Eur J Cardiothorac Surg 2000;17:488-491
© 2000 Elsevier Science NL
Surgical management of idiopathic subglottic tracheal stenosis
Antonis Loutsidisa,
Charalambos Zisisa,
Konstantia Larioub,
Ion Bellenisa
a Department of Thoracic and Vascular Surgery, Evangelismos Hospital, Athens, Greece
b Department of Pathology, Evangelismos Hospital, Athens, Greece
Corresponding author. Omirou 19, Glyfada, 16675, Athens, Greece. Tel.: +30-1-9650436
e-mail: dkakats{at}cc.uoa.gr
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Abstract
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A case of idiopathic subglottic tracheal stenosis in a 50-year-old female is presented. A procedure of single-stage resection of the lesion and end-to-end anastomosis was performed with excellent results 1 year after the operation. The clinical, paraclinical, diagnostic, therapeutic and histopathological aspects of this rare pathologic condition are discussed and the literature on this topic is reviewed.
Key Words: Fibrotic lesion • Circumferential stenosis • Laryngotracheal reconstruction • Thyrotracheal anastomosis • Keloidal-type tissue • Dilatation • Laser resection
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1. Introduction
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Idiopathic laryngotracheal stenosis (ILTS) has been accepted as a particular entity including lesions, which share typical features of location, configuration, clinical evolution and specific pathology [1]. The reported cases in the literature are very few. Grillo's series is the largest with 49 cases, whereas Pearson [2] has presented nine cases and Couraud [3] five cases. Circumferential fibrotic stenosis is the usual macroscopic appearance of the lesion, located most often in the subglottic larynx and extending at varying distances predominantly in women.
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2. Case report
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A 50-year-old woman was accepted with increasing stridor, dyspnea at rest, noisy breathing and wheezing. No change in her voice existed and her medical history was free. Intubation, inhalation, burn or traumatism of the neck in the past were not reported. During the last 2 years, she had been managed in another center with successive laser resection of the stenotic area and concomitant dilatations in increasing frequency.
Marked subglottic stenosis as circumferential fibrotic focal lesion, 3 cm long, resulting in a lumen 3 mm in diameter at its narrowest portion, was evident on X-ray plain and computerized tomographies (Fig. 1a,b). In fiberoptic examination, it was characterized macroscopically as inflammatory and bloody.
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Fig. 1. Laryngotracheal stenosis in computerized tomography: (a) transverse plane, (b) sagittal and coronal plane.
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Antineutrophil cytoplasmic antibody testing was negative and cultures obtained from the trachea did not identify B. Koch, fungi or non-anticipated respiratory flora. The particularities of the case and the risks of the scheduled surgical procedure were analyzed to the patient and single-stage resection-repair with laryngotracheal reconstruction was performed. A collar incision provided exposure of the proximal two-thirds of the trachea. The anterior and anterolateral aspects of the trachea from the cricoid to the mid-trachea were mobilized in order to avoid tension of the anastomosis. Although, bronchoscopically and radiographically the lesion was located 1.5 cm below the glottis, the intraoperative macroscopical finding in this position was narrow lumen and shrunken mucosa. So, the resection was extended up to a limit of a few mm from the inferior margins of the vocal folds, where the appearance of the tissue was normal. Recurrent laryngeal nerve injury was avoided by limited lateral dissection and development of the plain on the anterior surface of the posterior cricoid plate right up to the level of the cricoarytenoid joints. As a result, the first ring of the trachea and the anterior portion of the subglottic larynx were resected in an arcuate line to include part of the lateral laminae of the cricoid cartilage, its anterior portion and most of the cricothyroid membrane. There was no extension of the process beyond the tracheal wall into the surrounding tissue. An end-to-end thyrotracheal anastomosis was finally performed using continuous sutures in the posterior wall and interrupted sutures in the anterior. The patient was extubated in the operating room and postoperative course was uneventful without any problem in aspiration or deglutition. Head immobilization was performed with a thread between the chin and the midline of the chest over the manubrium for 7 days. Oral feeding begun on 3rd postoperative day and problems with liquid food did not exist. Ten days after the operation the patient was discharged doing well. The pathologic examination of the tracheal wall showed pronounced development of a cheloid-like tissue in the lamina propria. Hemorrhage and ulceration of the surface were also found. This finding is compatible with the diagnosis of idiopathic laryngotracheal stenosis (Fig. 2).
One year after the operation, clinical, radiological and bronchoscopic examinations of the patient confirm an excellent recovery, as the voice and the respiration are quite normal with normal diameter of the airway.
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3. Comment
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Laryngotracheal stenosis may be caused by a series of pathologic conditions, acquired or congenital. The former is manifested usually early in infancy, while the latter include prolonged intubation for ventilation purposes as the commonest reason, external trauma to the trachea, inhalation burns, irradiation and infective causes. Once all the other possible causes of tracheal stenosis have been excluded, then a diagnosis of a truly idiopathic tracheal stenosis can be made [4]. Although it is entirely possible that more than one cause may be responsible for ILTS, similarities are so great that a single origin seems likely [1].
It is recommended to manage these patients initially conservatively with laser resection and dilatation [1,5], but if dilatation is required frequently and it looks as if repair can be done safely, then surgical repair and reconstruction is preferred.
In Grillo's series, interesting is the fact that only three of their 49 patients are male. The data of other series are similar, but known cause for this female predilection does not exist.
The clinical manifestations are similar to those of postintubation or posttraumatic strictures, including progressive shortness of breath, accompanied by noisy breathing, wheezing, stridor and hoarseness.
The radiographic appearance can be variable: stenosis from 2 to 4 cm long and lumen between 3 and 5 mm in diameter at the narrowest portion. The narrowing can be concentric and shaped like an hourglass or eccentric. According to Bhalla et al. [6] CT scan did not provide additional clinically useful information when compared with the radiographs and plain tomograms.
Useful criteria in differential diagnosis of ILTS are the duration (average duration of symptoms of 2 years), radiological and bronchoscopic appearance with absence of a history of intubation or trauma, typical predilection of middle-aged women and finally histopathological findings.
As long as the choice of surgical single-stage laryngotracheal reconstruction prevails, emphasis is given to careful preoperative treatment of infection and inflammation, as well as a meticulous muco-mucosal approximation of healthy margins at the anastomosis [3]. According to Couraud et al., it is preferable to delay the surgical procedure to allow the tracheal resection anastomosis in health margins. Patients with frank mucosal ulcerations are best deferred for later correction [1].
On the other hand, later progression of stenosis after resection has not been observed, even though the proximal line of resection passes through abnormal tissue [1].
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References
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Grillo H.C., Mark E.J., Mathisen D.J., Wain J.C. Idiopathic laryngotracheal stenosis and its management. Ann Thorac Surg 1993;56:80-87.[Abstract]
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Maddaus M.A., Toth J.L.R., Gullane P.J., Pearson F.G. Subglottic tracheal resection and synchronous laryngeal reconstruction. J Thorac Cardiovasc Surg 1992;104:1443-1450.[Abstract]
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Couraud L., Jougon J., Velly J.-F. Surgical treatment of nontumoral stenoses of the upper airway. Ann Thorac Surg 1995;60:250-260.[Abstract/Free Full Text]
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Harries P.G., Mason P.S., Ramsay A.D., Carruth J.A.S. Idiopathic tracheal stenoses. J Laryngol Otol 1996;110:973-975.[Medline]
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Park S.S., Streitz J.M., Rebeiz E.E., Shapshay S.M. Idiopathic subglottic stenosis. Arch Otolaryngol Head Neck Surg 1995;121:894-897.
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Bhalla M., Grillo H.C., McLoud T.C., Shepard J.-A.O., Weber A.L., Mark E.J. Idiopathic laryngotracheal stenosis: radiologic findings. Am J Roentgenol 1993;161:515-517.[Abstract/Free Full Text]
Received August 9, 1999;
received in revised form December 29, 1999;
accepted February 7, 2000.
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Abstract
1. Introduction
