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28 January 2015 : Case report  USA

A Complication of Tracheobronchopathia Osteochondroplastica Presenting as Acute Hypercapnic Respiratory Failure

Unusual clinical course, Mistake in diagnosis, Management of emergency care, Educational Purpose (only if useful for a systematic review or synthesis)

Mauricio DanckersABCEF, Roy A. RaadBDE, Ronaldo ZamucoBCD, Aron PollackBCDEF, Scott RickertBDF, Caralee Caplan-ShawABCDEF

DOI: 10.12659/AJCR.892427

Am J Case Rep 2015; 16:45-49

Abstract

BACKGROUND: Tracheobronchopathia osteochondroplastica is a rare benign and often indolent disease. We report the first case of tracheobronchopathia osteochondroplastica (TBO) presenting as acute hypercarbic respiratory failure due to superimposed subglottic submucosal abscess.

CASE REPORT: A 27-year-old man presented to the emergency department in respiratory distress that required mechanical ventilation for acute hypercarbic respiratory failure. Upon extubation the next day, stridor was elicited with ambulation. Spirometry revealed fixed upper airway obstruction. Neck imaging showed a 2.8×2.0×4.0 cm partially calcified subglottic mass with cystic and solid component obstructing 75% of the airway. Surgical exploration revealed purulent drainage upon elevation of the thyroid isthmus and an anterolateral cricoid wall defect in communication with a subglottic submucosal cavity. Microbiology was negative for bacteria or fungi. Pathology showed chondro-osseous metaplasia compatible with tracheobronchopathia osteochondroplastica (TBO). The patient received a course of antibiotics and prophylactic tracheostomy. Since tracheostomy removal 3 days later, the patient remains asymptomatic.

CONCLUSIONS: Tracheobronchopathia osteochondroplastica is a rare disease with usually benign clinical course and incidental diagnosis. It may present as acute hypercarbic respiratory failure when subglottic infection is superimposed.

Keywords: Diagnosis, Differential, Hypercapnia - etiology, Laryngoscopy, Osteochondrodysplasias - diagnosis, Respiratory Insufficiency - etiology, Tomography, X-Ray Computed, Tracheal Diseases - diagnosis

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American Journal of Case Reports eISSN: 1941-5923
American Journal of Case Reports eISSN: 1941-5923