Hostname: page-component-cd9895bd7-jkksz Total loading time: 0 Render date: 2024-12-22T01:17:53.388Z Has data issue: false hasContentIssue false
  • English
  • Français

Idiopathic subglottic stenosis – a familial association and review of practice

Published online by Cambridge University Press:  29 April 2022

J M Hintze Open the ORCID record for J M Hintze [Opens in a new window] ,
C W Fitzgerald ,
I O'Riordan ,
E Keane ,
J Kinsella  and
C Timon
J M Hintze*
Affiliation:
Department of Otolaryngology – Head and Neck Surgery, St James’ Hospital, Dublin, Ireland Trinity College Dublin, University of Dublin, Dublin, Ireland Royal College of Surgeons in Ireland, Dublin, Ireland
C W Fitzgerald
Affiliation:
Department of Otolaryngology – Head and Neck Surgery, St James’ Hospital, Dublin, Ireland Trinity College Dublin, University of Dublin, Dublin, Ireland Royal College of Surgeons in Ireland, Dublin, Ireland
I O'Riordan
Affiliation:
Department of Otolaryngology – Head and Neck Surgery, St James’ Hospital, Dublin, Ireland Trinity College Dublin, University of Dublin, Dublin, Ireland Royal College of Surgeons in Ireland, Dublin, Ireland
E Keane
Affiliation:
Department of Otolaryngology – Head and Neck Surgery, St James’ Hospital, Dublin, Ireland Trinity College Dublin, University of Dublin, Dublin, Ireland Royal College of Surgeons in Ireland, Dublin, Ireland
J Kinsella
Affiliation:
Department of Otolaryngology – Head and Neck Surgery, St James’ Hospital, Dublin, Ireland Trinity College Dublin, University of Dublin, Dublin, Ireland
C Timon
Affiliation:
Department of Otolaryngology – Head and Neck Surgery, St James’ Hospital, Dublin, Ireland Trinity College Dublin, University of Dublin, Dublin, Ireland
*
Author for correspondence: Dr Justin M Hintze, Department of Otolaryngology – Head and Neck Surgery, St James’ Hospital, Dublin, Ireland E-mail: [email protected]
Get access Rights & Permissions [Opens in a new window]

Abstract

Objective

Idiopathic subglottic stenosis describes subglottic stenosis where no inflammatory, traumatic, iatrogenic or other causative aetiology can be identified. The present study aimed to outline our institution's experience of patients diagnosed with idiopathic subglottic stenosis and describe a very rarely reported familial association.

Methods

A retrospective review was conducted of prospectively maintained medical records from 2011 to 2020. Patient clinical, radiological and intra-operative data were reviewed to assess for defined endpoints.

Results

Ten patients with idiopathic subglottic stenosis were identified in this series. One familial pairing was identified, with two sisters presenting with the condition. Successful treatment with carbon dioxide laser and dilatation was achieved in most cases.

Conclusion

Idiopathic subglottic stenosis represents a rare, clinically challenging pathology. Management with endoscopic laser and balloon dilatation is an effective treatment. This paper highlights a very rare familial association, and describes our experience in treating idiopathic subglottic stenosis.

Keywords

Tracheal StenosisAcquired Suglottic StenosisAirway ObstructionTracheaLaser Therapy
Type
Main Article
Information
The Journal of Laryngology & Otology , Volume 136 , Issue 12 , December 2022 , pp. 1275 - 1277
Copyright
Copyright © The Author(s), 2022. Published by Cambridge University Press on behalf of J.L.O. (1984) LIMITED

Access options

Get access to the full version of this content by using one of the access options below. (Log in options will check for institutional or personal access. Content may require purchase if you do not have access.)

Footnotes

*

Joint first authors

Dr J Hintze takes responsibility for the integrity of the content of the paper

References

Aarnaes, MT, Sandvik, L, Brondbo, K. Idiopathic subglottic stenosis: an epidemiological single-center study. Eur Arch Otorhinolaryngol 2017;274:2225–8CrossRefGoogle ScholarPubMed
Gelbard, A, Donovan, DT, Ongkasuwan, J, Nouraei, SA, Sandhu, G, Benninger, MS et al. Disease homogeneity and treatment heterogeneity in idiopathic subglottic stenosis. Laryngoscope 2016;126:1390–6CrossRefGoogle ScholarPubMed
Gelbard, A, Shyr, Y, Berry, L, Hillel, AT, Ekbom, DC, Edell, ES et al. Treatment options in idiopathic subglottic stenosis: protocol for a prospective international multicentre pragmatic trial. BMJ Open 2018;8:e022243Google ScholarPubMed
Roediger, FC, Orloff, LA, Courey, MS. Adult subglottic stenosis: management with laser incisions and mitomycin-C. Laryngoscope 2008;118:1542–6CrossRefGoogle ScholarPubMed
Drake, VE, Gelbard, A, Sobriera, N, Wohler, E, Berry, LL, Hussain, LL et al. Familial aggregation in idiopathic subglottic stenosis. Otolaryngol Head Neck Surg 2020;163:1011–17CrossRefGoogle ScholarPubMed
Dumoulin, E, Stather, DR, Gelfand, G, Maranda, B, Maceachern, P, Tremblay, A. Idiopathic subglottic stenosis: a familial predisposition. Ann Thorac Surg 2013;95:1084–6CrossRefGoogle ScholarPubMed
Clement, P, Hans, S, de Mones, E, Sigston, E, Laccourreye, O, Brasnu, D. Dilatation for assisted ventilation-induced laryngotracheal stenosis. Laryngoscope 2005;115:1595–8CrossRefGoogle ScholarPubMed
Herrington, HC, Weber, SM, Andersen, PE. Modern management of laryngotracheal stenosis. Laryngoscope 2006;116:1553–7CrossRefGoogle ScholarPubMed
Shapshay, SM, Beamis, JF Jr, Hybels, RL, Bohigian, RK. Endoscopic treatment of subglottic and tracheal stenosis by radial laser incision and dilation. Ann Otol Rhinol Laryngol 1987;96:661–4CrossRefGoogle ScholarPubMed
Feinstein, AJ, Goel, A, Raghavan, G, Long, J, Chhetri, DK, Berke, GS et al. Endoscopic management of subglottic stenosis. JAMA Otolaryngol Head Neck Surg 2017;143:500–5CrossRefGoogle ScholarPubMed
Maronian, NC, Azadeh, H, Waugh, P, Hillel, A. Association of laryngopharyngeal reflux disease and subglottic stenosis. Ann Otol Rhinol Laryngol 2001;110:606–12CrossRefGoogle ScholarPubMed
Damrose, EJ. On the development of idiopathic subglottic stenosis. Med Hypotheses 2008;71:122–5CrossRefGoogle ScholarPubMed
Dedo, HH, Catten, MD. Idiopathic progressive subglottic stenosis: findings and treatment in 52 patients. Ann Otol Rhinol Laryngol 2001;110:305–11CrossRefGoogle ScholarPubMed
Valdez, TA, Shapshay, SM. Idiopathic subglottic stenosis revisited. Ann Otol Rhinol Laryngol 2002;111:690–5CrossRefGoogle ScholarPubMed
Morrison, RJ, Katsantonis, NG, Motz, KM, Hillel, AT, Garrett, CG, Netterville, JL et al. Pathologic fibroblasts in idiopathic subglottic stenosis amplify local inflammatory signals. Otolaryngol Head Neck Surg 2019;160:107–15CrossRefGoogle ScholarPubMed