Tracheobronchopathia osteochondroplastica: two cases and a review of the literature.

INTRODUCTION

Tracheobronchopathia osteochondroplastica (TO) is a rare disorder involving the lumen of the trachea-bronchial tree and characterized by multiple sub-mucosal osseous and cartilaginous nodules in the trachea and bronchus, sparing the posterior wall. We here report two cases of patients with tracheobronchopathia osteochondroplastica and review the relevant literature briefly.

CASE PRESENTATION

Case 1 was a 64-year-old woman with a history of Chronic Obstructive Pulmonary Disease (COPD) who presented with frequent non-productive cough for 2 years. Chest computed tomography (CT) showed signs consistent with COPD and evident irregular narrowing of the tracheal and both main bronchial lumen caused by calcific foci. Fibre optic bronchoscope (FOB) was performed and showed dozens of sub-mucosal nodules protruding into the lumen of lower half of the trachea and both main bronchi. Histopathological exam demonstrated sub-mucosal ossification and cartilage in the sample. Her follow-up has been uneventful for 3 years. Case 2 was a 37-year-old man presented with hoarseness, exertional dyspnea, and intermittent dry cough for about 3 years. Chest CT scans showed irregular nodules around the entire circumference of the trachea extending from sub-glottic region to lower trachea. FOB showed glottic stenosis and diffused sub-mucosal calcified nodules protruding from the antero-lateral portion of the trachea in the subglottic region. Over the following 12 months, his disease is stable.

CONCLUSIONS

TO is a rare, benign disease with slow progression, clinicians should be aware of TO and should consider it in patients with chronic cough, recurrent respiratory infection and evolving exertional dyspnea.