Hydroxychloroquine as Treatment for Inflammatory Subglottic Stenosis: A Second Successful Case

Desuter, Gauthier; Grégoire, A; Gardiner, Quentin; Houssiau, Frédéric

doi:10.1155/2012/754308

Cited by 4 publications

(1 citation statement)

References 7 publications

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“…Unfortunately, this treatment was discontinued after the patient developed toxic epidermal necrolysis 15. However, hydroxychloroquine has been used successfully in the management of two previously steroid-dependent patients with inflammatory subglottic stenosis and later weaned without disease recurrence in both cases 16 17. The potential efficacy of other systemic therapies used in the management of chronic inflammatory diseases, such as biologics and intravenous immunoglobulin, is uncertain and requires further research.…”

Section: Discussionmentioning

confidence: 99%

Chronic non-granulomatous supraglottitis of a male adolescent and its successful management with azathioprine

2019

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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.

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Section: Discussionmentioning

confidence: 99%