Ann Thorac Surg 2006;81:1480-1482
© 2006 The Society of Thoracic Surgeons
Case report
Tracheobronchopathia Osteoplastica: Cause of Difficult Tracheal Intubation
Ahmad Tadjeddein, MD,
Zhamak Khorgami, MD,
Hamed Akhlaghi, MD*
Thoracic Surgery Ward, Shariati Hospital, Tehran University of Medical Sciences, Tehran, Iran
Accepted for publication April 5, 2005.
* Address correspondence to Dr Akhlaghi, Thoracic Surgery Ward, Shariaty Hospital, No. 45 Sahar Ln, Shirkhorshidi Ln, Naderi Shomali St, Qazvin, 34137-77578 Tehran, Iran (Email: hamed.akhlaghi{at}gmail.com).
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Abstract
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Tracheobronchopathia osteochondroplastica is a rare benign disorder affecting the trachea and the bronchi. We report a case presenting as difficulty during tracheal intubation. Chest roentgenogram revealed nothing before surgery. Findings on computed tomographic imaging scans show calcified nodular densities protruding into the tracheal lumen, with an abnormally irregular tracheal morphology and decreased lateral diameter. The fiber optic bronchoscopy, which was unable to pass more than 4 to 5 cm, showed enormous prominent protrusion with significant narrowing of the tracheal lumen. The diagnosis was confirmed by virtual bronchoscopy, which showed a tracheal narrowing with a beaded appearance and an irregular border that extended into the trachea and main bronchi.
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Introduction
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Tracheobronchopathia osteochondroplastica (TO) known as tracheopathia osteoplastica is a rare benign disorder characterized by multiple cartilaginous or osseous submucosal nodules that project into the tracheobronchial lumen [1]. Patients usually present with cough, hemoptysis, and recurrent respiratory infection [2, 3]. Because this disorder is usually benign without any specific symptoms, most of the cases in the past were detected incidentally on autopsy, but presently it is more frequently diagnosed during bronchoscopy. The diagnosis of TO is very rarely made during a difficult intubation [4].
A 39-year-old woman was referred to the thoracic surgery ward at Shariati Hospital because intubation had been impossible when she was to undergo surgery. The patient was scheduled for myringoplasty due to chronic suppurative otitis media with general anesthesia. On the day of surgery, after induction of anesthesia, an anesthesiologist was unable to pass even a 5-mm endotracheal tube through the trachea. The induction was reversed and the surgery was cancelled. In past medical history, the patient had no respiratory problem, expect asthmatic type attacks after heavy exercise and during pregnancy with no medical evaluation. There was no history of disphagia, aspiration, or regurgitation.
The patient was referred for computed tomographic imaging and fiber optic bronchoscopy. Computed tomographic image findings were asymmetric airways, calcified nodular densities protruding into the tracheal lumen, abnormally irregular tracheal morphology, and decreased lateral diameter with no occupied lesion (Fig 1). The bronchoscopy report noted that visualizing more than 4 to 5 cm of trachea was impossible due to enormous prominent calcified protrusions with significant narrowing of the tracheal lumen. The larynx and vocal cords were normal.
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Fig 1. Computed tomographic image showing calcified nodular densities protruding into the tracheal lumen.
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The virtual bronchoscopy was performed, which showed a beaded appearance and irregular border that extended from the larynx into the trachea and main bronchi, and segmental bronchi was seen (Figs 2–4).
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Fig 2. Cronal section of helical computed tomographic image showing irregular border of trachea and main bronchi.
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Fig 3. Reconstructed helical computed tomographic images showing the three-dimensional state of the trachea with an irregular border that extended into the main and segmental bronchi.
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Tracheobronchopathia osteochondroplastica was diagnosed radiologically and the patient was discharged with the suggestion to use the epiglottic mask intubation for further operation.
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Comment
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Tracheobronchopathia osteochondroplastica is a rare, benign condition and is characterized by the presence of bony and cartilaginous nodules in the tracheal and bronchial mucosa [1]. This was first described by Wilks (a physician at Guy’s Hospital in 1875) when he found at autopsy that the larynx, trachea, and bronchi of a 38-year-old man (who had died of tuberculosis) was covered with a number of bony plates. He also noted that these bony deposits were predominately anterior to the trachea and lay between the cartilaginous rings [4, 5].
The cause of the condition is unknown, but several theories have been postulated. Ecchondroses and endocondroses from the tracheal rings were first suggested by Virchow in 1863. In 1910, Aschoff suggested that metaplasia of the elastic tissue may be the cause [4, 5]. More recently, Sakula theorized that tracheobronchopathia might be a form of primary localized amyloidosis of the lower respiratory tract that has undergone ossification. However, histological findings do not support this suggestion [6]. A possible association with atrophic rhinitis and pharyngitis was found by Harma and Suurkari, who suggested that the lesions were due to a build-up of calcium salts within the tracheal mucosa that led to the development of local bony nodules [4].
Most patients are asymptomatic and symptoms may include dyspnea, cough, hemoptysis, hoarseness, and wheezing [2, 4, 5]. The chest roentgenogram is normal most of the time [1, 5]; however irregularity and thickening of the trachea or a lobar collapse may be seen. Hodges and Israel reported two cases of TO presenting as a right middle lobe collapse [7]. At times, calcification of the tracheobronchial tree may be evident, especially in a lateral view. Computed tomography of the chest may show calcified nodules protruding into the tracheal lumen from the anterior and lateral walls of the trachea, sparing the posterior membranous portion, and when present, this finding is considered diagnostic of TO [1, 2, 8].
Computed tomography is an important imaging modality in the diagnosis of TO. It can reveal multiple nodular submucosal irregularities, some of which are calcified, with sparing of the posterior tracheal wall [3, 6–9].
The diagnosis of TO is made on the basis of gross appearance of the lesions during bronchoscopy. The presence of sessile cartilaginous or bony nodules, or both, with normal overlying mucosa producing a beaded appearance of the tracheobronchial tree constitutes the classical bronchoscopic finding [5]. Endoscopic features are typical and pathognomonic. The lesions are found most frequently in the distal two thirds of the trachea and major bronchi, but are also detected in the proximal trachea, the lobar, and the segmental bronchi, such as in our case in which the lesions were present up to the segmental bronchi [1, 2].
We found only three cases that have described tracheobronchopathia osteoplastica as the cause for an unexpected difficult intubation due to subglottic obstruction. In two of them, as in our case, the disease was extensive and caused significant problems with intubation [4].
The prognosis of TO is generally good and depends on the extent and location of the lesions. In cases of advanced lesions, the airway obstruction may produce obstructive pneumonitis and atelectasis. Treatment is usually conservative and consists of antibiotics for recurrent respiratory infections. Surgical treatment is necessary when conservative management fails [1, 3, 5].
There is no specific therapy for this entity. Treatment options have varied from a conservative management, laser resection, and radiation therapy to a more aggressive surgical repair or placement of stents to maintain the tracheal lumen. The treatment must be tailored to the patient’s needs [1, 2, 4, 6].
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References
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Abstract
Introduction
