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  Table of Contents    
LETTER TO EDITOR  
Year : 2016  |  Volume : 59  |  Issue : 1  |  Page : 129-130
Tracheobronchopathia osteochondroplastica: An unusual presentation with near total occlusion of airways


1 Department of Pathology and Laboratory Medicine, Deen Dayal Upadhyay Hospital, New Delhi, India
2 Department of ENT, Deen Dayal Upadhyay Hospital, New Delhi, India

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Date of Web Publication27-Jan-2016
 

How to cite this article:
Sood N, Khandelia BK, Bugnait G. Tracheobronchopathia osteochondroplastica: An unusual presentation with near total occlusion of airways. Indian J Pathol Microbiol 2016;59:129-30

How to cite this URL:
Sood N, Khandelia BK, Bugnait G. Tracheobronchopathia osteochondroplastica: An unusual presentation with near total occlusion of airways. Indian J Pathol Microbiol [serial online] 2016 [cited 2019 Nov 22];59:129-30. Available from: http://www.ijpmonline.org/text.asp?2016/59/1/129/174882


Editor,

Tracheobronchopathia osteochondroplastica (TPO) is an uncommon, idiopathic benign disease of large airways characterized by the presence of multiple, sessile cartilaginous, and osseous submucosal nodules. It rarely presents as a single mass lesion arising from anterolateral wall projecting into the tracheobronchial lumen. TPO was the first reported by Wilks in 19 th century. It has a reported incidence of 0.01-4.2%. It is seen more commonly in males in the fourth to sixth decade. Clinical presentation varies from productive cough, hemoptysis, dyspnea, and wheeze, to obstructive respiratory symptoms caused by a single mass lesion. [1],[2],[3]

A 40-year-old man presented with difficult breathing and stridor for the past 6 months. There was no history of tuberculosis, asthma, chronic obstructive pulmonary disorder, or trauma. The patient had to be tracheostomized to relieve his symptoms. He was given similar palliative treatment 8 years back. Computed tomogram (CT) scan showed a well-defined heterogeneous lesion measuring 28 mm 27 mm 24 mm lifting the left vocal cord superiorly and causing occlusion of the subglottic air space with peripheral calcified rim and central coarse calcification [Figure 1]a and b. On fiberoptic bronchoscopy (FOB), a large fleshy intraluminal mass was visualized in the subglottic region with posterior attachment.
Figure 1: (a and b) Computed tomogram scan shows a mass lesion causing near total occlusion of the subglottic airspace (arrows). (c and d) Pseudostratified columnar to metaplastic stratified squamous epithelium with underlying central chondroid islands and peripheral osseous metaplasia. (c: ×100 H and E, d: ×400 H and E). (e) Excised specimen measuring (2.8 cm × 2.6 cm × 1 cm). (f) Postsurgery X - ray showing T-tube insertion

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Histopathological examination of the biopsy taken from the mass revealed pseudostratified columnar to metaplastic stratified squamous epithelial lining, underlying subepithelial islands of chondroid elements, central myxoid degeneration, and peripheral osseous metaplasia consistent with a diagnosis of TPO [Figure 1]c and d. The mass was excised followed by T-tube insertion. Histological features of this mass were similar to the biopsy [Figure 1]e and f.

The etiopathogenesis of TPO remains elusive. However, chronic infections, inflammation, trauma, amyloidosis, silicosis, ecchondrosis, and exostosis have been suggested as possible etiological factors. [4] TPO has been associated with thymoma, atrophic rhinitis, lymphoma, epidermal cysts, and myasthenia gravis. [1] No such associations were found in our patient.

TPO needs to be differentiated from calcified lesions secondary to tuberculosis, carcinoma, papilloma, fibroma, endobronchial sarcoidosis, polychondritis, and Wegener's granulomatosis of the proximal airways. [4] These were ruled out in this case on the basis of clinical history, physical examination, histopathological examination, and sputum microscopy and culture.

On FOB, it is commonly seen as multiple sessile submucosal nodules protruding into the lumen of the trachea and main bronchi, sparing the posterior wall. CT shows calcific lesions and biopsy shows the presence of submucosal osseous and cartilaginous metaplasia, and metaplastic squamous epithelium confirming the diagnosis. Unlike the reported incidence, it was a single mass arising from the posterior wall in this case. [1],[3],[5]

Treatment options of TPO range from medications for symptomatic relief to laser ablation, surgical resection and mechanical/laser debulking for larger obstructive lesions following urgent tracheostomy. [5] This patient was tracheostomized followed by mechanical debulking and T-tube insertion 1-month later. The patient is currently asymptomatic in a 6 months follow-up.

This case is being presented due to its rarity and unusual presentation as a single mass lesion arising from the posterior wall of the larynx causing near total obliteration of the tracheal lumen.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Tatar D, Senol G, Demir A, Polat G. Tracheobronchopathia osteochondroplastica: Four cases. Chin Med J (Engl) 2012;125:2942-4.  Back to cited text no. 1
    
2.
Raess PW, Cowan SW, Haas AR, Zhang PJ, Litzky LA, Miller WT Jr, et al. Tracheobronchopathia osteochondroplastica presenting as a single dominant tracheal mass. Ann Diagn Pathol 2011;15:431-5.  Back to cited text no. 2
    
3.
Prakash UB. Tracheobronchopathia osteochondroplastica. Semin Respir Crit Care Med 2002;23:167-75.  Back to cited text no. 3
    
4.
Thakur A, Yang T, Chen T, Rana N, Zhu B, Wei X, et al. Atypical presentation of tracheobronchopathia osteochondroplastica: Is chronic inflammation a perpetrator? Med Princ Pract 2013;22:503-5.  Back to cited text no. 4
    
5.
Zhang XB, Zeng HQ, Cai XY, Zhang YJ. Tracheobronchopathia osteochondroplastica: A case report and literature review. J Thorac Dis 2013;5:E182-4.  Back to cited text no. 5
    

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Correspondence Address:
Binit Kumar Khandelia
F-302, BPTP Freedom Park Life, Sector 57, Gurgaon - 122 003, Haryana
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.174882

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