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IgG4-Related Laryngeal Disease as a Possible Cause of Idiopathic Subglottic Stenosis: A Case Report

INTRODUCTION: Immunoglobulin G4-related disease (IgG4-RD) is a systemic fibro-inflammatory disorder. Laryngotracheal manifestation is very rare; therefore, it is usually associated with complex diagnostic and therapeutic problems. CASE REPORT: Herein, we report the case of a 35-year-old woman with i...

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Autores principales: Bach, Ádám, Ambrus, Andrea, Iványi, Béla, Tóbiás, Zoltán, Alim Marvasti, Gholam Hossein, Rovó, László
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Mashhad University of Medical Sciences 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8052487/
https://www.ncbi.nlm.nih.gov/pubmed/33912489
http://dx.doi.org/10.22038/ijorl.2020.47106.2547
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author Bach, Ádám
Ambrus, Andrea
Iványi, Béla
Tóbiás, Zoltán
Alim Marvasti, Gholam Hossein
Rovó, László
author_facet Bach, Ádám
Ambrus, Andrea
Iványi, Béla
Tóbiás, Zoltán
Alim Marvasti, Gholam Hossein
Rovó, László
author_sort Bach, Ádám
collection PubMed
description INTRODUCTION: Immunoglobulin G4-related disease (IgG4-RD) is a systemic fibro-inflammatory disorder. Laryngotracheal manifestation is very rare; therefore, it is usually associated with complex diagnostic and therapeutic problems. CASE REPORT: Herein, we report the case of a 35-year-old woman with idiopathic subglottic stenosis (ISGS) treated with one-step laryngotracheal reconstruction surgery. Postoperatively, the lesion was found to be a part of the IgG4-RD spectrum. Objective and subjective phoniatric tests, spirometry, and Quality of Life Questionnaire were used for the evaluation of postoperative functional results. Slide laryngotracheoplasty as a one-step surgery without stenting and tracheostomy ensured a sufficiently wide subglottic space with no adverse effect on voice quality. During a follow-up period of 22 months, endoscopy and computed tomography scan revealed no significant restenosis. The patient was able to return to premorbid activities of daily living without any further medical treatment. CONCLUSION: The laryngeal involvement of IgG4-RD is uncommon; however, it is a manifestation that should be included in the differential diagnosis of subglottic stenoses (SGS). Furthermore, subglottic IgG4-RD might be a potential etiological factor of ISGS and acquired airway stenosis after short-term intubation. Slide laryngotracheoplasty might be a favorable solution without stenting and tracheostomy even in special cases of SGS.
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spelling pubmed-80524872021-04-27 IgG4-Related Laryngeal Disease as a Possible Cause of Idiopathic Subglottic Stenosis: A Case Report Bach, Ádám Ambrus, Andrea Iványi, Béla Tóbiás, Zoltán Alim Marvasti, Gholam Hossein Rovó, László Iran J Otorhinolaryngol Case Report INTRODUCTION: Immunoglobulin G4-related disease (IgG4-RD) is a systemic fibro-inflammatory disorder. Laryngotracheal manifestation is very rare; therefore, it is usually associated with complex diagnostic and therapeutic problems. CASE REPORT: Herein, we report the case of a 35-year-old woman with idiopathic subglottic stenosis (ISGS) treated with one-step laryngotracheal reconstruction surgery. Postoperatively, the lesion was found to be a part of the IgG4-RD spectrum. Objective and subjective phoniatric tests, spirometry, and Quality of Life Questionnaire were used for the evaluation of postoperative functional results. Slide laryngotracheoplasty as a one-step surgery without stenting and tracheostomy ensured a sufficiently wide subglottic space with no adverse effect on voice quality. During a follow-up period of 22 months, endoscopy and computed tomography scan revealed no significant restenosis. The patient was able to return to premorbid activities of daily living without any further medical treatment. CONCLUSION: The laryngeal involvement of IgG4-RD is uncommon; however, it is a manifestation that should be included in the differential diagnosis of subglottic stenoses (SGS). Furthermore, subglottic IgG4-RD might be a potential etiological factor of ISGS and acquired airway stenosis after short-term intubation. Slide laryngotracheoplasty might be a favorable solution without stenting and tracheostomy even in special cases of SGS. Mashhad University of Medical Sciences 2021-03 /pmc/articles/PMC8052487/ /pubmed/33912489 http://dx.doi.org/10.22038/ijorl.2020.47106.2547 Text en https://creativecommons.org/licenses/by/3.0/This is an Open Access article distributed under the terms of the Creative Commons Attribution License, (http://creativecommons.org/licenses/by/3.0/ (https://creativecommons.org/licenses/by/3.0/) ) which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Case Report
Bach, Ádám
Ambrus, Andrea
Iványi, Béla
Tóbiás, Zoltán
Alim Marvasti, Gholam Hossein
Rovó, László
IgG4-Related Laryngeal Disease as a Possible Cause of Idiopathic Subglottic Stenosis: A Case Report
title IgG4-Related Laryngeal Disease as a Possible Cause of Idiopathic Subglottic Stenosis: A Case Report
title_full IgG4-Related Laryngeal Disease as a Possible Cause of Idiopathic Subglottic Stenosis: A Case Report
title_fullStr IgG4-Related Laryngeal Disease as a Possible Cause of Idiopathic Subglottic Stenosis: A Case Report
title_full_unstemmed IgG4-Related Laryngeal Disease as a Possible Cause of Idiopathic Subglottic Stenosis: A Case Report
title_short IgG4-Related Laryngeal Disease as a Possible Cause of Idiopathic Subglottic Stenosis: A Case Report
title_sort igg4-related laryngeal disease as a possible cause of idiopathic subglottic stenosis: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8052487/
https://www.ncbi.nlm.nih.gov/pubmed/33912489
http://dx.doi.org/10.22038/ijorl.2020.47106.2547
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