SESSION TYPE: Bronchology Cases
PRESENTED ON: Wednesday, October 24, 2012 at 11:15 AM - 12:30 PM
INTRODUCTION: Tracheobronchopathia Osteochondroplastica (TO) is a rare large airway disorder of unknown etiology characterized by formation of multiple submucosal osteo-cartilaginous nodules. Since the mid 19th century approximately 300 cases have been reported world-wide. It is usually found incidentally and is benign, but airway obstruction may cause respiratory symptoms. We present a case of an adult with HIV diagnosed with TO.
CASE PRESENTATION: A 57 year-old man with HIV (CD4> 500 and VL undetectable) on anti-retroviral therapy was found incidentally on chest CT to have nodular abnormalities in the large airways. He was asymptomatic and denied dyspnea, cough, hemoptysis, fevers, chills, and weight loss. He has no history of opportunistic infections, and is a non-smoker. On physical exam vital signs were stable. He had no lymphadenopathy, and clear breath sounds. Routine laboratory data was within normal limits. CXR was unremarkable. Chest CT revealed multiple small noncalcified nodular protrusions in the large airways, sparing the distal airways and lung parenchyma. On bronchoscopy numerous hard and glossy submucosal nodules were visualized in the antero-lateral aspect of large airways, sparing the posterior walls. Histology from bronchoscopic endobronchial biopsy and needle aspiration showed respiratory epitheilium. All culture data were negative.
DISCUSSION: The etiology of TO is unknown, and reported associations include familial, congenital, chronic inflammatory, and infectious origins. Our case is unique because of the association with HIV. This association may suggest that immunodeficiency plays a contributory role in development of TO. TO generally has a benign natural history. Airway obstruction may cause cough, dyspnea, hemoptysis, recurrent pulmonary infections, atelectasis, and bronchiectasis. The diagnosis of TO is made by characteristic imaging, bronchoscopic, and histologic findings. Characteristic histologic findings include submucosal cartilage and ossification; these were not found in our patient due to the technically challenging aspect of obtaining a needle biopsy from these hard nodules. Asymptomatic patients are managed conservatively, and symptomatic patients may benefit from bronchoscopic palliation.
CONCLUSIONS: Tracheobronchopathia osteochondroplastica is a rare large airways disorder of unknown cause and was diagnosed in a patient with HIV, suggesting a possible association with immunodeficiency.
1) Abu-Hijleh M, Lee D, Braman SS. Tracheobronchopathia osteochondroplastica: a rare large airway disorder. Lung. 2008 Nov-Dec;186(6):353-9
DISCLOSURE: The following authors have nothing to disclose: Karthik Jothianandan, Amy Chi
No Product/Research Disclosure InformationTufts Medical Center, Boston, MA