Cytotoxic Agents in the Treatment of Laryngeal Sarcoidosis: A Case Report and Review of the Literature

Agrawal, Yuri; Godin, David A.; Belafsky, Peter C.

doi:10.1016/j.jvoice.2005.10.010

Cited by 18 publications

(20 citation statements)

References 14 publications

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“…To our knowledge, this case represents the first use of azathioprine in the management of CNGS, though its successful use in chronic supraglottitis more generally is not without precedent; azathioprine has previously been used successfully in the management of laryngeal sarcoidosis. In one case, a 35-year-old man with laryngeal sarcoidosis had close to complete resolution of his supraglottic oedema at approximately 1 year of azathioprine therapy 14. We propose that azathioprine’s side effect profile, particularly in children and adolescents, is favourable to that of long-term glucocorticoid use and warrants consideration in the future management of CNGS.…”

Section: Discussionmentioning

confidence: 93%

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: 93%

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

2019

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“…Sarcoidosis is a multisystem granulomatous disorder of unknown aetiology 3. It is characterised by non-caseating granulomas in involved organs and tissues and remains a diagnosis of exclusion 3 4. It typically affects young adults and has an estimated prevalence of 10–35 cases per 100 000 population 3.…”

Section: Discussionmentioning

confidence: 99%

“…The disease is usually systemic, most commonly affecting the respiratory tract, however in 30%–60% of cases, patients are asymptomatic 3. Laryngeal involvement of sarcoid is rare, affecting only 0.5%–5% of those suffering with the disease 4 5. It typically presents with supraglottic involvement and appears as a pale pink, oedematous, diffusely hypertrophied epiglottis and associated supraglottic structures 4 6–8.…”

Section: Discussionmentioning

confidence: 99%

“…Laryngeal involvement of sarcoid is rare, affecting only 0.5%–5% of those suffering with the disease 4 5. It typically presents with supraglottic involvement and appears as a pale pink, oedematous, diffusely hypertrophied epiglottis and associated supraglottic structures 4 6–8. This can be very easily mistaken for infectious supraglottitis or many other causes for supraglottic inflammation.…”

Section: Discussionmentioning

confidence: 99%

“…This can be very easily mistaken for infectious supraglottitis or many other causes for supraglottic inflammation. Diagnosis is made by characteristic appearance of the larynx on endoscopic examination, histology of laryngeal biopsies showing non-caseating granulomatous inflammation and the exclusion of other granulomatous diseases (for example granulomatosis with polyangiitis or tuberculosis) 4 7…”

Section: Discussionmentioning

confidence: 99%

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Supraglottic laryngeal sarcoidosis masquerading as supraglottitis

et al. 2020

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Sarcoidosis is a rare multisystem disorder of unknown aetiology characterised by non-caseating granulomas in involved organs; it is a diagnosis of exclusion. Laryngeal involvement affects only 0.5%–5% of those with sarcoidosis. It is an uncommon but important cause of supraglottic inflammation and oedema and should be considered in the differential diagnosis in patients with supraglottitis. This case describes a 30-year-old man who presented with stridor and shortness of breath. Flexible nasendoscopic examination revealed a grossly oedematous, pale pink, diffusely hypertrophied epiglottis. Surgical biopsy revealed non-caseating granulomatous inflammation. In the context of exclusion of hepatitis, anti-neutrophil cytoplasmic antibody (ANCA) positivity, malignancy and mycobacterial infection, the diagnosis of supraglottic laryngeal sarcoid was made. He is being treated with azathioprine immunosuppression with symptomatic improvement.

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