CRA42 COBBLESTONE AIRWAYS: SERIES OF RECOGNIZING THE RARE TRACHEOBRONCHOPATHIA OSTEOCHONDROPLASTICA

Nor Syamimi Mohd Isa, Lalitha Pereirasamy.
Hospital Pulau Pinang, Pulau Pinang, Malaysia

INTRODUCTION

Tracheobronchopathia osteochondroplastica (TO) is a rare, benign condition characterised by presence of numerous submucosal chondro-osseous nodules on the anterolateral wall of distal trachea and main bronchi. Most TOs are either asymptomatic or had non-specific symptoms, leading to misdiagnoses and its under-recognition. We described our experience with 2 cases of this rare entity in the past 3 years.

CASE 1

A 52 years-old non-smoker man was referred to Respiratory clinic for recalcitrant cough with CT Thorax showing right middle-lobe sub-segmental collapse. He was subjected to bronchoscopy which revealed luminal narrowing of trachea and main bronchi due to multiple tiny cobblestone-like nodules. Histopathological biopsy identified benign respiratory epithelium with squamous metaplastic changes alongside sub-epithelial nodules of osseous element. Biopsy was consistent with TO and his lavage grew Pseudomonas Aerigunosa. He was initiated on course of antibiotic and improved with symptomatic cough management. 

CASE 2 

A 58 years-old ex-smoker man was reviewed for incidental Right upper-lobe nodule. Further questioning revealed 3-months history of chronic cough without constitutional symptoms. He underwent CT-guided biopsy which showed granulomatous inflammation and empirical anti-TB was started. Patient’s cough however didn’t improved, and he was troubled by multiple cutaneous adverse reactions to anti-TB. Re-investigation with a bronchoscopy revealed multiple calcified nodules involving distal trachea and bilateral main bronchi. Similarly, biopsy showed benign osseo-cartilagenous lesion suggestive of TO. Patient’s anti-TB was ceased and he was prescribed with inhaled bronchodilators and symptomatic treatment.

DISCUSSION

There is no recognized guidelines highlighting diagnostic and treatment approach for TO. In our experience, diagnoses were incidental through bronchoscopy and clinched histopathologically. Moreover, it is difficult to deduce if chronic infections e.g. tuberculosis caused TO or vice versa. Plausibly, better understanding of TO will improve its recognition, diagnostic process and prevent long-term sequelae.