Ann Thorac Surg 2008;85:1438-1439. doi:10.1016/j.athoracsur.2007.10.032
© 2008 The Society of Thoracic Surgeons
Case Reports
Idiopathic Tracheal Stenosis: Successful Outcome With Antigastroesophageal Reflux Disease Therapy
Ricardo Mingarini Terra, MDa,*,
Israel Lopes de Medeiros, MDa,
Hélio Minamoto, MDa,
Ary Nasi, MDb,
Paulo Manuel Pego-Fernandes, MDa,
Fábio Biscegli Jatene, MDa
a Division of Thoracic Surgery, Hospital das Clínicas, University of São Paulo Medical School, São Paulo, Brazil
b Division of Gastroenterology, Hospital das Clínicas, University of São Paulo Medical School, São Paulo, Brazil
Accepted for publication October 8, 2007.
* Address corrrespondence to Dr Terra, Al. Fernão Cardim 161, ap. 61, São Paulo, SP CEP 01403-020, Brazil (Email: rmterra{at}uol.com.br).
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Abstract
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There is controversial evidence that gastroesophageal reflux disease (GERD) is an etiologic factor for idiopathic laryngotracheal stenosis. We present the case of a 44-year-old woman with symptomatic tracheal stenosis managed as idiopathic stenosis. She underwent six endoscopic dilations during 1 year, and before surgery she underwent 24-hour esophageal pH monitoring that documented GERD. Anti-GERD treatment was started, which was confirmed as effective with 24-hour esophageal pH monitoring 3 months later. At 2-year follow-up the patient remained free of symptoms and no additional airway procedure was necessary. A close relationship between anti-GERD therapy and clinical outcome was noted.
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Introduction
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Idiopathic laryngotracheal stenosis (ILTS) is a rare disease characterized by inflammatory cicatricial stenosis at the level of the cricoid cartilage and upper trachea. It is more frequent in women in their fourth and fifth decades who have no identifiable cause of airway stenosis [1]. Many authors have implicated gastroesophageal reflux disease (GERD) as a cause of idiopathic tracheal stenosis [2, 3]. We present a case of tracheal stenosis that improved with clinical treatment of GERD.
A 44-year-old woman was admitted to our institution with dyspnea on exertion that had developed during 2 years. In the last 6 months her symptoms had progressed to dyspnea at rest, with noisy breathing and stridor. She had no medical history of airway infections; intubation; or inhalation, burn, or other trauma to the neck. Physical examination revealed an obese patient (body mass index, 36 kg/m2) with stridor.
Stenosis of the upper part of the trachea, 6 mm in diameter at its narrowest portion and 2 cm long, was evident at computed tomography. At bronchoscopy, the mucosa had an inflammatory aspect. Biopsy specimens were obtained, and histopathologic analysis showed focal squamous metaplasia and fibrosis. Testing for antineutrophil cytoplasmic antibody with indirect immunofluorescence assay, for antinuclear antibodies with the HEp-2 cell assay, and for rheumatoid factor yielded normal results. Cultures obtained from the trachea did not reveal Mycobacterium tuberculosis or fungi. On the basis of clinical and laboratory findings, idiopathic tracheal stenosis was diagnosed.
At 1-year follow-up the patient had undergone 6 endoscopic dilations, with transient relief of symptoms. Surgical treatment with tracheal resection and reconstruction was planned. The preoperative workup included ambulatory esophageal three-site pH monitoring, which revealed pathologic levels of gastroesophageal reflux during horizontal decubitus and supraesophagic reflux.
Clinical treatment of GERD was initiated and included life-style modifications, prokinetic agents (30 mg/d of bromopride), and proton pump inhibitors (40 mg/d of omeprazole). We opted to determine the outcome with GERD treatment before performing tracheal reconstruction. After 3 months the esophageal pH monitoring was repeated and the pathologic alterations were no longer detected (Table 1). Clinical treatment of GERD resulted in resolution of symptoms, and the patient remained asymptomatic at 2-year follow-up. A computed tomography scan revealed a stable tracheal lumen 10 mm in diameter. During this period, no additional airway procedures were necessary.
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Comment
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Idiopathic laryngotracheal stenosis includes lesions that share typical features of location, configuration, clinical evolution, and pathologic findings. This patient had some of these features, as well as female sex, fifth decade of life, and upper tracheal stenosis, with no history of intubation, trauma, infection, or collagen vascular disease (eg, Wegener granulomatosis or relapsing polychondritis). Therefore, a diagnosis of ILTS was made [1]. Because of good results of surgical treatment in patients with similar findings, as shown in some series [4, 5], we considered our patient a candidate for surgery.
Many authors have implicated GERD as the cause of ILTS. Maronian and colleagues [2], using three- to four-port pH probes, recorded pH less than 4.0 in 5 of 7 patients with isolated idiopathic subglottic stenosis. Toohill and colleagues [3], using ambulatory 24-hour pH monitoring, studied 12 patients with laryngotracheal stenosis and 34 healthy volunteers and found that the incidence of pharyngeal acid reflux events in the patient group was higher than in the control group. Neither in these series nor in the literature have we found information about GERD treatment in this subset of patients. No previous report has addressed the relationship between GERD management and symptomatic improvement in patients with ILTS.
Our patient had no typical symptoms of GERD; however, 24-hour esophageal pH monitoring demonstrated pathologic reflux, as frequently observed in other extraesophageal GERD manifestations (eg, asthma and sinusitis) [6]. In our patient a successful outcome was obtained, and GERD clinical treatment resulted in regression of tracheal stenosis, demonstrated by absence of symptoms at 2-year follow-up, and, more important, no need for further endoscopic dilation. The suggestion that GERD is an etiologic factor for ILTS remains controversial. In our patient a close relationship was observed between anti-GERD treatment and resolution of symptoms.
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References
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- Grillo HC, Mark EJ, Mathisen DJ, Wain JC. Idiopathic laryngotracheal stenosis and its management Ann Thorac Surg 1993;56:80-87.[Abstract]
- Maronian NC, Azadeh H, Waugh P, Hillel A. Association of laryngopharyngeal reflux disease and subglottic stenosis Ann Otol Rhinol Laryngol 2001;110:606-612.[Medline]
- Toohill RJ, Ulualp SO, Shaker R. Evaluation of gastroesophageal reflux in patients with laryngotracheal stenosis Ann Otol Rhinol Laryngol 1998;107:1010-1014.[Medline]
- Ashiku SK, Kuzucu A, Grillo HC, et al. Idiopathic laryngotracheal stenosis: effective definitive treatment with laryngotracheal resection J Thorac Cardiovasc Surg 2004;127:99-107.[Abstract/Free Full Text]
- Loutsidis A, Zisis C, Lariou K, Bellenis I. Surgical management of idiopathic subglottic tracheal stenosis Eur J Cardiothorac Surg 2000;17:488-491.[Abstract/Free Full Text]
- Wong WN, Fass R. Extraesophageal and atypical manifestations of GERD J Gastroenterol Hepatol 2004;19(suppl 3):S33-S43.[Medline]
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Abstract
Introduction
