September 2019 HSC Section 1 Congenital and Pediatric Problems

CONCLUSION After surgery for pediatric laryngotracheal stenosis, significant voice disturbance is a common finding. Preop- erative glottic involvement of the stenosis and the pres- ence of comorbidities are important factors for poorer voice-related quality of life outcome. Voice quality and voice-related quality of life should be part of the stan- dard pre- and postoperative assessment in patients suf- fering from laryngotracheal stenosis. BIBLIOGRAPHY 1. Pullens B, Hoeve LJ, Timmerman MK, van der Schroeff MP, Joosten KF. Characteristics and surgical outcome of 98 infants and children surgical- ly treated for a laryngotracheal stenosis after endotracheal intubation: excellent outcome for higher grades of stenosis after SS-LTR. Int J Pediatr Otorhinolaryngol 2014;78:1444–1448. 2. Pullens B, Pijnenburg MW, Hoeve HJ, et al. Long-term functional airway assessment after open airway surgery for laryngotracheal stenosis. Laryngoscope 2016;126:472–477. 3. Pullens B, Dulfer K, Buysse CM, Hoeve LJ, Timmerman MK, Joosten KF. Long-term quality of life in children after open airway surgery for lar- yngotracheal stenosis. Int J Pediatr Otorhinolaryngol 2016;84:88–93. 4. Bailey CM, Clary RA, Pengilly A, Albert DM. Voice quality following lar- yngotracheal reconstruction. Int J Pediatr Otorhinolaryngol 1995; 32(suppl):S93–S95. 5. Clary RA, Pengilly A, Bailey M, et al. Analysis of voice outcomes in pediat- ric patients following surgical procedures for laryngotracheal stenosis. Arch Otolaryngol Head Neck Surg 1996;122:1189–1194. 6. de Alarcon A. Voice outcomes after pediatric airway reconstruction. Laryn- goscope 2012;122(suppl 4):S84–S86. 7. Kelchner LN, Miller CK. Current research in voice and swallowing out- comes following pediatric airway reconstruction. Curr Opin Otolaryngol Head Neck Surg 2008;16:221–225. 8. Zacharias SR, Weinrich B, Brehm SB, et al. Assessment of vibratory char- acteristics in children following airway reconstruction using flexible and rigid endoscopy and stroboscopy. JAMA Otolaryngol Head Neck Surg 2015;141:882–887. 9. Zur KB, Cotton S, Kelchner L, Baker S, Weinrich B, Lee L. Pediatric Voice Handicap Index (pVHI): a new tool for evaluating pediatric dysphonia. Int J Pediatr Otorhinolaryngol 2007;71:77–82. 10. Wuyts FL, De Bodt MS, Molenberghs G, et al. The dysphonia severity index: an objective measure of vocal quality based on a multiparameter approach. J Speech Lang Hear Res 2000;43:796–809. 11. Hakkesteegt MM, Brocaar MP, Wieringa MH. The applicability of the dysphonia severity index and the voice handicap index in evaluating effects of voice therapy and phonosurgery. J Voice 2010;24:199–205. 12. Hakkesteegt MM, Brocaar MP, Wieringa MH, Feenstra L. Influence of age and gender on the dysphonia severity index. A study of normative val- ues. Folia Phoniatr Logop 2006;58:264–273. 13. Wibke Freudenhammer GK. Development of Vocal Performance in 5th Grade Children: A Longitudinal Study of Choral Class Singing. Proceedings of the 7th Triennial Conference of European Society for the Cognitive Scien- ces of Music (ESCOM 2009). Jyvaskyla, Finland; 2009: 134–140. 14. De Bodt M, Heylen L, Mertens F, Vanderwegen J, Van de heyning P. De Dys- phonia Severity Index (DSI) een objectieve maat voor stemkwaliteit. In: DeBodt M, Heylen L, Mertens F, Vanderwegen J, Van de heyning P. Stem- stoornissen. Antwerpen-Apeldoorn, Nederland: Garant;. 2008: p. 163–164. 15. Baudonck N, D’Haeseleer E, Dhooge I, Van Lierde K. Objective vocal qual- ity in children using cochlear implants: a multiparameter approach. J Voice 2011;25:683–691. 16. Pebbili GK, Kidwai J, Shabnam S. Dysphonia severity index in typically developing Indian children. J Voice 2016. pii: S0892-1997(16)00002-3. doi: 10.1016/j.jvoice.2015.12.017. 17. Baker S, Kelchner L, Weinrich B, et al. Pediatric laryngotracheal stenosis and airway reconstruction: a review of voice outcomes, assessment, and treatment issues. J Voice 2006;20:631–641. 18. Kelchner LN, Weinrich B, Brehm SB, Tabangin ME, de Alarcon A. Char- acterization of supraglottic phonation in children after airway recon- struction. Ann Otol Rhinol Laryngol 2010;119:383–390. 19. de Alarcon A, Brehm SB, Kelchner LN, Meinzen-Derr J, Middendorf J, Weinrich B. Comparison of pediatric voice handicap index scores with perceptual voice analysis in patients following airway reconstruction. Ann Otol Rhinol Laryngol 2009;118:581–586. 20. Monnier P. Pediatric airway surgery. In: Monnier P, ed. Pediatric Airway Surgery: Management of Laryngotracheal Stenosis in Infants and Children. Berlin, Germany: Springer; 2011: 234–239.

data on the influence of stenting on postoperative voice quality is lacking. Our study cohort predominately consists of grade II and III stenoses for which a single stage laryngotracheal reconstruction was performed using posterior grafts. Subsequently, our results reflect mainly voice outcome in patients with grade II and III stenosis treated with a single-stage LTR and posterior graft. Using a posterior cartilage graft very well could influence the quality of the voice. Because we have used a posterior graft in almost all of our patients (51 out of 55 cases), an honest evaluation on the effect of the poste- rior graft on voice outcome cannot be made with our data. To our opinion, however, it is more likely that the extensive scarring of the glottis predominates a bad voice after surgery regardless of the type of graft or sur- gical technique used. In this regard, it would be very interesting to have a series of pre- and postoperative voice measurements in order to identify the true influ- ence that an airway reconstruction has on the quality of the voice. This will be the subject of future research. Strengths and Limitations This study is the first to evaluate long-term voice outcome in a cohort of patients after airway surgery, regardless of their subjective voice complaints. We have identified operative factors associated with poor voice- related quality of life after surgery. These findings have important implications for the counseling of patients and parents suffering from pediatric laryngotracheal steno- sis. Our results indicate that there is a clear need for an assessment of the voice quality and voice-related quality of life at long-term follow-up. We have used the DSI as an objective measure for voice quality and found a low DSI score in a large portion of our study cohort. With regard to limitations, this is still a relatively small cohort for assessing such a heterogenic patient population. There is a significant difference in severity of the stenosis between the complete cases and noncom- plete cases. However, we feel that this is of no influence on the voice outcome analysis and conclusions. The demographical and operative data was collected retro- spectively from the hospital records, carrying the risk of incomplete data. The use of the DSI as a means to quantify voice quality in children is difficult because no norm values exist for this age group, and we encountered a large number of patients who could not comply with the proto- col. This underlines the difficulty when trying to quanti- fy voice quality in the pediatric population. We have determined a DSI score lower than 2,0 to correlate to significant voice disturbance. Although there is no robust evidence for this, we feel that it is a very prudent estimate and that the actual norm score will be higher than two.

Laryngoscope 127: July 2017

Pullens et al.: Voice Outcome After Pediatric Airway Surgery

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