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Clinical Trial
. 2021 Jul;131(7):1663-1669.
doi: 10.1002/lary.29290. Epub 2020 Dec 2.

Redefining Success by Focusing on Failures After Pediatric Hypoglossal Stimulation in Down Syndrome

Phoebe K Yu  1 , Asitha D L Jayawardena  1 , Matthew Stenerson  1 , Margaret B Pulsifer  1   2 , Julie A Grieco  2 , Leonard Abbeduto  3 , Raj C Dedhia  4 , Ryan J Soose  5 , Allison Tobey  5 , Nikhila Raol  6 , Stacey L Ishman  7 , Sally R Shott  7 , Michael S Cohen  1 , Brian G Skotko  8   9 , Thomas B Kinane  8 , Donald G Keamy Jr  1 , Christopher J Hartnick  1
Affiliations
Clinical Trial

Redefining Success by Focusing on Failures After Pediatric Hypoglossal Stimulation in Down Syndrome

Phoebe K Yu et al. Laryngoscope. 2021 Jul.

Abstract

Objectives/hypothesis: Patients with Down syndrome have a high incidence of obstructive sleep apnea (OSA) and limited treatment options. Hypoglossal stimulation has shown efficacy but has not yet been approved for pediatric populations. Our objective is to characterize the therapy response of adolescent patients with down syndrome and severe OSA who underwent hypoglossal stimulation.

Study design: Prospective longitudinal trial.

Methods: We are conducting a multicenter single-arm trial of hypoglossal stimulation for adolescent patients with Down syndrome and severe OSA. Interim analysis was performed to compare objective sleep and quality of life outcomes at 12 months postoperatively for the first 20 patients.

Results: The mean age was 15.5 and baseline AHI 24.2. Of the 20 patients, two patients (10.0%) had an AHI under 1.5 at 12 months; nine patients of 20 (45.0%) under five; and 15 patients of 20 (75.0%) under 10. The mean decrease in AHI was 15.1 (P < .001). Patients with postoperative AHI over five had an average baseline OSA-18 survey score of 3.5 with an average improvement of 1.7 (P = .002); in addition, six of these patients had a relative decrease of apneas compared to hypopneas and seven had an improvement in percentage of time with oxygen saturation below 90%.

Conclusions: Patients with persistently elevated AHI 12 months after hypoglossal implantation experienced improvement in polysomnographic and quality of life outcomes. These results suggest the need for a closer look at physiologic markers for success beyond reporting AHI as the gold standard.

Level of evidence: 4 Laryngoscope, 131:1663-1669, 2021.

Keywords: Down syndrome; hypoglossal nerve stimulation; pediatric obstructive sleep apnea.

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Conflict of interest statement

The authors have no other funding, financial relationships, or conflicts of interest to disclose.

Figures

Fig. 1.
Fig. 1.
Postoperative change in a) apnea-hypopnea index (AHI) b) OSA-18 survey scores c) Epworth Sleepiness (ESS) scores.
See this image and copyright information in PMC

References

    1. Shott SR. Down syndrome: common otolaryngologic manifestations. Am J Med Genet C Semin Med Genet 2006;142C:131–140. - PubMed
    1. Knauert M, Naik S, Gillespie MB, Kryger M. Clinical consequences and economic costs of untreated obstructive sleep apnea syndrome. World J Otorhinolaryngol Head Neck Surg 2015;1:17–27. - PMC - PubMed
    1. Horne RS, Wijayaratne P, Nixon GM, Walter LM. Sleep and sleep disordered breathing in children with down syndrome: effects on behaviour, neurocognition and the cardiovascular system. Sleep Med Rev 2019;44:1–11. - PubMed
    1. Ingram DG, Singh AV, Ehsan Z, Birnbaum BF. obstructive sleep apnea and pulmonary hypertension in children. Paediatr Respir Rev 2017;23:33–39. - PubMed
    1. Breslin J, Spano G, Bootzin R, Anand P, Nadel L, Edgin J. Obstructive sleep apnea syndrome and cognition in Down syndrome. Dev Med Child Neurol 2014;56:657–664. - PubMed

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