Literature DB >> 32847880

Treatment-recalcitrant laryngeal sarcoidosis responsive to sirolimus.

Karen J Kelleher1, John Russell2, Orla G Killeen3,4, Timothy Ronan Leahy5,6.   

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. © BMJ Publishing Group Limited 2020. No commercial re-use. See rights and permissions. Published by BMJ.

Entities:  

Keywords:  otolaryngology / ENT; paediatrics; rheumatology

Mesh:

Substances:

Year:  2020        PMID: 32847880      PMCID: PMC7451481          DOI: 10.1136/bcr-2020-235372

Source DB:  PubMed          Journal:  BMJ Case Rep        ISSN: 1757-790X


  20 in total

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Journal:  Sarcoidosis Vasc Diffuse Lung Dis       Date:  2014-10-20       Impact factor: 0.670

Review 2.  Current and future immunosuppressive strategies in renal transplantation.

Authors:  Karen L Hardinger; Matthew J Koch; Daniel C Brennan
Journal:  Pharmacotherapy       Date:  2004-09       Impact factor: 4.705

3.  TORmented macrophages spontaneously form granulomas.

Authors:  Antonio J Pagán; Lalita Ramakrishnan
Journal:  Nat Immunol       Date:  2017-02-15       Impact factor: 25.606

Review 4.  Off-Label Use of Sirolimus and Everolimus in a Pediatric Center: A Case Series and Review of the Literature.

Authors:  M Bevacqua; F Baldo; S Pastore; E Valencic; Alberto Tommasini; A Maestro; M Rabusin; A Arbo; E Barbi
Journal:  Paediatr Drugs       Date:  2019-06       Impact factor: 3.022

5.  Rapamycin (AY-22,989), a new antifungal antibiotic. II. Fermentation, isolation and characterization.

Authors:  S N Sehgal; H Baker; C Vézina
Journal:  J Antibiot (Tokyo)       Date:  1975-10       Impact factor: 2.649

Review 6.  Sirolimus, a new, potent immunosuppressive agent.

Authors:  P A Kelly; S A Gruber; F Behbod; B D Kahan
Journal:  Pharmacotherapy       Date:  1997 Nov-Dec       Impact factor: 4.705

7.  Infliximab therapy in patients with chronic sarcoidosis and pulmonary involvement.

Authors:  Robert P Baughman; Marjolein Drent; Mani Kavuru; Marc A Judson; Ulrich Costabel; Roland du Bois; Carlo Albera; Martin Brutsche; Gerald Davis; James F Donohue; Joachim Müller-Quernheim; Rozsa Schlenker-Herceg; Susan Flavin; Kim Hung Lo; Barry Oemar; Elliot S Barnathan
Journal:  Am J Respir Crit Care Med       Date:  2006-07-13       Impact factor: 21.405

8.  Spontaneous improvement of laryngeal sarcoidosis resistant to systemic corticosteroid administration.

Authors:  Kazuya Tsubouchi; Naoki Hamada; Kayo Ijichi; Toshiro Umezaki; Koichi Takayama; Yoichi Nakanishi
Journal:  Respirol Case Rep       Date:  2015-08-13

9.  Upper Airway Obstruction Requiring Emergent Tracheostomy Secondary to Laryngeal Sarcoidosis: A Case Report.

Authors:  Changwan Ryu; Erica L Herzog; Hongyi Pan; Robert Homer; Mridu Gulati
Journal:  Am J Case Rep       Date:  2017-02-13

Review 10.  The Treatment of Activated PI3Kδ Syndrome.

Authors:  Tanya I Coulter; Andrew J Cant
Journal:  Front Immunol       Date:  2018-09-07       Impact factor: 7.561

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  2 in total

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Authors:  Julie H Wu; Sotonye Imadojemu; Avrom S Caplan
Journal:  Am J Clin Dermatol       Date:  2022-05-18       Impact factor: 6.233

2.  Uncommon presentation of isolated laryngeal sarcoidosis in a young man.

Authors:  Bayan O Besharah; Revan A Mujahed; Haddad H AlKaf; Sherif K Abdelmonim; Ali Aboloyoun Mohamed; Hasheema Alsulami; Mohammad A Al-Essa
Journal:  Ann Med Surg (Lond)       Date:  2022-07-31
  2 in total

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