A case of subglottic and diffuse tracheal stenoses appearing responsive to macrolide therapy

We present an atypical case of subglottic stenosis with diffuse tracheal stenoses in a child responsive only to steroid and azithromycin (AZI) therapy. A 12-year-old boy presented with acute biphasic stridor on the background of an 18-month history of progressive shortness of breath, decreased exercise tolerance and snoring. Subsequent laryngoscopy and bronchoscopy revealed granulation tissue in the subglottic area, two circumferential stenoses of the trachea and a number of fibrous bands at the carina and at the aperture if the right main bronchus were seen. A battery of serological and histological investigations did not reveal a specific aetiology. In the acute phase this patient only responded to steroid therapy. In the medium term, repeat laryngoscopies were performed with sharp division of stenotic bands and balloon dilatation. The patient's condition was unresponsive to non-steroidal anti-inflammatories, multiple first-line antibiotics, and surgical treatment of the tracheal lesions. However definitive treatment was found with the macrolide antibiotic AZI used for its anti-inflammatory properties. This highly unusual case of diffuse tracheal stenoses in a child proved to be a management challenge. Definitive treatment was found with the use of AZI. From our literature search this appears to be the first reported case of AZI successfully treating subglottic and tracheal stenoses.