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 wit...

Full description

Bibliographic Details
Main Authors: Ádám Bach, Andrea Ambrus, Béla Iványi, Zoltán Tóbiás, Gholam Hossein Alim Marvasti, László Rovó
Format: Article
Language:English
Published: Mashhad University of Medical Sciences 2021-03-01
Series:Iranian Journal of Otorhinolaryngology
Subjects:
Online Access:https://ijorl.mums.ac.ir/article_17598_d9d57ad753907c16c464f5daa1c0b375.pdf
_version_ 1818366404833837056
author Ádám Bach
Andrea Ambrus
Béla Iványi
Zoltán Tóbiás
Gholam Hossein Alim Marvasti
László Rovó
author_facet Ádám Bach
Andrea Ambrus
Béla Iványi
Zoltán Tóbiás
Gholam Hossein Alim Marvasti
László Rovó
author_sort Ádám Bach
collection DOAJ
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.
first_indexed 2024-12-13T22:35:38Z
format Article
id doaj.art-8157ac80dd774865a739a73f27eacdb7
institution Directory Open Access Journal
issn 2251-7251
2251-726X
language English
last_indexed 2024-12-13T22:35:38Z
publishDate 2021-03-01
publisher Mashhad University of Medical Sciences
record_format Article
series Iranian Journal of Otorhinolaryngology
spelling doaj.art-8157ac80dd774865a739a73f27eacdb72022-12-21T23:28:59ZengMashhad University of Medical SciencesIranian Journal of Otorhinolaryngology2251-72512251-726X2021-03-0133211912510.22038/ijorl.2020.47106.254717598IgG4-Related Laryngeal Disease as a Possible Cause of Idiopathic Subglottic Stenosis: A Case ReportÁdám Bach0Andrea Ambrus1Béla Iványi2Zoltán Tóbiás3Gholam Hossein Alim Marvasti4László Rovó5Department of Otorhinolaryngology & Head- Neck Surgery, University of Szeged; Tisza Lajos krt. 111., 6725, Szeged, Hungary.Department of Otorhinolaryngology & Head- Neck Surgery, University of Szeged; Tisza Lajos krt. 111., 6725, Szeged, Hungary.Department of Pathology, University of Szeged, Állomás u. 2, 6725, Szeged, Hungary.Department of Otorhinolaryngology & Head- Neck Surgery, University of Szeged; Tisza Lajos krt. 111., 6725, Szeged, Hungary.Department of Otorhinolaryngology & Head- Neck Surgery, University of Szeged; Tisza Lajos krt. 111., 6725, Szeged, Hungary.Department of Otorhinolaryngology & Head- Neck Surgery, University of Szeged; Tisza Lajos krt. 111., 6725, Szeged, Hungary.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.https://ijorl.mums.ac.ir/article_17598_d9d57ad753907c16c464f5daa1c0b375.pdffibro-inflammatory disorderidiopathic subglottic stenosisigg4laryngotracheoplasty
spellingShingle Ádám Bach
Andrea Ambrus
Béla Iványi
Zoltán Tóbiás
Gholam Hossein Alim Marvasti
László Rovó
IgG4-Related Laryngeal Disease as a Possible Cause of Idiopathic Subglottic Stenosis: A Case Report
Iranian Journal of Otorhinolaryngology
fibro-inflammatory disorder
idiopathic subglottic stenosis
igg4
laryngotracheoplasty
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 fibro-inflammatory disorder
idiopathic subglottic stenosis
igg4
laryngotracheoplasty
url https://ijorl.mums.ac.ir/article_17598_d9d57ad753907c16c464f5daa1c0b375.pdf
work_keys_str_mv AT adambach igg4relatedlaryngealdiseaseasapossiblecauseofidiopathicsubglotticstenosisacasereport
AT andreaambrus igg4relatedlaryngealdiseaseasapossiblecauseofidiopathicsubglotticstenosisacasereport
AT belaivanyi igg4relatedlaryngealdiseaseasapossiblecauseofidiopathicsubglotticstenosisacasereport
AT zoltantobias igg4relatedlaryngealdiseaseasapossiblecauseofidiopathicsubglotticstenosisacasereport
AT gholamhosseinalimmarvasti igg4relatedlaryngealdiseaseasapossiblecauseofidiopathicsubglotticstenosisacasereport
AT laszlorovo igg4relatedlaryngealdiseaseasapossiblecauseofidiopathicsubglotticstenosisacasereport