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J Thorac Cardiovasc Surg 2006;132:714-716
© 2006 The American Association for Thoracic Surgery
Brief Communication |
a Department of Pulmonary, and Albert Einstein College of Medicine, Montefiore & Jacobi Medical Center, Bronx, NY
b Department of Pathology, and Albert Einstein College of Medicine, Montefiore & Jacobi Medical Center, Bronx, NY
c Department of Radiology, and Albert Einstein College of Medicine, Montefiore & Jacobi Medical Center, Bronx, NY
Received for publication March 29, 2006; revisions received April 27, 2006; accepted for publication May 19, 2006. * Address for reprints: A. M. Khan, MBBS, FRCS, MSc, 3600 Fieldston Rd 7C, Bronx, New York 10463 (Email: dramirkhan{at}hotmail.com).
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Tracheobronchopathia osteochondroplastica, or tracheopathia osteoplastica (TPO), is characterized by multiple cartilaginous or bony submucosal nodules that project into the tracheal and bronchial lumina and may or may not cause respiratory symptoms. The nodules always originate in airway cartilages and typically spare the posterior membranous wall.1
TPO was described by Wilks in the mid-1800s; by the 1970s more than 245 cases of TPO had been reported. TPO does not produce any characteristic symptoms or signs to indicate the diagnosis. The disease frequently manifests as chronic cough and wheezing, which frequently leads to the mistaken diagnosis of asthma or chronic bronchitis. Many patients remain free of symptoms. Larger nodules project into the airway lumen causing the commonly reported symptoms of chronic cough, hoarseness, dyspnea, hemoptysis, and recurrent respiratory infections. In rare instances, TPO may progress to tracheal stenosis with critical airway obstruction.
We herein describe a case of TPO complicated by tracheal stenosis, "TPO stenosis," necessitating surgical resection. To date only a handful of cases in which surgical intervention was required have been described in the literature.2-5
Clinical Summary
A 47-year-old woman with a medical history of asthma (mild, intermittent, occasional use of albuterol, one prior intubation) and alcoholic liver disease presented with progressive stridor of 3 months' duration. The patient's symptoms had progressively worsened over the past few months, and she had noted increasing difficulty in breathing for the past few days. There was no history of arthritis, arthralgias, nasal/sinus abnormalities, exposures, smoking, allergies, or any other surgical intervention. Her physical examination was unrevealing. Routine laboratory investigations were unremarkable, and a computed tomography (CT) scan of the neck was requested.
CT of the trachea revealed a stenosis of approximately 3 mm in diameter at the level of the mid trachea extending from the third to the fifth ring (see Figure 1). No surrounding enlarged mass was seen. Bronchoscopic examination confirmed the stenosis to be approximately 4 cm below the vocal cords. Surgical exploration through an extended collar incision was carried out.
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For the past 5 years the patient has been followed up with yearly CT of neck with no symptoms. She was initially (first 2 years) followed up with bronchoscopic examination and CT of the neck where a proximal stenosis at the site of the anastomosis, 1.2 x 1 cm, was noted after surgery (Figure 1). This has remained unchanged for the past 7 years and is the only other narrow part of the trachea, besides the original TPO stenosis. The original stenosis has not changed and has required no further surgical intervention. No recurrence of the disease has been seen in any other part of the tracheobronchial tree.
Histologic examination confirmed a 1 x 2 cm (breadth and length, respectively) tracheal segment. The proximal end had a lumen of 0.9 cm whereas the distal segment was 0.3 cm in diameter. Thickened wall was noticed (ranging from 0.5-1 cm) from proximal to distal (Figure 2). Microscopic examination showed squamous metaplasia of the bronchial epithelium. There were multiple submucosal nodules of mature and immature cartilage associated with osseous metaplasia (Figure 2) confirming the diagnosis of "tracheobronchopathia osteochondroplastica."
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An early bronchoscopic description of TPO was given by Killian in 1897. Von Schroetter reported the diagnosis in vivo by using a laryngeal mirror in 1896. Bronchoscopy firmly establishes its diagnosis. TPO typically involves the distal trachea and its bifurcation, but involvement of the proximal trachea, subglottis, and larynx has been described. Its characteristic appearance on the anterolateral surface of the trachea, with posterior sparing and hard gritty feel on bronchoscopy, establishes the diagnosis. The nodules may project into the airway lumen and lead to tracheal stenosis and airway obstruction.
A standard chest radiograph is usually of little help in identifying the etiology. Conventional CT of the trachea may reveal a "beaded" or scalloped appearance of the airways. However, CT scan of the trachea and 3-dimensional reconstruction is helpful in clarifying the anatomy and extent of the stenosis and thus is valuable in preoperative mapping for surgical intervention.
Treatment of TPO is symptomatic because it is usually not possible to remove all nodules or prevent their recurrence. Treatment of TPO stenosis with nonoperative modalities, for example, external beam radiation and bronchoscopic intervention (forceps removal, dilation, stent, laser ablation, and cryotherapy) has been attempted with very limited success.
The treatment of progressive TPO stenosis with critical airway obstruction mandates surgical excision, usually with an end-to-end anastomosis. On an average, approximately 5 cm of trachea can be removed; this can be increased depending on the peritracheal soft tissue. If a large segment has to be removed, resection is facilitated by release of the larynx, freeing up the trachea from surrounding structures in the mediastinum, and tracheoplasty. To date, very little literature is available to establish the best surgical approach and technique for TPO stenosis, because most of this is based on case reports.2-5
In summary, TPO is a benign disease, but critical airway obstruction resulting from TPO stenosis may prompt surgical intervention. The best surgical approach and techniques are yet to be established because little is known about the progression and prevention of the disease.
References
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