September 2019 HSC Section 1 Congenital and Pediatric Problems

Reprinted by permission of Laryngoscope. 2017; 127(7):1707-1711.

The Laryngoscope V C 2016 The American Laryngological, Rhinological and Otological Society, Inc.

Voice Outcome and Voice-Related Quality of Life After Surgery for Pediatric Laryngotracheal Stenosis

Bas Pullens, MD; Marieke Hakkesteegt, PhD; Hans Hoeve, PhD; Marieke Timmerman, MD; Koen Joosten, PhD

Objectives: To evaluate the long-term outcome of voice quality and voice-related quality of life after open airway surgery for pediatric laryngotracheal stenosis. Study Design: Prospective cohort study. Methods: Children under the age of 18 years at time of follow-up and with a history of open airway surgery for acquired laryngotracheal stenosis were included in this analysis. To assess voice-related quality of life, the pediatric voice handicap (pVHI) index was completed by the patients’ parents. The dysphonia severity index (DSI) was used as an objective measurement for voice quality. Results: Fifty-five parents completed the pVHI, and 38 children completed the DSI. This showed high pVHI values and low total DSI scores, indicating significant voice disturbance. After multivariate analysis, the presence of comorbidities and glottic involvement of the stenosis are associated with poor long-term voice-related quality of life. Conclusion: Significant voice disturbance is common after surgery for pediatric laryngotracheal stenosis. Glottic involve- ment of the stenosis and comorbidities is associated with poor voice-related quality of life. Evaluation of pre- and postoperative voice quality and voice-related quality of life is advised for children treated for laryngotracheal stenosis. Key Words: Laryngotracheal stenosis, laryngotracheal reconstruction, cricotracheal resection, surgical results, voice outcome, dysphonia, children. Level of Evidence: 2B. Laryngoscope , 127:1707–1711, 2017

INTRODUCTION Pediatric laryngotracheal stenosis (LTS) is a life- threatening condition for which a tracheostomy is often necessary. Surgery focuses on creating an adequate airway for spontaneous breathing and aims to relieve the patient of the tracheostomy. Using open airway surgery such as laryngotracheal reconstruction (LTR) and cricotracheal resection (CTR), excellent outcomes were achieved as far as decannulation is concerned. However, long-term func- tional deficits and deficits in health-related quality of life (HR-QoL) may still exist. 1–3 Acquired LTS often involves the glottis, impairing glottic movement with extensive scarring of the posterior aspect of the glottis and ankylosis of the arytenoids. Surgery aims to enlarge the airway by opening the scarred glottis. In theory, this could contribute to postoperative dysphonia. From the Department of Otorhinolaryngology and Head and Neck Surgery ( B . P ., M . H ., H . H ., M . T .); and the Department of Pediatrics, Pediatric Intensive Care Unit ( K . J .), Erasmus Medical Center, Sophia Children’s Hospital, Rotterdam, The Netherlands. Editor’s Note: This Manuscript was accepted for publication September 9, 2016. Financial Disclosure: Erasmus University Rotterdam Trust Fund, reference number 97030.23/11.0530. The authors have no other funding, financial relationships, or conflicts of interest to disclose. Send correspondence to B. Pullens, Department of Otorhinolaryngology, Erasmus Medical Center, Sophia Children’s Hospital, Room SP 1455, Dr Molewaterplein 60, 3015 GJ, Rotterdam, the Netherlands. E-mail: b.pullens@erasmusmc.nl

Over the past decades, a number of centers have published on voice outcome after pediatric airway sur- gery. Although there is a large heterogeneity, the studies are unanimous in reporting a high incidence of voice dis- turbance ranging from mild dysphonia to severe voice disorders. Endoscopy usually reveals an abnormal lar- ynx and abnormal phonation. Until now, no study has identified significant factors associated with voice outcome. 4–8 The aim of this study is to describe the long-term voice outcome and voice-related quality of life after open airway surgery for pediatric laryngotracheal stenosis in our tertiary referral center. We try to identify factors associated with voice outcome. MATERIALS AND METHODS This study is part of a large follow-up project. All chil- dren who were treated for acquired LTS between 1994 and 2009 by means of an LTR or a CTR were approached for this study and invited for an outpatient, follow-up visit. Children younger than the age of 4 years and those still with a trache- ostomy were excluded from this study. Children under 18 years at time of follow-up visit were included in this voice analysis. Baseline characteristics (gender, age at time of follow-up) and operative factors (Cotton-Myer [CM] grade of stenosis, preoperative tracheostomy, presence of comorbidities, congenital syndromes, localization of stenosis, age at time of surgery, and grafts used) were retrospectively extracted from the hospital records.

DOI: 10.1002/lary.26374

Laryngoscope 127: July 2017

Pullens et al.: Voice Outcome After Pediatric Airway Surgery

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