Prevalence and incidence of idiopathic subglottic stenosis in southern and central Alberta: a retrospective cohort study

Abstract Background Subglottic stenosis (SGS) is a reportedly rare disease that causes recurrent severe airway obstruction. Etiologies reported for SGS include idiopathic, iatrogenic, autoimmune, congenital, and traumatic, with variable ratios among different centres. From empiric observation, south...

Full description

Bibliographic Details
Main Authors: Ryan K. Chan, Beau Ahrens, Paul MacEachern, J. Douglas Bosch, Derrick R. Randall
Format: Article
Language:English
Published: BMC 2021-11-01
Series:Journal of Otolaryngology - Head and Neck Surgery
Subjects:
Online Access:https://doi.org/10.1186/s40463-021-00544-8
_version_ 1797972391071055872
author Ryan K. Chan
Beau Ahrens
Paul MacEachern
J. Douglas Bosch
Derrick R. Randall
author_facet Ryan K. Chan
Beau Ahrens
Paul MacEachern
J. Douglas Bosch
Derrick R. Randall
author_sort Ryan K. Chan
collection DOAJ
description Abstract Background Subglottic stenosis (SGS) is a reportedly rare disease that causes recurrent severe airway obstruction. Etiologies reported for SGS include idiopathic, iatrogenic, autoimmune, congenital, and traumatic, with variable ratios among different centres. From empiric observation, southern and central Alberta was hypothesized to have a disproportionate distribution of SGS driven by increased idiopathic SGS (iSGS) compared to previous literature. Identification of causative agents of iSGS will help understand and guide future management options, so this study aimed to characterize the demographics of SGS subtypes, define prevalence and incidence rates of iSGS in southern Alberta, and geographically analyze for clustering of iSGS prevalence. Methods SGS patients from Alberta census divisions No. 1–9 and 15 were retrospectively reviewed. Patients were subtyped according to etiology of SGS and characterized. Idiopathic SGS prevalence and incidence was assessed; prevalence was further geographically segregated by census division and forward sortation area (FSA). Significant clustering patterns were assessed for using a Global Moran’s I analysis. Results From 2010 to 2019 we identified 250 SGS patients, who were substantially overrepresented by idiopathic patients (80.4%) compared to autoimmune (10.0%), iatrogenic (7.6%), congenital (1.2%), and traumatic (0.8%). The total iSGS prevalence was 9.28/100,000 with a mean annual incidence rate of 0.71/100,000 per year. Significant clustering was observed (Moran’s index 0.125; z-score 2.832; p = 0.0046) and the highest rates of prevalence were observed in southern Alberta and in rural communities heterogeneously dispersed around Calgary FSAs. Conclusion In southern and central Alberta, iSGS patients were disproportionately over-represented in contrast to other subtypes with the highest prevalence in southern Alberta. There was a three-fold higher annual incidence compared to previous literature demonstrating the highest rates of disease reported worldwide. Future research aims to expand the geographical scope and to assess for demographic or environmental differences within significant clusters that may contribute to disease pathophysiology. Level of evidence III. Graphical Abstract
first_indexed 2024-04-11T03:47:46Z
format Article
id doaj.art-95eed5de4e1b4646a9bf14d55315ad95
institution Directory Open Access Journal
issn 1916-0216
language English
last_indexed 2024-04-11T03:47:46Z
publishDate 2021-11-01
publisher BMC
record_format Article
series Journal of Otolaryngology - Head and Neck Surgery
spelling doaj.art-95eed5de4e1b4646a9bf14d55315ad952023-01-02T02:29:19ZengBMCJournal of Otolaryngology - Head and Neck Surgery1916-02162021-11-0150111110.1186/s40463-021-00544-8Prevalence and incidence of idiopathic subglottic stenosis in southern and central Alberta: a retrospective cohort studyRyan K. Chan0Beau Ahrens1Paul MacEachern2J. Douglas Bosch3Derrick R. Randall4Cumming School of Medicine, University of CalgaryInterdisciplinary PhD Program, Dalhousie UniversityInterventional Pulmonary Medicine, Division of Respirology – Thoracic Surgery and Medical Oncology, Cumming School of Medicine, University of CalgarySection of Otolaryngology – Head and Neck Surgery, Department of Surgery, Cumming School of Medicine, University of CalgarySection of Otolaryngology – Head and Neck Surgery, Department of Surgery, Cumming School of Medicine, University of CalgaryAbstract Background Subglottic stenosis (SGS) is a reportedly rare disease that causes recurrent severe airway obstruction. Etiologies reported for SGS include idiopathic, iatrogenic, autoimmune, congenital, and traumatic, with variable ratios among different centres. From empiric observation, southern and central Alberta was hypothesized to have a disproportionate distribution of SGS driven by increased idiopathic SGS (iSGS) compared to previous literature. Identification of causative agents of iSGS will help understand and guide future management options, so this study aimed to characterize the demographics of SGS subtypes, define prevalence and incidence rates of iSGS in southern Alberta, and geographically analyze for clustering of iSGS prevalence. Methods SGS patients from Alberta census divisions No. 1–9 and 15 were retrospectively reviewed. Patients were subtyped according to etiology of SGS and characterized. Idiopathic SGS prevalence and incidence was assessed; prevalence was further geographically segregated by census division and forward sortation area (FSA). Significant clustering patterns were assessed for using a Global Moran’s I analysis. Results From 2010 to 2019 we identified 250 SGS patients, who were substantially overrepresented by idiopathic patients (80.4%) compared to autoimmune (10.0%), iatrogenic (7.6%), congenital (1.2%), and traumatic (0.8%). The total iSGS prevalence was 9.28/100,000 with a mean annual incidence rate of 0.71/100,000 per year. Significant clustering was observed (Moran’s index 0.125; z-score 2.832; p = 0.0046) and the highest rates of prevalence were observed in southern Alberta and in rural communities heterogeneously dispersed around Calgary FSAs. Conclusion In southern and central Alberta, iSGS patients were disproportionately over-represented in contrast to other subtypes with the highest prevalence in southern Alberta. There was a three-fold higher annual incidence compared to previous literature demonstrating the highest rates of disease reported worldwide. Future research aims to expand the geographical scope and to assess for demographic or environmental differences within significant clusters that may contribute to disease pathophysiology. Level of evidence III. Graphical Abstracthttps://doi.org/10.1186/s40463-021-00544-8Subglottic stenosisIdiopathicAlberta
spellingShingle Ryan K. Chan
Beau Ahrens
Paul MacEachern
J. Douglas Bosch
Derrick R. Randall
Prevalence and incidence of idiopathic subglottic stenosis in southern and central Alberta: a retrospective cohort study
Journal of Otolaryngology - Head and Neck Surgery
Subglottic stenosis
Idiopathic
Alberta
title Prevalence and incidence of idiopathic subglottic stenosis in southern and central Alberta: a retrospective cohort study
title_full Prevalence and incidence of idiopathic subglottic stenosis in southern and central Alberta: a retrospective cohort study
title_fullStr Prevalence and incidence of idiopathic subglottic stenosis in southern and central Alberta: a retrospective cohort study
title_full_unstemmed Prevalence and incidence of idiopathic subglottic stenosis in southern and central Alberta: a retrospective cohort study
title_short Prevalence and incidence of idiopathic subglottic stenosis in southern and central Alberta: a retrospective cohort study
title_sort prevalence and incidence of idiopathic subglottic stenosis in southern and central alberta a retrospective cohort study
topic Subglottic stenosis
Idiopathic
Alberta
url https://doi.org/10.1186/s40463-021-00544-8
work_keys_str_mv AT ryankchan prevalenceandincidenceofidiopathicsubglotticstenosisinsouthernandcentralalbertaaretrospectivecohortstudy
AT beauahrens prevalenceandincidenceofidiopathicsubglotticstenosisinsouthernandcentralalbertaaretrospectivecohortstudy
AT paulmaceachern prevalenceandincidenceofidiopathicsubglotticstenosisinsouthernandcentralalbertaaretrospectivecohortstudy
AT jdouglasbosch prevalenceandincidenceofidiopathicsubglotticstenosisinsouthernandcentralalbertaaretrospectivecohortstudy
AT derrickrrandall prevalenceandincidenceofidiopathicsubglotticstenosisinsouthernandcentralalbertaaretrospectivecohortstudy