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CASE REPORT
Chronic non-granulomatous supraglottitis of a male adolescent and its successful management with azathioprine
  1. Aaron Kovacs,
  2. Shankar Haran and
  3. Paul Paddle
  1. Department of Ear, Nose and Throat Surgery, Monash Health, Clayton, Victoria, Australia
  1. Correspondence to Mr Paul Paddle, Paul.Paddle{at}monashhealth.org

Abstract

Chronic non-granulomatous supraglottitis (CNGS) is a rare disorder of the supraglottic larynx, characterised by chronic supraglottic inflammation in the absence of granulomata, vasculitis, neoplasia, autoimmune disease or infective changes on histology. We present the case of a male adolescentwho attended with progressively worsening exertional dyspnoea, stridor and symptoms of obstructive sleep apnoea. Flexible nasendoscopy revealed marked supraglottic subepithelial thickening sparing the glottis and subglottis, confirmed on microlaryngoscopy. MRI of the head and neck demonstrated diffuse, homogenous supraglottic oedema. At the peak of his symptomology, the patient was admitted for further investigations and intravenous steroid therapy, and switched to prolonged oral steroids on discharge. Tracheostomy was avoided. After 3 months, he was successfully weaned from steroids to azathioprine with gradual symptomatic improvement. This case represents the first successful use of a steroid-sparing agent in the management of CNGS.

  • ear, nose and throat
  • otolaryngology / ENT

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Footnotes

  • Contributors AK is the primary author of the manuscript. SH was the primary author of the conference presentation on which the manuscript was built, and has contributed to this manuscript. PP is the research supervisor and treating surgeon of the case’s subject.

  • Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.

  • Competing interests None declared.

  • Provenance and peer review Not commissioned; externally peer reviewed.

  • Patient consent for publication Parental/guardian consent obtained.