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Ann Thorac Surg 1996;61:1824-1826
© 1996 The Society of Thoracic Surgeons

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Case Report

Successful Complete Tracheal Resection in a Three-Month-Old Infant

Great Ormond Street Hospital for Children, London, England, and University of Bonn, Bonn, Germany

Accepted for publication December 23, 1995.

	Abstract

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We report an infant with severe long-segment tracheal stenosis in whom the posterior trachea was formed by complete cartilage rings and the anterior trachea was almost totally formed by a solid cartilage plate. The child was successfully treated initially by complete resection of the trachea and primary end-to-end repair and subsequently with tracheal homograft transplantation for secondary stenosis.

	Introduction

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See also page 1827.

Tracheal stenosis, a life-threatening problem in young children, is often associated with pulmonary vascular sling and intracardiac lesions [1]. Long-segment congenital tracheal stenosis (LSCTS) represents the most challenging anatomic subgroup of this population. Although initial treatment of tracheal stenosis often involves resection of the stenosed segment with reanastomosis, this treatment is difficult in stenoses involving longer segments [2]. Recurrent tracheal stenosis is especially problematic.

We report a case of severe LSCTS treated with complete resection of the trachea and primary end-to-end repair with subsequent tracheal homograft transplantation (THT) for secondary stenosis.

A 3-month-old female infant presented with severe stridor. Investigation revealed LSCTS, pulmonary vascular sling, and ventricular septal defect. Exploration revealed severe LSCTS with the posterior trachea formed by complete cartilage rings and the anterior trachea almost totally formed by a solid cartilage plate (Fig 1a).

View larger version (29K): [in this window] [in a new window]   Fig 1. . (a) The initial tracheal pathology involved severe long-segment tracheal stenosis in which the posterior trachea was formed by complete cartilage rings and the anterior trachea was almost totally formed by a solid cartilage plate. (b) The first operation entailed complete resection of the trachea with anastomosis of the cricoid to the carina. (c) Over the ensuing months, the airway between the cricoid and the carina had lengthened but severely narrowed. (d) The anterior aspect of the airway was removed and the cricoid was left intact but split longitudinally along the posterior surface to allow the airway to splay open. (e) The tracheal homograft was trimmed to cover the remaining posterior airway and was sutured to the front surface of the airway.

Her postoperative course was complicated by airway instability, and she was extubated 15 days after the operation. Frequent pulmonary infections developed related to gastroesophageal reflux and aspiration. Bronchoscopy revealed severe recurrent tracheal stenosis at the anastomotic site, which was unresponsive to endoscopic dilation. A silicone endoluminal stent was inserted to maintain the airway, but this eventually became unstable.

At the age of 18 months, intubation was necessary. One week later, she underwent THT to manage her airway instability and severe recurrent tracheal stenosis. The procedure was performed through a redo median sternotomy using cardiopulmonary bypass with iliac cannulation before sternotomy. Extremely dense adhesions, calcification, and cartilage formation were encountered at the previous cricoid-to-carina anastomosis. The airway between the cricoid and the carina had lengthened but severely narrowed (Fig 1c). The airway was opened anteriorly, the laryngeal cartilage was split in the midline, and the cricoid cartilage was split in four places around its perimeter. The anterior aspect of the airway was removed from the cricoid cartilage to the carina. The cricoid wall was left intact but split longitudinally along the posterior surface, allowing the airway to splay open.

Stored cadaveric tracheal homograft tissue (fixed in formalin, washed in methiolate and alcohol, and stored in acetone) was fashioned and trimmed to cover the remaining posterior airway (Fig 1d). The homograft was then sutured to the front surface of the airway using interrupted absorbable monofilament sutures (Fig 1e). Before completion of the anastomosis, a silicone intraluminal stent (Dumon stent; Axiom, Lyon, France) was placed.

After bronchoscopy to confirm graft patency and perform bronchial toilet, the graft was sealed with Tisseel fibrin glue (Immuno AG, Vienna, Austria). Cardiopulmonary bypass was weaned. Hemostasis and routine closure followed.

The postoperative course was complicated by laryngeal and pharyngeal edema treated with steroids. Frequent bronchoscopy was required to remove granulation tissue until the airway stabilized and epithelialized. The stent was removed endoscopically 14 weeks after transplantation. Subsequently, the patient has remained free of any airway problems. Bronchoscopy performed 13 months postoperatively revealed a completely epithelialized homograft with a normal airway caliber from cricoid to carina (Fig 2). She is currently at home, asymptomatic, and enjoying a normal activity level 16 months after transplantation.

View larger version (100K): [in this window] [in a new window]   Fig 2. . Bronchoscopy performed 13 months postoperatively revealed a completely epithelialized homograft with a normal airway caliber from cricoid to carina.

	Comment

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Herberhold [7] initially described THT in the German otolaryngology literature. Remarkable success has been achieved using this technique through a cervical incision for proximal tracheal stenoses in adults and some children. Distal tracheal and proximal bronchial lesions can be managed with THT via a median sternotomy. We have performed THT in 5 children, 4 of whom achieved functional airways and ventilator independence after transplantation [8]. Cardiopulmonary bypass facilitates the dissection necessary for THT in more distal lesions and obviates the need for airway maintenance. Postoperative management requires a multidisciplinary approach involving cardiac surgeons, intensivists, otolaryngologists, and pediatricians. Immunosuppression is not necessary because the chemically treated and preserved trachea is a biocompatible implant that has no cellular viability [7]. Frequent bronchoscopy is necessary to remove granulation tissue around the intraluminal stent until eventual airway stabilization and epithelialization. Stent removal is performed endoscopically. Subsequent endoscopy usually reveals an epithelialized and functional airway. Biopsy demonstrates this epithelium to be ciliated columnar respiratory epithelium.

In summary, THT provides a technique of tracheal reconstruction for patients with no useful tracheal tissue of their own. This case report demonstrates the technical feasibility of removing the entire trachea in an infant.

	Footnotes

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Address reprint requests to Dr Elliott, Cardiothoracic Unit, Great Ormond Street Hospital for Children, London, England WCIN 3JH.

	References

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1. Andrews TM, Cotton RT, Bailey WW, Myer CM, Vester SR. Tracheoplasty for congenital complete tracheal rings. Arch Otolaryngol Head Neck Surg 1994;120:1363–9.[Abstract/Free Full Text]
2. Grillo HC. Slide tracheoplasty for long-segment congenital tracheal stenosis. Ann Thorac Surg 1994;58:613–21.[Abstract]
3. Messineo A, Forte V, Joseph T, Silver MM, Filler RM. The balloon posterior tracheal split: a technique for managing tracheal stenosis in the premature infant. J Pediatr Surg 1992;8:1142–4.
4. Cosentino CM, Backer CL, Idriss FS, et al. Pericardial patch tracheoplasty for severe tracheal stenosis in children: intermediate results. J Pediatr Surg 1991;26:879–85.[Medline]
5. Willner A, Velez FJ. Rib-muscle flap for the repair of congenital tracheal stenosis. Am Otol Rhinol Laryngol 1994;103:601–8.
6. Tsugawa C, Nishijima E, Muraji T, Matsumoto Y. The use of omental pedicle flap for tracheobronchial reconstruction in infants and children. J Pediatr Surg 1991;26:762–5.[Medline]
7. Herberhold C. Transplantation von Larynx und Trachea beim Menschen. Verh Dtsch Ges Hals-Nasen-Ohren-Heilkunde Eur Arch Oto-Rhinol-Laryngol 1992;2(Suppl 1):247–55.
8. Elliott MJ, Haw MP, Jacobs JP, Bailey CM, Evans JNG, Herberhold C. Successful tracheal replacement in children using cadaveric human tracheal homograft. Eur J Cardiothorac Surg (in press).

This article has been cited by other articles:

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				C. L. Backer, C. Mavroudis, M. E. Dunham, and L. D. Holinger Repair of congenital tracheal stenosis with a free tracheal autograft J. Thorac. Cardiovasc. Surg., April 1, 1998; 115(4): 869 - 874. [Abstract] [Full Text] [PDF]

				J. P. Jacobs, M. J. Elliott, M. P. Haw, C. M. Bailey, C. Herberhold, and S. M. R. de Leval PEDIATRIC TRACHEAL HOMOGRAFT RECONSTRUCTION: A NOVEL APPROACH TO COMPLEX TRACHEAL STENOSES IN CHILDREN J. Thorac. Cardiovasc. Surg., December 1, 1996; 112(6): 1549 - 1560. [Abstract] [Full Text]

This Article Abstract 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): Jeffrey P. Jacobs Martin J. Elliott Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Jacobs, J. P. Articles by Elliott, M. J. Search for Related Content PubMed PubMed Citation Articles by Jacobs, J. P. Articles by Elliott, M. J.