Ann Thorac Surg 2003;75:1325-1328
© 2003 The Society of Thoracic Surgeons
Case report
Management of complex tracheo-aortic fistula following neonatal tracheal reconstruction
Marco Ricci, MDa,
Gordon A. Cohen, MD, PhDa,
Derek Roebuck, MDa,
Martin J. Elliott, MDa*
a Cardiothoracic Unit and Tracheal Team, Great Ormond Street Hospital for Children NHS Trust, London, United Kingdom
Accepted for publication September 27, 2002.
* Address reprint requests to Dr Elliott, Cardiothoracic Unit, Great Ormond Street Hospital for Children NHS Trust, Great Ormond Street, London WC1N 3JH, UK
e-mail: elliom1{at}gosh.nhs.uk
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Abstract
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We describe an unusual case of tracheo-aortic fistula, which occurred after tracheal surgery and tracheal stenting. The management of this complex case and the surgical technique used for repair are discussed and illustrated. Repair of the aortic arch was accomplished using a modified technique of regional low-flow perfusion, similar to that described for neonatal aortic arch reconstruction. This strategy allowed maintenance of cerebral, myocardial, and systemic perfusion during arch repair, thus avoiding total circulatory arrest.
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Introduction
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Catastrophic bleeding due to erosion of major vascular structures has been recognized as an unusual but potentially lethal complication of tracheostomy [1], complex tracheal surgery [2, 3] and tracheal stenting [4–6]. Most often, this complication involves pressure necrosis on the innominate artery as a result of the intimate relationship between this vessel and the anterior surface of the trachea [2]. Involvement of vascular structures other than the innominate artery is exceedingly rare and only a few cases of pulmonary artery rupture have been reported [4, 5]. In a historical series reported by Deslaurier and colleagues in 1975 [2], innominate artery rupture occurred in as many as 8% of the patients undergoing major resective/reconstructive tracheal surgery. Although the incidence reported in more recent series has been somewhat lower [7, 8], this dreadful complication of tracheal surgery has not completely disappeared. Furthermore, its occurrence has been observed recently after tracheal stenting [4, 5], which is being used with increasing frequency in the management of tracheal disease, alone or in combination with surgery [9].
The initial management of patients with tracheo-innominate fistula is difficult. Success rates are variable and largely depend on the patient’s condition at presentation [2]. Bleeding outside a hospital setting is often fatal. Timely intervention, prompt airway establishment, and rapid control of bleeding are all significant factors in the outcome. Further surgical management of those patients who initially survive is relatively straightforward. Management usually encompasses reconstruction (or ligation) of the innominate artery, and occasionally tracheal reconstruction [2].
In contrast to the isolated rupture of the innominate artery, involvement of the aortic arch poses an even greater threat to patient survival and increases substantially the complexity of surgical repair. The aim of this brief report is to describe the surgical technique used in the management of tracheo-aortic fistula following tracheal surgery, which was observed in a patient who previously underwent tracheal reconstruction and stenting. In this patient, reconstruction of the aortic arch was performed under hypothermic regional low-flow perfusion, avoiding total circulatory arrest.
Our experience of tracheo-aortic fistula following tracheal surgery consists of one patient who was born with left pulmonary artery sling and associated long segment tracheal stenosis. At 1 month old, the child underwent one-stage repair of pulmonary artery sling and patch tracheoplasty using autologous pericardium. During the following 2 years he made good progress, although he developed recurrent episodes of airway obstruction due to tracheo-bronchomalacia and granulation tissue within the tracheal stent. These required numerous bronchoscopic investigations with repeated laser vaporization, balloon dilatation, and insertion of balloon-expandable metallic tracheo-bronchial stents (Palmaz, Johnson and Johnson Interventional Systems Co., Warren, NJ) (Fig 1A).
Of note, one of the metallic stents was placed within the distal trachea, with the distal end at about 2 cm from the carina (Fig 1A). Granulation tissue seemed to recur particularly within the distal portion of the tracheal stent, requiring multiple sessions of balloon dilatation (Fig 1A).
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Fig 1. (A) Multiple tracheo-bronchial stents placed after tracheal reconstruction. The radio-opacity within the tracheal stent corresponds to the area where granulation tissue recurred. (B) Contrast-enhanced chest computed tomography scan, revealing possible erosion of the aortic arch. (C) Aortic angiography, suggesting aneurysmal dilatation at the base of the left carotid artery.
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At 2 years old, the child presented with mild hemoptysis, which promptly resolved spontaneously. A few days later, because of the reappearance of hemoptysis and recurrent pattern of distal tracheal obstruction, a contrast-enhanced computed tomography scan (CT) of the chest was performed (Fig 1B). The CT suggested compression and possibly erosion of the aortic arch by the stented portion of trachea. Further investigation included an aortic angiography with selective injection of the innominate and left common carotid arteries (Fig 1C), in order to further delineate the anatomy and the relationship between airway and vascular structures. Neither the aortogram nor the selective injections conclusively demonstrated injury to the innominate artery or aortic arch, although mild aneurysmal dilatation of the left carotid artery at the take-off from the aorta was noted (Fig 1C). These findings were confirmed by simultaneous bronchoscopy and angiography, after which the patient was taken to surgery for urgent repair.
After the sternum was reopened in the midline, the heart and great vessels were dissected from the previous adhesions. To avoid massive bleeding, care was taken to avoid dissection of the innominate artery and distal ascending aorta/aortic arch at this stage. After full heparinization, the proximal and distal ascending aorta were separately cannulated with two DLP arterial cannulas (DLP, Medtronic, Minneapolis, MN) (Fig 2B),
which were both connected to the cardiopulmonary bypass (CPB) machine. Double aortic cannulation would subsequently allow myocardial preservation and cerebral perfusion during arch repair and avoidance of total hypothermic circulatory arrest. Venous drainage was obtained by a single two-stage venous cannula inserted in the right atrium (Fig 2B). CPB was established, and the patient was cooled down to 16°C. Further dissection was carried out. Once the heart stopped ejecting, a left atrial vent was inserted to avoid distension of the left-sided chambers.
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Fig 2. (A) Full-thickness erosion of the aortic arch by the stented portion of the trachea. (B) Strategy of cannulation for cardiopulmonary bypass (CPB). Double aortic cannulation (proximal and distal ascending aorta) and right atrial cannulation with single two-stage venous cannula. Arrow indicates direction of blood flow. (C) At 16°C, the ascending aorta is clamped, the distal aortic cannula is advanced into the left carotid artery, CPB flow is diminished to 50 mL·kg-1·min-1, the head vessels are snared, and the innominate artery is clamped and divided distally. A Foley is inserted into the descending thoracic aorta. The arrows indicate the area of erosion of the aortic arch, which is now safe to dissect. (D) Reconstruction of the aortic arch under regional hypothermic low-flow perfusion. The brain and myocardium are being perfused. (E) Full CPB flow is reestablished, and rewarming is begun. The trachea is reconstructed with a tracheal homograft. (F) At last, the innominate artery is reconstructed with a Gore-tex graft.
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At 16°C, the ascending aorta was cross-clamped between the two aortic cannulas (Fig 2C). The distal arterial cannula originally placed in the distal ascending aorta was advanced into the left carotid artery (Fig 2C). As CPB flow was diminished to approximately 50 ml · kg-1 · min-1, the head vessels were dissected further, and snared. Thus, hypothermic low-flow perfusion of the brain and myocardium was established. At this stage, the base of the innominate artery and the posterior arch were separated from the anterior surface of the trachea (Fig 2C). This was the area where the vascular injury was expected to be; this maneuver was greatly facilitated by division of the innominate artery at its origin from the aorta, and by the placement of a large Foley catheter through the aortic opening (Fig 2D). The balloon of the catheter was inflated within the proximal descending thoracic aorta so as to avoid back bleeding (Fig 2D). Of note, this strategy allowed to perform the critical part of the operation (i.e., dissection of innominate artery and aortic arch from the trachea) under controlled conditions, avoiding the risk of exsanguination. Furthermore, total circulatory arrest and cardioplegic arrest were avoided altogether. Once the full extent of aortic arch involvement was established, arch reconstruction was undertaken using a bovine pericardial patch, which was sewn to the edges of the aorta using 6/0 Prolene, as illustrated in Figure 2D. As arch repair was completed, the head vessels were unsnared, full CPB support was resumed, and rewarming was begun.
Tracheal reconstruction was performed at this stage by using a cadaveric tracheal homograft, as illustrated in Figure 2E. This preceded reconnection of the innominate artery to the aorta (Fig 2F) to facilitate exposure of the distal trachea. In our patient, the tracheal stent had completely eroded through the tracheal and aortic walls, causing a considerable fibrotic reaction. As a result, a portion of anterior tracheal wall along with the stent had to be removed. After reconstructing the trachea, the innominate artery was reconnected to the aorta using a 6- mm Gore-tex graft (Gore-tex, Flagstaff, AZ) (Fig 2F). Rewarming was completed, and CPB was discontinued. Before closing the chest, a Gore-tex sheet was placed between trachea and great arteries.
Despite the postoperative course being complicated by the occurrence of severe mediastinitis due to Pseudomonas aeruginosa (previously isolated from the airway), the child made a full recovery.
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Comment
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Data regarding the epidemiology, diagnosis, and surgical treatment of tracheo-innominate fistula after adult and pediatric tracheal surgery are widely available in the literature [1, 2]. In contrast, information regarding the management of patients with tracheo-aortic fistula is lacking, perhaps because this condition is almost always lethal, and patients often succumb before treatment can be instituted. In our series of pediatric patients who underwent major resective/reconstructive tracheal surgery (51 patients), massive bleeding due to vascular erosion was observed in 4 patients, and in only 1 patient was due to involvement of the aorta.
Although rare, involvement of the aortic arch changes considerably the strategy of intervention. Most of the cases of innominate artery rupture and tracheo-innominate fistula can be managed simply by dealing directly with the innominate artery (ligation or reconstruction) [2]. In contrast, when the presence of an injury extending to the aortic arch is suspected, full support with CPB and a complex strategy of repair are required. As our experience is limited to only 1 patient, to report definite conclusions regarding clinical presentation, diagnosis, and surgical management of this dreadful complication of tracheal surgery are impossible. However, a few points may be worthy of discussion. In our patient, arch repair was undertaken while using a technique of modified regional low-flow perfusion [10–12]. This technique, designed to avoid total circulatory arrest in the setting of neonatal aortic arch operations, closely resembles that originally described by Asou and colleagues [10], and subsequently popularized by others [11, 12]. By adapting this technique to our patient, we were able to avoid total circulatory arrest. Also, this strategy allowed us to safely dissect the innominate artery and the aorta from the trachea, avoiding the risk of a catastrophic hemorrhage. We also wish to emphasize the value of the use of tracheal homograft tissue in redo-tracheal surgery [13].
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Acknowledgments
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We would like to thank all the members of the Tracheal Team at Great Ormond Street Hospital for Children NHS Trust. Their work was instrumental in obtaining a successful outcome. They are: Niyi Ade-Ajayi, Martin Bailey, Catherine Dunne, Hilary Glaisyer, Ben Hartley, Clare McLaren, Quen Mok, Clair Noctor, Nick Piggott, and Steven Scuplack. Research at the Institute of Child Health and Great Ormond Street Hospital for Children NHS Trust benefits from Research and Development funding received from the NHS Executive.
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References
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Abstract
Introduction
