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Case report
Treatment-recalcitrant laryngeal sarcoidosis responsive to sirolimus
  1. Karen J Kelleher1,
  2. John Russell2,
  3. Orla G Killeen3,4 and
  4. Timothy Ronan Leahy1,5
  1. 1Paediatric ID and Immunology, CHI at Crumlin, Dublin, Ireland
  2. 2Paediatric Otorhinolaryngology, CHI at Crumlin, Dublin, Ireland
  3. 3Paediatric Rheumatology, CHI at Crumlin, Dublin, Ireland
  4. 4Department of Paediatrics, University College Dublin, Dublin, Ireland
  5. 5Department of Paediatrics, University of Dublin, Trinity College, Dublin, Ireland
  1. Correspondence to Dr Timothy Ronan Leahy; ronan.leahy{at}olchc.ie

Abstract

A 15-year-old girl presented with gradual-onset dysphonia and dysphagia. Laryngoscopy revealed significant supraglottic airway obstruction with swelling of both the epiglottis and arytenoids. After emergency tracheostomy, biopsy of the epiglottis revealed lymphoid hyperplasia with focal non-necrotising granulomata, leading to a presumed diagnosis of laryngeal sarcoidosis. Treatment with prednisolone and methotrexate produced minimal clinical improvement. A switch to sirolimus was followed by significant reduction in the laryngeal swelling, allowing decannulation of the tracheostomy. Treatment with sirolimus should be considered as a steroid sparing agent in laryngeal sarcoidosis, particularly in the presence of lymphoid hyperplasia on biopsy.

  • paediatrics
  • otolaryngology / ENT
  • rheumatology

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Footnotes

  • Contributors KJK and TRL collected the relevant data for the case report, prepared the manuscript and undertook relevant literature review. OGK reviewed and revised the manuscript. JR prepared laryngoscopic photographs for figure 1, and was also involved in review and revision of manuscript. TRL, OGK and JR were all involved in patient care.

  • 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.

  • Patient consent for publication Parental/guardian consent obtained.

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

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