Primary Immunoglobulin G4-Related Laryngeal Disease: A Case Series and Review of Literature

Clinical Medicine Insights. Case Reports
Elizabeth F MaughanChadwan Al Yaghchi

Abstract

Immunoglobulin G4-related disease (IgG4-RD) is an increasingly recognised cause of various systemic fibro-inflammatory conditions. However, laryngeal involvement as a primary feature is extremely rare. We aimed to report on a case series of such patients and examine the global literature relating to laryngeal involvement. Having previously reported a case of IgG4-RD laryngeal pseudotumour, we describe a case series of further 4 patients with primary laryngeal IgG4-RD managed by our UK quaternary airway service and provide a brief overview of laryngeal IgG4-RD. Including our cases, 14 cases of primary laryngeal IgG4-RD have been reported. Vocal cord involvement is relatively uncommon. Repeat biopsies may be required to achieve histological diagnosis. Remission is achievable by commencement of immunomodulatory treatment, following which laryngeal reconstruction may be necessary. Laryngeal involvement is a rare presentation of IgG4-RD, itself a rare and difficult-to-diagnose condition. A high and prolonged index of suspicion is necessary from both surgical and pathological specialists for correct diagnosis and management.

References

Mar 17, 2010·Archives of Otolaryngology--head & Neck Surgery·Colin R ButlerGuri S Sandhu
Jun 4, 2010·The Journal of Laryngology and Otology·L MastersonE Rytina
Aug 25, 2010·Advances in Anatomic Pathology·Wah Cheuk, John K C Chan
Feb 10, 2012·The New England Journal of Medicine·John H StoneVikram Deshpande
May 19, 2012·Modern Pathology : an Official Journal of the United States and Canadian Academy of Pathology, Inc·Vikram DeshpandeJohn H Stone
Sep 19, 2012·Archives of Otolaryngology--head & Neck Surgery·Jagdeep Singh VirkGurpreet Sandhu
Nov 15, 2012·Radiographics : a Review Publication of the Radiological Society of North America, Inc·Akifumi FujitaHideharu Sugimoto
Sep 11, 2014·The Annals of Otology, Rhinology, and Laryngology·Lindsay RederPhillip Song
Dec 8, 2014·Lancet·Terumi KamisawaJohn H Stone
Mar 27, 2015·Arthritis & Rheumatology·A KhosroshahiUNKNOWN Second International Symposium on IgG4-Related Disease
May 20, 2015·Arthritis & Rheumatology·Zachary S WallaceJohn H Stone
Nov 26, 2016·Best Practice & Research. Clinical Rheumatology·Pilar Brito-ZerónJohn H Stone
Jan 18, 2017·JAMA Otolaryngology-- Head & Neck Surgery·Sergio S FerranteJames J Daniero
Feb 20, 2018·Arthritis Care & Research·Zachary S WallaceJohn H Stone
Dec 5, 2019·Annals of the Rheumatic Diseases·Zachary S WallaceUNKNOWN Members of the ACR/EULAR IgG4-RD Classification Criteria Working Group

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Methods Mentioned

BETA
biopsy
biopsies

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