SESSION TITLE: Bronchology Global Case Reports
SESSION TYPE: Global Case Report
PRESENTED ON: Tuesday, October 29, 2013 at 01:30 PM - 02:30 PM
INTRODUCTION: Tracheobronchopathiaosteochondroplastica is a rare disorder of unknown cause affecting the large airways.This disease has been written in case reports. However, the association with active pulmonary tuberculosis has not been reported in recent literature.
CASE PRESENTATION: Patient is E.R. a 57 yo female hypertensive, non-smoker seen at the outpatient department for 4 weeks history of non-productive cough and hoarseness. There was note of fever, weightloss and anorexia. She sought consult and was treated as a case of pnuemonia and was given several courses of antibiotics with no relief of symptoms. 1week prior to admission, there was persistence of symptoms now associated with difficulty of breathing . She notes difficulty in sleeping described as choking sensation. Pertinent physical examination,revealed stable vital signs, afebrile, BMI 22 , 98% O2 saturation at room air, there was note of basal ronchi on auscultation. Laryngoscopy showed hyperemic vocal cords with a polypoid mass on the right vocal fold impression was Chronic laryngitis Cxray showed infiltrates on the left upper lobe suggestive of tuberculosis. Ct scan showed ground glass opacities in the superior segment of both lower lobes, small nodule posterior segment of right upper lobe, fibrosis left apical lobe and narrowing of the right main stem bronchus. There were no lymphadenopathies. Neck Ct scan showed no masses, but with irregular borders on the right bronchus, with collapse of the right upper lobe Patient underwent bronchosocpy which revealed edematous vocal cords, cobble stoning with hard protruding cartilaginous structures covered with cottony exudates seen from the trachea to the right main bronchus. Bronchial washing was positive for Kliebsiella Pnuemonia. Biopsy showed fragments of degenerated epithelilal cells, necrotic debris. Cartilage and fibrin material with acute and chronic inflammatory cells consistent with ulcer bed and necrotic debris. Special AFB stain is positive for numerous acid fast bacilli consistent with Tuberculosis. She was started on INH/ RIF/PZA/EMB. Patient still had difficulty of breathing and underwent bronchosopy to remove the remaining cartilaginous material which was done using rigid bronchoscopy. Histopathology confimed the diagnosis of Tracheobronchopathiaosteochondroplastica revealing cartilage with areas of ossification. There was note of improvement of symptoms and patient continued treatment for 6 months.
DISCUSSION: Tracheobronchopathiaosteochondroplastica is a rare disorder of unknown cause affecting the large airways of adults affecting both genders. It is described a multiple osseous and cartilaginous nodules in the submucosa of the trachea and main bronchi.Presentation of such entity ranges from an asymptomatic patient to cough, hemoptysis and recurrent respiratory infections. It may cause significant airway obstruction and difficulty of intubation.Kliebsiella is frequently isolated however there is no clear relationship. Theories of its etiology includes ecchondrosis, exositosis from tracheal rings or metaplasia of submucosal and connective tissue cartialage and/ or bone tissue .
CONCLUSIONS: Tracheobronchopathiaosteochondroplastica is a rare disorder described as multiple osseous and cartilaginous nodules in the submucosa of the trachea and main bronchi. The patient presented with recurrent respiratory infection and upper airway obstruction. Biopsy revealed cartilage with ossifiction and acid fast bacilli consisitent with tuberculosis. Rigid bronchoscopy was done to remove the obstruction. The association of these two disease has not been reported in recent literature.
Reference #1: Harikrishna Doshi .J Thorac Cardiovasc Surg 2005;130:901-902 Tracheobronchopathia osteochondroplastica presenting as an isolated nodule in the right upper lobe bronchus with upper lobe collapse
DISCLOSURE: The following authors have nothing to disclose: Angelene Taleon-Parazo
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