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Ann Thorac Surg 2001;72:1898-1901
© 2001 The Society of Thoracic Surgeons

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Original article: general thoracic

Aortopexy in severe tracheal instability: short-term and long-term outcome in 29 infants and children

^a Department of Thoracic and Cardiovascular Surgery, University Hospital RWTH Aachen, Aachen, Germany
^b Department of Pediatric Cardiology, University Hospital RWTH Aachen, Aachen, Germany
^c Department of Anesthesiology and Operative Intensive Care, Children’s Hospital, Cologne, Germany

Accepted for publication August 14, 2001.

* Address reprint requests to Dr Vazquez-Jimenez, Department of Thoracic and Cardiovascular Surgery, University Hospital, Pauwelsstr 30, D-52057 Aachen, Germany
e-mail: jvazquez-jimenez{at}post.klinikum.rwth-aachen.de

	Abstract

Top Abstract Introduction Material and methods Results Comment References

 
Background. Tracheal instability is a hazardous situation after operation for esophageal atresia. In cases with life-threatening apneas, aortopexy is a therapeutic option. To assess efficacy, short-term and long-term outcome was analyzed retrospectively.

Methods. Between 1985 and 2000, 29 patients (age, 1.5 months to 5.2 years) were operated on. A flaccid trachea after operation for esophageal atresia was the cause for life-threatening apneas in 27, and there was external vascular compression in 2 patients. The operative procedure consisted of ventropexy of the aortic arch to the sternum and ventral thoracic wall.

Results. There was neither early nor late mortality. A reversible lesion of the phrenic nerve was observed in 2 patients, a pneumothorax in 3, and secondary wound healing in 1. In all but 1 patient symptoms improved markedly or disappeared within days or within the first 3 months postoperatively. An increased susceptibility to respiratory infections was observed in long-term follow-up.

Conclusions. Aortopexy can be performed with no mortality and low morbidity. Aortopexy is effective to prevent further life-threatening apneas, but does not prevent an increased susceptibility to respiratory infections.

	Introduction

Top Abstract Introduction Material and methods Results Comment References

 
Instability of the trachea is a hazardous situation after operation for esophageal atresia [1, 2]. Furthermore, this condition may be caused by compression owing to abnormal position of the great vessels [3]. In relation to the intensity of tracheal instability, respiratory symptoms occur. In cases with severe tracheal instability leading to life-threatening apneas, aortopexy has been shown to be a therapeutic option [1, 2]. To assess the efficacy of this therapy, short-term and long-term outcome of our patients was analyzed retrospectively.

	Material and methods

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Patients
Between May 1985 and December 2000, 29 patients (22 male), 24 of whom were younger than 1 year of age and 21 younger than 6 months (mean age, 9.3 ± 14.8 months, range, 1.5 months to 5.2 years), were operated on for severe tracheal instability. A flaccid trachea after operation for esophageal atresia with congenital tracheoesophageal fistula at the level of the atresia was the main cause for life-threatening apneas in 27 cases; compression by the innominate artery or the distal aortic arch was the cause in 1 patient each. Diagnosis was verified by tracheal endoscopy in all patients. Anatomic structures were studied by computed tomographic scan in 5 patients and magnetic resonance imaging in 2 patients; angiography was performed in 14 patients. Beginning in the early 1990s, cardiac structures and great vessels were examined by echocardiography, thus avoiding angiography in most of these patients. All patients had a left-sided aortic arch. Operative procedure consisted of fixation of the aortic arch to the sternum and ventral thoracic wall. This was performed through a left anterior thoracotomy in 28 patients and a median sternotomy in 1 patient.

Operative technique
A left anterior thoracotomy is performed in the bed of the fourth rib. The thymus is partially resected. Pericardium is left closed (Fig 1). Four to five pledget-supported 3-0 to 5-0 sutures, depending on the age of the patient and the strength of the tissue, of a nonresorbable material are placed on the ventral surface of the aortic arch and the origin of the innominate artery (Fig 1). Only adventitia and media are caught by the sutures to avoid bleeding.

View larger version (29K): [in this window] [in a new window]   Fig 1. Position of the sutures on the ventral portion of the aortic arch.

View larger version (26K): [in this window] [in a new window]   Fig 2. By pulling the aortic arch ventrally (A) and to the right (B), the compression is relieved and the trachea is suspended by the connective tissue layer between aorta and trachea.

Follow-up was obtained by the outpatient department, the family doctor, or the patient or their parents by telephone interview.

	Results

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There was neither early nor late mortality. No intraoperative complications (eg, severe bleeding) were observed. The 6 preoperatively intubated patients were extubated within 8 days postoperatively. The remaining patients were extubated within the first 3 postoperative days. A reversible lesion of the phrenic nerve was noted in 2 patients, a pneumothorax in 3, and secondary wound healing in 1.

Follow-up was complete. The mean follow-up time was 8.1 ± 5.5 years (median, 10 years; range, 1 month to 15.4 years). In all but 1 patient the symptoms improved markedly or disappeared within days and the first 3 months of follow-up. In 1 patient the clinical condition did not improve postoperatively. Gastroesophageal reflux was found to be the major problem; after fundoplication, symptoms improved markedly. Control tracheal endoscopy was performed in all patients during the first 6 months after operation showing an almost complete resolution of the tracheal collapse in 27 patients. In 2 patients a mild to moderate tracheal obstruction remained, but was not symptomatic. In patients with a follow-up of more than 1 year (24 of 29 patients; 83%) an increased susceptibility to respiratory infections was found in 14 of 24 (58%) patients, requiring intermittent inhalational therapy with saline solution aerosol and oral antibiotic therapy if indicated. In 8 of these 14 patients (57%), these symptoms disappeared between the age of 3 to 6 years. All patients but 1 are reported to take part normally in everyday life and to participate in school sports. One patient operated on 6 years ago is still suffering from neurologic deficits related to preoperative apnea attacks. A scoliosis probably related to the left-sided anterior (aortopexy) or right-sided posterior (esophagus) thoracotomy was reported in 5 patients (17%).

	Comment

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In 1945 Mercer [4] described respiratory disturbances such as stridor, cyanosis, and apneas after surgical correction of esophageal atresia. The histopathologic investigations of Wailoo and Emery [5] revealed that the pathomorphologic correlative of the flaccid trachea in these patients is a primary dysplasia or hypoplasia of the cartilaginous elements and a prolongation of the muscular elements of the membranous part of the trachea. In 1976 Benjamin and coworkers [1] and Filler and associates [2] published their clinical observations in patients with respiratory complications after repair of esophageal atresia and described aortopexy as a surgical option in severe cases with life-threatening apnea attacks. The later use of aortopexy for this indication [6–11] is mainly related to the technique introduced by Gross and Neuhäuser [12] in 1948 and by Mustard and colleagues [13] in 1969. The incidence of severe tracheal instability requiring aortopexy after surgical correction of esophageal atresia ranges between 1.8% and 25.4% [1, 11, 14]. In 1990 we reported 83 consecutive children operated on for tracheoesophageal malformations. Seventy-three of these patients had esophageal atresia with tracheoesophageal fistula. Of the surviving 63 patients, 16 (25.4%) experienced a severely pronounced flaccid trachea defined by the presence of life-threatening apneas and an endoscopically verified tracheal collapse of more than two thirds of the lumen [14]. Taking all forms of esophageal atresia into account, this is by far the most common one. As the incidence of tracheal instability is markedly high in the group with the fistula, one has to assess whether prophylactic stabilization of the trachea during the atresia repair may be a therapeutic option.

As in our series, the vast majority of operations were deemed necessary during the first months of life [8, 9]; tracheal collapse as a result of pathologic tissue defect usually occurs early after operation for esophageal atresia.

Left anterior thoracotomy is the preferred surgical approach for aortopexy [1, 8, 9, 15]. Mustard and associates [13] used a right anterior thoracotomy in the bed of the second rib. This approach might be unfavorable because of adhesions related to the repair of the esophageal atresia. A sternotomy is used by some authors [1, 6, 8]. This again might be unfavorable as it will cause adhesions, which will complicate future mediastinal access. However, median sternotomy was used in one of our patients in whom additional intracardiac repair was necessary. Bullard and coworkers [16] reported on a new approach for aortopexy: An anterior mediastinal window is created using a small transverse incision over the left second and third intercostal space followed by subchondral excision of these costal cartilages to expose the mediastinum. This method might be more effective to prevent scoliosis and adhesions, but there might be less of an operative view. Although all patients in our series had a left-sided aortic arch, in case of a right-sided aortic arch a right anterior thoracotomy has to be considered.

An important point to mention is the vulnerability of the aorta. For this reason, Spitz [17] introduced a Dacron patch aortopexy as a less traumatizing method preventing adventitia trauma, especially in infants, but as seen in our series no trauma occurred if the aortic wall was handled with caution. Furthermore, this method is associated with leaving foreign material in the body, which is not necessary. Airway splinting offers semiinvasive options to reduce upper airway obstructions in patients with tracheal instability [18]. Tazuke and associates [19] reported on an infant with congenital cardiac anomalies including an enlarged aorta and additional esophageal atresia. After operation for esophageal atresia, tracheal instability occurred and aortopexy was performed at the age of 2 months, but the infant was not able to be weaned from the ventilator. At the age of 4.5 months an expandable (Palmaz) stent was placed, and extubation was performed 18 hours later. After 9 months of follow-up the clinical situation was satisfactory.

Partial tracheal resection as an alternative is not considered as a good therapeutic option because it does not change the morphologic abnormalities of the tracheal wall and the trachea may be at risk for an anastomosis stenosis.

Comparable good short-term and midterm results as found in our series were reported by other investigators [1, 2, 6, 8, 15]. Damage to the phrenic nerve as observed in some series is also known [9]. This may be caused by tension to the thymic tissue during dissection.

Until now long-term results of a large series have not been reported. We found that our long-term results are good with regard to the relief of airway obstruction. Agrawal and colleagues [20] found in patients who underwent aortopexy respiratory function and clinical findings at school age not to be suggestive of malacia. Hence, during the first 3 to 6 years there seems to be an increased susceptibility to respiratory infections, requiring intermittent specific therapy, an observation also reported by others [8]. An explanation for this finding might be the minor stability of the tracheal wall as described by Wailoo and Emery [5], leading to mucous retention and infection. An age-dependent stabilization of the tracheal wall may resolve this problem, but this remains speculative. We are concerned about the incidence of scoliosis, but this late complication is probably related to the thoracotomy technique and not to the aortopexy itself. In fact scoliosis could be related to the combination of left (aortopexy) and right (esophageal atresia operation) thoracotomies as the incidence of scoliosis in our institution was only 2.8% [21] after left posterolateral thoracotomy for closure of a patent ductus arteriosus. Another point to be mentioned is the potential need for mediastinal reoperation. If a patient who has undergone aortopexy requires sternotomy, as happened in one of our patients for closure of a ventricular septal defect, a special strategy is necessary to avoid severe bleeding.

In this patient a left anterior rethoracotomy was performed, and the ascending aorta and aortic arch were freed from the sternum, which was technically easy. Then, a routine median sternotomy was performed without difficulty.

In contrast to patients with esophageal atresia in whom structural changes of the trachea seem to be the major cause for tracheal instability, vascular abnormalities as seen in 2 of our patients may be the cause of severe respiratory compromise. Although our 2 patients showed an unremarkable long-term course, early repair is mandatory in these patients because, as reported by McElhinney and coworkers [22], an increase of duration of the airway compression leads to tracheomalacia and persistent symptoms even after the compression is relieved.

Aortopexy is a good option for patients with severe tracheal instability after operation for esophageal atresia and can be performed with no mortality and low morbidity. Aortopexy is effective to prevent further life-threatening apneas but does not prevent increased susceptibility to respiratory infections.

	References

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This Article Abstract Full Text (PDF) Alert me when this article is cited Alert me if a correction is posted 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): Jaime F. Vazquez-Jimenez Oliver J. Liakopoulos Bruno J. Messmer Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Vazquez-Jimenez, J. F. Articles by Messmer, B. J. Search for Related Content PubMed PubMed Citation Articles by Vazquez-Jimenez, J. F. Articles by Messmer, B. J. Related Collections Trachea and bronchi