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Ann Thorac Surg 2004;77:1420-1422
© 2004 The Society of Thoracic Surgeons

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Case report

Stenting of the mainstem bronchus in children: a word of caution

^a Divisions of Cardiothoracic Surgery and Pediatric Cardiology, Keck School of Medicine, University of Southern California and Childrens Hospital Los Angeles, Los Angeles, California, USA

Accepted for publication May 14, 2003.

^* Address reprint requests to Dr Wells, Division of Cardiothoracic Surgery, Childrens Hospital Los Angeles, 4650 Sunset Blvd, MS 66, Los Angeles, CA 90027, USA.

	Abstract

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We present two patients with critical hemorrhage following expandable metallic stenting of the left mainstem bronchus in children. Stent migration with erosion into a bronchial artery leading to mycotic pseudoaneurysm formation and overwhelming pulmonary hemorrhage occurred in both patients. One patient died from uncontrollable hemoptysis. The second patient was salvaged by left pneumonectomy and patch repair at the site of mycotic aortic rupture. Stenting of a mainstem bronchus, particularly in cyanotic children must be approached with extreme caution.

	Introduction

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Airway obstruction from tracheo or bronchomalacia may pose life-threatening problems. When the etiology is aortic or great vessel compression, then aortopexy may relieve the problem [1]. In patients where this is not successful or feasible, placement of a metallic expandable stent has emerged as an alternative therapy [2–6]. Stents have most frequently been used for relief of tracheal obstruction, but occasionally they have been deployed in the mainstem bronchus. The potential for stent erosion through the thin bronchial wall has been discussed, but not well documented in the clinical setting. Our experience with two severe complications of mainstem bronchus stenting in cyanotic children is described and discussed below.

	Case reports

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Patient 1
A 4-year-old girl with a dilated cardiomyopathy and documented severe pulmonary hypertension required a tracheostomy for respiratory failure. The patient had persisting collapse of the left lung and computed tomographic (CT) scan revealed left main bronchus narrowing. Bronchoscopy revealed severe left mainstem bronchomalacia 12-mm in length. A 2-cm Wallstent (Boston Scientific, Watertown, MA) was placed in the left mainstem bronchus and dilated to 7 mm.

One-month later the patient presented to the emergency department with copious bright red blood through tracheostomy. History was positive for cough and secretions, but negative for fever or other suggestion of upper respiratory infection. Chest roentgenogram was clear. Following immediate transfer to our institution repeat films revealed evidence of a left lower lobe infiltrate. While being prepared for bronchoscopy the patient had massive hemoptysis requiring transfusion and vasopressor support. Emergency arteriography illustrated a ruptured pseudoaneurysm adjacent to the stented left bronchus with active bleeding into the lung (Fig 1). Despite coil embolization of the involved bronchial artery, the patient had progressive desaturation and hemodynamic instability. She expired within hours of the embolization procedure.

View larger version (123K): [in this window] [in a new window]   Fig 1. Angiogram of patient 1. The bronchial artery pseudoaneurysm is demonstrated with contrast injection, and constrast from the pseudoaneurysm is seen in the airways of the left lung.

At 24 months the patient underwent a bidirectional Glenn with an additional 3-mm modified Blalock shunt to augment pulmonary flow. The left lung appeared normal on chest roentgenogram.

When she was 3 years old the patient had a cardiac catheterization in anticipation of a complete Fontan. Pulmonary artery mean pressure was 15 mm Hg and systemic saturation was in the high 70s. The left lung appeared normal and the patient was felt to be a Fontan candidate.

Three-days after the catheterization there was acute deterioration with respiratory distress and hypoxia. The left lung was opacified on chest roentgenogram. After transfer to our facility bronchoscopy revealed a complete left mainstem bronchus obstruction due to organized thrombus. Magnetic resonance imaging (MRI) demonstrated a contained rupture of the proximal descending aorta with a large pseudoaneurysm (Fig 2). Major airways on the left side were obliterated.

View larger version (109K): [in this window] [in a new window]   Fig 2. Contrast magnetic resonance imaging of patient 2 demonstrating the bronchial artery pseudoaneurysm and site of mycotic descending aorta rupture.

	Comment

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Endoairway stenting has been used in difficult patients with malacia or other forms of central trachobronchial obstruction. Although recurrent stenosis due to granulation tissue has been the main complication of stents, erosion and migration of the metallic foreign body has also occurred. Autopsy studies have revealed full thickness stent erosion into the mediastinum [2]. Erosion and migration would be expected to be more common from the mainstem bronchus as opposed to the trachea because the airway wall is thinner and has less cartilage support.

The potential for stent erosion is of particular concern in the cyanotic patient who may have dilated bronchial collateral vessels in close proximity to the stent site. It appears that in both of our patients erosion into a bronchial vessel led to a pseudoanuurysm and airway hemorrhage. In patient 2, infection within the mediastinal pseudoaneurysm extended to the proximal descending aorta causing a mycotic rupture. The site of aortic disruption was well away from the stent itself.

In the acute setting, fluoroscopic, MRI, and CT angiography are all diagnostic. In the presence of active hemorrhage, interventional catheterization offers potential hemostasis through covered-stent or intravascular coil placement. However, in subacute or chronic presentation, three-dimensional reconstructions produced by MRI or CT better define bronchial-arterial relationships and surgical roadmaps. CT angiography is fast and simple, but poorly suited for serial evaluation because of nontrivial radiation exposure. Contrasted-enhanced magnetic resonance angiography offers outstanding anatomic detail, without ionizing radiation, making it the modality of choice for screening or serial examinations. Metal artifact from wall stents is minimal and presents no safety hazard to the patients.

Despite the risks, metallic stenting may be the only alternative in patients with complex airway obstruction. Once vascular compression has been minimized, stenting may be the only option to avoid the complications of ongoing mechanical ventilation. Without positive pressure ventilation these patients have persisting pulmonary collapse, consolidation, and problems with infection. In some patients, including both patients described in this report, it may be difficult to keep the lung up even with assisted ventilation. Stents are highly effective in correcting airway obstruction and allowing patients to come off the ventilator. However, as noted by Jacobs and coworkers [7], wire stents, particularly in the bronchus, are almost always nonremovable and they present an ongoing risk of erosion. Given the experience noted in this report we are avoiding expandable metallic stents in the mainstem bronchus in cyanotic children where the likelihood of large bronchial collateral vessels is high.

Nicolai and colleagues [8] has recently reported an experience with 13 bronchial stenting events in 6 children using a soft mesh stent (SS34 to 90; Boston Scientific) or nitinol (Nitinol briliary or Ultraflex bronchial; Boston Scientific). These devices were not subject to the erosion and migration problems that have been associated with the Palmaz stent, and have also been successfully removed on several occasions. The authors have concluded that nitinol stents are probably the best choice for the pediatric airway.

	References

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1. Filler R.M. Current approaches in tracheal surgery. Pediatr Pulmonol Suppl 1999;18:105-108.[Medline]
2. Filler R.M., Forte V., Chait P., et al. Tracheobronchial stenting for the treatment of airway obstruction. J Pediatr Surg 1998;33:304-311.[Medline]
3. Fraga J.C., Filler R.M., Forte V., Bahoric A., Smith C. Experimental trial of balloon-expandable, metallic Palmaz stent in the trachea. Arch Otolaryngol Head Neck Surg 1997;123:522-528.[Medline]
4. Furman R.H., Backer C.L., Dunham M.E., Donaldson J., Mavroudis C., Holinger L.D. The use of balloon-expandable metallic stents in the treatment of pediatric tracheomalacia and bronchomalacia. Arch Otolaryngol Head Neck Surg 1999;125:203-207.[Abstract/Free Full Text]
5. Orons P.D., Amesur N.B., Dauber J.H., Zajko A.B., Keenan R.J., Iacono A.T. Balloon dilation and endobronchial stent placement for bronchial strictures after lung transplantation. J Vasc Interv Radiol 2000;11:89-99.[Medline]
6. Wood D.E. Airway stenting. Chest Surg Clin N Am 2001;11:841-860.[Medline]
7. Jacobs J., Quinttessenza J.A., Botero L.M., et al. The role of airway stents in the management of pediatric tracheal, carinal, and bronchial disease. Eur J Cardiothorac Surg 2000;18(5):505-512.[Abstract/Free Full Text]
8. Nicolai T., Huber R.M., Reiter K., Merkenschlager A., Hautmann H., Mantel K. Metal airway stent implantation in children: follow-up of seven children. Pediatr Pulmonol 2001;31:289-296.[Medline]

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This Article Abstract Full Text (PDF) Alert me when this article is cited Alert me if a correction is posted Citation Map 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): Winfield J. Wells Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Wells, W. J. Articles by Wood, J. C. Search for Related Content PubMed PubMed Citation Articles by Wells, W. J. Articles by Wood, J. C. Related Collections Pleura