Pulmonary Procedures |

An Unusual Case of Tracheobronchopathia Osteochondroplastica Presenting With Specific Antibody Deficiency FREE TO VIEW

Mohammad Khan, DO; Olena Lineberry, MD; Candace Huebert, MD; James Tracy, MD
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Pulmonary and Critical Care, University of Nebraska Medical Center, Omaha, NE

Chest. 2014;146(4_MeetingAbstracts):759A. doi:10.1378/chest.1988762
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SESSION TITLE: Bronchology/Interventional Procedures Case Report Posters

SESSION TYPE: Affiliate Case Report Poster

PRESENTED ON: Tuesday, October 28, 2014 at 01:30 PM - 02:30 PM

INTRODUCTION: Tracheobronchopathia Osteochondroplastica (TO) is a chronic, benign disease characterized by development of multiple osteocartilaginous nodules in the trachea and main bronchi. Patients usually present with cough, dyspnea, recurrent respiratory tract infections, and occasionally hemoptysis. While the incidence rates of TO remain uncertain, its occurrence is probably more common than currently recognized. We present a case of TO coexisting with specific antibody deficiency.

CASE PRESENTATION: A non-smoking, 45 year-old female, asthmatic was referred for evaluation of persistent dry cough and recurrent sinopulmonary infections for the past five years. The patient had been treated with inhaled corticosteroids, bronchodilators, antibiotics, antitussives and nasal decongestants without relief. More recently she was found to have specific antibody deficiency with only 40% response to pneumococcal antigen. Physical examination revealed focal right lower lobe wheezing. Serial chest roentgenograms and pulmonary function tests were normal. Computed tomography (CT) of the chest showed scattered areas of mucous impaction along the trachea and larger airways with mild bronchiectasis. Bronchoscopy revealed multiple cartilaginous nodules within the trachea, main and lobar bronchi consistent with TO on microscopy.

DISCUSSION: The etiology of TO remains elusive but associations with chronic infections, inflammation, amyloidosis, silicosis, and genetic predisposition have been hypothesized. CT is the imaging modality of choice while bronchoscopy is the most definitive diagnostic procedure. The hallmark of bronchoscopy is the presence of sessile submucosal nodules protruding into the tracheal and/or bronchial lumen while sparing the posterior tracheal wall. The mucosa can be described as having a ‘rock garden’ or ‘cobblestone’ appearance. Prognosis is good, but associated with degree of airway involvement and luminal narrowing.

CONCLUSIONS: This case highlights the significance of radiologic imaging and bronchoscopy in a patient with chronic cough unresponsive to usual empiric therapy. Multiple factors are involved in the pathogenesis of TO with specific antibody deficiency as an interesting association in this case. Familiarizing clinicians with this entity will result in increased recognition and appropriate treatment strategies.

Reference #1: Leske V, Lazor R, Coetmeur D, et al. Tracheobronchopathia osteochondroplastica: a study of 41 patients. Medicine 2001;80:378-90

DISCLOSURE: The following authors have nothing to disclose: Mohammad Khan, Olena Lineberry, Candace Huebert, James Tracy

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