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Allergy and Airway |

Tracheobronchopathia Osteochondroplastica: An Incidental Finding Associated With Right Middle Lobe Collapse

Kovi Levin, MBBS; Eli Dabscheck, MBBS
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Alfred Hospital, Melbourne, VIC, Australia


Chest. 2013;144(4_MeetingAbstracts):25A. doi:10.1378/chest.1702046
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Abstract

SESSION TITLE: Bronchology Case Report Posters II

SESSION TYPE: Affiliate Case Report Poster

PRESENTED ON: Tuesday, October 29, 2013 at 01:30 PM - 02:30 PM

INTRODUCTION: Tracheobronchopathia Osteochondroplastica (TO) is a rare non-neoplastic condition of the trachea, major airways, and occasionally larynx. It manifests as benign growths of osseous and cartilaginous nodules arising in the submucosa. Frequently asymptomatic, TO has been associated with numerous respiratory manifestations, including haemoptysis, dyspnoea, cough, lobar collapse, and lower respiratory infections.

CASE PRESENTATION: A 57 year old man was found incidentally to have right middle lobe (RML) bronchiectasis and partial collapse on computerized tomography (CT) following a motor vehicle accident. A chronic cough productive of discoloured sputum was reported. The patient emigrated from Turkey to Australia 24 years earlier. He worked as a metal welder and painter. His father had tuberculosis as a child. Comorbidities include a 10-pack year ex-smoking history and hypertension. He is a fit man who swims daily. While Quantiferon testing was positive, 3 sputum cultures for mycobacteria were negative. Spirometry and single breath gas transfer were normal. Bronchoscopy demonstrated multiple irregular nodules along the trachea and upper main bronchi. RML washings grew Pseudomonas aeruginosa and Klebsiella ozaena. Narrowing of the RML origin was not seen. Biopsies of a tracheal nodule demonstrated mucosa containing metaplastic bone and surface squamous metaplasia. Macroscopic and histological findings were consistent with TO. Antibiotics were prescribed. Four months later, repeat CT demonstrated improved RML changes, while repeat bronchoscopy one year later demonstrated stability of tracheal nodules.

DISCUSSION: Prior to CT and bronchoscopy, TO was diagnosed only on autopsy. Its true incidence remains unknown. The differential diagnoses include relapsing polychondritis, amyloidosis, Wegener's granulomatosis, endobronchial sarcoid, malignancy and tuberculosis. Biopsy for histopathological confirmation is therefore important. TO associated with lower respiratory infections has been well described. A previous case study described 2 patients with TO and RML collapse. In contrast to this case, both had RML origin narrowing.

CONCLUSIONS: Despite being a benign condition, TO is associated with a variety of chronic respiratory manifestations. Patients presenting in this manner warrant bronchoscopy as part of the diagnostic algorithm. Additionally, biopsy of endobronchial and endotracheal abnormalities for pathological diagnosis is vital.

Reference #1: Pounder DJ, Pieterse AS. Tracheopathia osteoplastica: report of four cases. Pathology. 1982 Oct;14(4):429-33

Reference #2: Lundgren R, Stjernberg NL. Tracheobronchopathia osteochondroplastica. A clinical bronchoscopic and spirometric study. Chest. 1981 Dec;80(6):706-9.

Reference #3: Hodges MK, Israel E. Tracheobronchopathia osteochondroplastica presenting as right middle lobe collapse. Diagnosis by bronchoscopy and computerized tomography. Chest. 1988 Oct;94(4):842-4.

DISCLOSURE: The following authors have nothing to disclose: Kovi Levin, Eli Dabscheck

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