervention due to recurrent coarctation were analyzed by Kaplan-Meier curves. The SAS statistical software (SAS Institute, Cary, North Carolina) was utilized for basic statistical measures.

Surgical Technique
The coarctation repair is approached through a limited, serratus-sparing, left posterolateral thoracotomy through the third or fourth interspace. Single-lung ventilation is not undertaken; the lung is simply retracted anteriorly. The dissection is begun by incising the pleura overlying the aorta in the region of the coarctation. Care is taken to identify and preserve the vagus and phrenic nerves. The pleural opening is extended up the left subclavian and down to the mid-descending aorta. The aorta is then mobilized from the distal ascending aorta to the mid-descending aorta, as are the bracheocephalic vessels. If the patient's distal circulation is not dependent upon a patent ductus arteriosus, division of the ductus or ligamentum with preservation of the recurrent laryngeal nerve can facilitate the exposure. Collaterals and other branches from the aorta are preserved as is feasible. Once the dissection is complete, a tension-free anastomosis of the proximal descending aorta to the underside of the arch can be easily accomplished.

During anesthesia induction and the dissection of the aorta, the patient is allowed to passively surface cool to approximately 36°C to lessen the risk of postoperative paralysis. A heparin dose of 100 units/kg is given. Control of the proximal aorta is accomplished tangentially below the left carotid artery, thereby maximizing exposure to the underside of the arch and maintaining flow to the artery. The left subclavian can either be controlled in the same clamp or separately. The descending aorta is controlled distal to the coarctation. The entire area from the left subclavian artery to an area of normal proximal descending aorta is resected. The proximal aorta is spatulated along the lesser curve of the arch to a point immediately below the origin of the left carotid artery. The distal aorta is appropriately beveled, and anastomosed to the underside of the arch. The anastomosis is flushed and circulation is gradually restored.

For patients who require repair of more proximal arch hypoplasia, the approach is through a standard median sternotomy. During the time period of the study, deep hypothermic circulatory arrest was utilized. Standard techniques of cardiopulmonary bypass were used, with active cooling to 18°C over at least 20 minutes. During cooling, the ascending aorta, arch, bracheocephalic vessels, and proximal descending aorta are widely mobilized to allow a tension-free reconstruction. After delivering cardioplegia and initiating circulatory arrest, the aortic isthmus and coarctation are resected. The lesser curve of the arch is spatulated proximally to a point of normal caliber aorta. The proximal descending aorta is beveled or spalulated to perform the primary anastomosis. Alternatively, the aorta may be ligated just distal to the left subclavian creating a "type A interrupted aortic arch." The proximal descending aorta can then be anastomosed to the proximal arch/distal ascending aorta. Thus, the hypoplastic portion of the arch is required only to sequentially perfuse the left carotid and subclavian arteries.


   Results
Top
 Abstract
 Introduction
 Material and Methods
 Results
Comment
 References
 
Patient Characteristics
Between 1990 and 2000, 83 patients with isolated coarctation of the aorta underwent repair at less than 1 year of age through extended resection and end-to-end anastomosis (Table 1). The study population included patients with bicuspid aortic valve, patent ductus arteriosus, atrial septal defects, small hemodynamically insignificant ventricular septal defects, and mild Shone's variants (such as a slightly abnormal mitral valve or chordal attachments but no supravalvar ring and no significant mitral stenosis at the time of coarctation repair). Patients were excluded if they had critical aortic stenosis, long segment thoracic coarctation, all features of Shone's complex, a left ventricle of questionable size and were being considered for univentricular repair, or if they underwent simultaneous repair of other significant associated lesions. Exclusion criteria were conservative, because the goal of this study was to evaluate management of isolated aortic coarctation. Specifically, patients with a ventricular septal defect and coarctation in which the ventricular septal defect was hemodynamically significant and required simultaneous closure were excluded. Five patients had named genetic syndromes (2 Turner, 1 Down, 1 CHARGE, 1 Soto), whereas 10 others had associated extracardiac congenital anomalies including tracheoesophageal fistula, single kidney, agenesis of the corpus callosum, cerebral heterotopia, hypospadias, and sacral dimple.


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  		Table 1. Patient Characteristics
 
There were 52 boys and 31 girls. Median age at surgery was 21 days (range, 2 to 365). The patient population was skewed toward neonates with ductal dependent lesions: 50% of patients were 21 days old or less at the time of surgery and 75% were 78 days old or less. Median weight at surgery was 3.7 kg (range, 1.7 to 9.3). Forty-one patients (49%) presented in shock preoperatively. Forty-three patients (52%) were managed with preoperative prostaglandin E1 administration. Of note, the ductus arteriosus did not open in response to prostaglandin administration in 3 neonates, but all 3 were more than 2 weeks old. Pressor support was used preoperatively in 29 patients (35%). Patients were resuscitated and stabilized before being taken to the operating room. Seizures occurred before surgery in 4 patients (5%). One patient had preoperative necrotizing enterocolitis and underwent an exploratory laparotomy.

Operative Data
All patients underwent repair through extended resection and end-to-end anastomosis (Fig 1). Seventy-two patients (87%) underwent thoracotomy whereas 11 patients (13%) underwent median sternotomy. For 1 patient, the operation was started as a thoracotomy but then was converted to a median sternotomy. Median cross-clamp time was 17 minutes (range, 9 to 72). Median circulatory arrest time for the 11 patients who underwent sternotomies was 18 minutes (range, 11 to 30). Circulatory arrest times were less than 20 minutes for 9 of 11 of these patients. Two patients from the thoracotomy group had significant residual coarctation gradients noted in the operating room and consequently were converted intraoperatively to subclavian flap angioplasty repairs.



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  		Fig 1. Extended resection and end-to-end anastomosis. Key technical points: (A) beveled anastomosis, brought up under the transverse arch; (B) extended as far proximally as deemed necessary to achieve relief of stenosis; (C) no prosthetic material used; (D) if the arch hypoplasia extends proximal to the left carotid artery and would require clamping of the innominate artery, then the repair is approached through a median sternotomy and deep hypothermic circulatory arrest is used; otherwise, the repair is performed through a lateral thoracotomy.

 
Perioperative Morbidity and Mortality
Postoperative low cardiac output was present in 9 patients (11%). Two patients (2%) experienced seizures in the postoperative period. One patient (1%), from the sternotomy group, underwent mediastinal exploration for postoperative bleeding. Technique-related complications were noted in 4 patients (5%). Two patients had chylothorax and required thoracic duct ligation. One patient had vocal cord paralysis. These 3 patients had their coarctation repair through lateral thoracotomy. One patient, who was repaired through median sternotomy, had left mainstem bronchial compression necessitating a thoracotomy with further mobilization of the descending aorta. There was 1 perioperative death, and thus 1% early mortality. The death occurred secondary to pulmonary hypertensive crises in a patient diagnosed by autopsy with alveolar capillary dysplasia. That patient had no residual coarctation at a cardiac catheterization performed in the immediate postoperative period. At the time of discharge, 9 patients (11%) had residual coarctation gradients of 15 mm Hg or greater by both physical examination and echocardiogram. All but 1 of these patients were 2 months old or less.

Outcomes
Follow-up data were available for 66 patients (80%) (Fig 2). Mean duration of follow-up was 4.5 years (SD ± 3.1). Sixty-two patients (94%) had no recurrent aortic arch gradient, with a significant gradient defined as 15 mm Hg or greater either by echocardiogram or by systolic blood pressure gradient on physical examination. There was 1 late death from pneumonia in a patient with multiple noncardiac anomalies who had no aortic arch gradient. The remaining 61 patients were asymptomatic and on no antihypertensive medications at their last follow-up visit.



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  		Fig 2. Outcomes of extended resection and end-to-end anastomosis for isolated aortic coarctation in infants less than 1 year of age from 1990 to 2000. (ASX = asymptomatic; coarct = coarctation; f/u = follow-up; HTN = hypertension.)

 
Four patients underwent interventions due to residual or recurrent arch gradients, for an overall reintervention rate of 6% (Fig 3). Of these patients, 3 underwent balloon angioplasty, and 1 underwent reoperation. Six patients underwent cardiac catheterization owing to concern for possible recoarctation based on echocardiography. Of these, 3 had significant peak systolic ejection gradients at catheterization (30, 40, and 72 mm Hg). Balloon angioplasty successfully relieved the gradient in all 3 patients. Each of these patients was under a month old at the time of original repair and was repaired through lateral thoracotomy. Reintervention was performed at 3 months, 5 months, and 7 months respectively. The patient who underwent surgical intervention had a residual coarctation gradient of 35 mm Hg noted by both blood pressure cuff and echocardiogram after her original repair, which was performed at 13 days of age through lateral thoracotomy. One month later she underwent reoperation with repair with subclavian flap angioplasty. This patient subsequently developed recoarctation, with long segment narrowing which was not amenable to balloon angioplasty, and had to undergo another surgical repair 16 months later. Of the 4 patients who needed reintervention, 3 of the 4 had residual coarctation gradients of 15 mm Hg or greater at hospital discharge.



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  		Fig 3. Actuarial freedom from reintervention for recurrent coarctation (Kaplan-Meier) for 66 infants repaired through extended resection and end-to-end anastomosis.

 
Associated Hypertension and Late Valvar Morbidity
Postoperative paradoxical hypertension was noted in 15 patients (18%) and was treated acutely with either esmolol or nitroprusside. When hypertension persisted, captopril or propranolol was used once the patient could take oral medications. Twelve patients (14%) were on antihypertensive medication at hospital discharge, 9 of whom had had paradoxical hypertension immediately postoperatively. Right arm blood pressure was 95th percentile or higher in 28 patients at the 6-week outpatient visit (elevated in 42% of patients with available data for that visit). Antihypertensive medication was discontinued within a year in most patients. No patient in the entire follow-up group was on antihypertensive medication at last follow-up.

Thus far, only 2 patients have undergone subsequent cardiac operations. One patient had subaortic stenosis requiring resection, which was complicated by complete heart block necessitating pacemaker placement. The other underwent atrial septal defect closure. Because of associated noncardiac anomalies, patients within the study group underwent the following noncardiac procedures: open lung biopsy, pyloromyotomy, tracheostomy, and repair of a small anterior congenital diaphragmatic hernia.

Mild mitral stenosis was present at last follow-up in 3 patients. Two patients had a subaortic ridge and mild mitral stenosis. One patient had a subaortic membrane with a 26 mm Hg gradient, and 1 had a subaortic ridge without a gradient. A supravalvar mitral ring with minimal gradient was noted in 1 patient. Although 57 patients had bicuspid aortic valves, only 1 patient had developed aortic valve morbidity by the time of last follow-up, which was mild to moderate aortic stenosis.


   Comment
Top
 Abstract
 Introduction
 Material and Methods
 Results
 Comment
References
 
During the decade of the 1990s, 83 patients with isolated coarctation of the aorta underwent repair at less than 1 year of age through extended resection and end-to-end anastomosis at our institution. Patients with other associated lesions that would require median sternotomy for complete repair, including the combination of ventricular septal defect and coarctation, were excluded, since the intent of the study was to focus solely on isolated aortic coarctation. Seventy-two patients were repaired through a lateral thoracotomy, and 11 patients underwent median sternotomy owing to severe aortic arch hypoplasia. Mortality and morbidity were low, as they are with all techniques in patients with isolated coarctation. There was 1 postoperative death and 1 late death; both were in patients with other medical problems who had no recurrent aortic arch obstruction.

Follow-up data were available for 66 patients (80%; Fig 2). Residual or recurrent coarctation was defined as a gradient of 15 mm Hg or greater, either by systolic blood pressure gradient on physical examination or by echocardiogram. Only 4 patients underwent interventions owing to residual or recurrent arch gradients for an overall reintervention rate of 6% (Figs 2 and 3). Of note, 3 of these 4 patients had residual arch gradients of 15 mm Hg or greater at hospital discharge. All 4 were less than a month old at the time of surgery and were repaired through lateral thoracotomy. None of the patients who were repaired through median sternotomy required reintervention during the study period. These results support our current tailored approach using median sternotomy in patients with severe arch hypoplasia, although the number of patients in the sternotomy group was small. The remaining 61 patients were asymptomatic and on no antihypertensive medications at last follow-up.

The presence of a residual aortic arch gradient of 15 mm Hg or greater at hospital discharge warrants particular attention. In our study, 9 of 83 patients had residual gradients at hospital discharge. Three of the 4 patients who required reintervention came from this group with residual gradients. Conversely, only 1 of 74 patients with no significant gradient at discharge later required reintervention. Although these numbers are too small for valid statistical analyses, a similar finding was noted in another recent study of extended arch aortoplasty in infants [15]. In that report, all 4 patients who required reintervention had some residual gradient at discharge. In both studies, when reintervention was needed it occurred within the first 7 months after initial repair.

For patients with isolated aortic coarctation, the move toward repair in infancy has been driven by the finding that patients repaired at earlier ages had lower risk of long-term hypertension [9–11]. Our findings appear consistent with this goal in that no patient was on antihypertensive medication at last follow-up. However, it remains to be seen whether this will be the case as these children age, since the mean duration of follow-up is just under 5 years. In a study of long-term complications in patients who underwent coarctation surgery at later ages, Toro-Salazar and colleagues [16] noted a correlation between elevations in right arm blood pressure at the 6-week postoperative follow-up visit and later development of hypertension. In our group of infants, 42% of patients with available data had right arm blood pressures at the 95th or higher percentile for age at that visit, although only 14% went home on antihypertensive medications. Determining whether this subset of the patients is at increased risk of hypertension in adulthood will require further long-term investigation.

Our review demonstrates that tailored surgical repair for aortic coarctation using extended resection and end-to-end anastomosis has a low rate of residual and recurrent coarctation even in infants. The 6% recurrence rate compares favorably with outcomes in infants with earlier techniques and approaches the low recurrence rate reported for older children with many approaches. In comparison, at our institution from 1960 to 1985, for infants who underwent coarctation repair at less than a year of age with either subclavian flap angioplasty or end-to-end anastomosis, the risk of reoperation by the fifth postoperative year was 16% and by the tenth postoperative year, it was 30% [12].

There has been continued debate about the preferred technique of surgical repair in infants, particularly with respect to addressing aortic arch hypoplasia. Some institutions have had excellent results with the end-to-side anastomosis with a 4% reintervention rate at 2-year follow-up [17, 18]. Other authors have reported recurrence rates ranging from 2% to 24% with extended end-to-end anastomosis without prosthetic material [5, 15,19–28]. In 1993, Zannini and associates [22] described 32 patients less than 3 months old repaired by this technique with a recurrence rate of 13%. Van Heurn and colleagues [23] reported a 17% recurrence rate at 4 years using extended resection and end-to-end anastomosis in 77 infants. In 1995, Conte and coworkers [24] reported a 9.8% recurrence rate at 5-year follow-up in neonates, including 95 patients with isolated coarctation. Using a variation of an extended end-to-end repair termed "aortic arch advancement," Elgamel and coworkers [26] noted only a 2% reintervention rate in neonates, but that was achieved using a median sternotomy with deep hypothermic circulatory arrest for all patients. Similarly, at our institution, in neonates undergoing single-stage repair of aortic arch obstruction and associated intracardiac defects through median sternotomy, recurrent arch obstruction was noted in 3.8% of patients [27]. Recently, Wood and colleagues [15] and Pearl and colleagues [28] reported recurrence rates of 2% in infants repaired through lateral thoracotomy.

Our results add to the growing body of literature supporting extended resection and end-to-end anastomosis as the optimal surgical approach to isolated aortic coarctation in the infant. This technique allows for wide excision of ductal tissue, and the beveled anastomosis addresses tubular hypoplasia of the arch and diminishes the likelihood of circumferential suture line scarring. The results presented here, with a 6% reintervention rate, support a tailored approach that includes consideration of repair through median sternotomy in selected neonates with severe arch hypoplasia.

Like other contemporary studies, this study is limited by the duration of follow-up available thus far. The mean follow-up time is 4.5 years (SD ± 3.1). Based on the history of surgical repair of aortic coarctation, caution is warranted until this approach has stood the test of time. Although these intermediate term results are very promising, it will be important to compare recurrence rates at 15 years or longer. This study is also limited by incomplete follow-up. In our academic practice, only one third of the surgical patients are local referrals. Despite multiple attempts to reach nonlocal families, 20% of the original infant cohort no longer could be contacted to obtain consent. Consequently, no follow-up information could be used for these patients. A small number of adverse outcomes within this group would significantly alter these results. Because we focused solely on infants with isolated aortic coarctation, the findings of this study may not be generalizable to neonates with associated lesions. This narrow focus was intentionally selected, however, as there is continued debate about the optimal management of even this most straightforward group of patients. Additionally, there continues to be concern about whether there is a neurodevelopmental cost to this tailored approach when a short period of circulatory arrest is used. Further study to evaluate neurodevelopmental outcomes in patients undergoing median sternotomy versus lateral thoracotomy is needed. The recent introduction of regional cerebral perfusion allows a median sternotomy approach without the need for deep hypothermic circulatory arrest, although neurologic outcomes after use of this technique are as yet unknown.

In summary, a tailored surgical approach to aortic coarctation using extended resection and end-to-end anastomosis that includes consideration of repair through median sternotomy in neonates with severe arch hypoplasia has a low rate (6%) of residual and recurrent coarctation, even when performed in infants. Thus far, this low recurrence rate approaches that reported with other techniques in older children.


   References
Top
 Abstract
 Introduction
 Material and Methods
 Results
 Comment
 References
 

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C. L. Backer
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Ann. Thorac. Surg., August 1, 2007; 84(2): 715 - 716.
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H.-G. Lim, W.-H. Kim, W.-S. Jang, C. Lim, J. G. Kwak, C. Lee, S. W. Hwang, and C.-H. Lee
One-stage total repair of aortic arch anomaly using regional perfusion
Eur. J. Cardiothorac. Surg., February 1, 2007; 31(2): 242 - 248.
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