Long-term stability of hypoglossal nerve stimulation for the treatment of obstructive sleep apnea in children with Down syndrome

Stenerson, Matthew; Yu, Phoebe K.; Kinane, T. Bernard; Skotko, Brian G.; Hartnick, Christopher J.

doi:10.1016/j.ijporl.2021.110868

Cited by 13 publications

(17 citation statements)

References 22 publications

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“…Five (45.5%) of these 11 patients responded to therapy, compared to 44.4% of patients with a BMI under the 85th percentile ( P = 0.96) ( 21 ). Age- and sex-adjusted BMIs ranged from 19.2 to 24.6 kg/m 2 at baseline, and from 19.8 to 34.6 kg/m 2 , and BMI percentiles increased for 3 of the four patients ( 23 ).…”

Section: Resultsmentioning

confidence: 99%

“…Five studies used the OSA-18 (a validated, disease-specific quality of life instrument for OSA) and Epworth Sleepiness Scale (ESS) questionnaires, to examine the improvements in treatment durations ( 18 , 21 , 23 , 25 , 26 ). As a result, it improved their sleep quality and subjective feelings.…”

Section: Resultsmentioning

confidence: 99%

“…Four investigations examined the necessity to optimize stimulation voltage for arousal during treatment ( 15 , 18 , 20 , 23 ). According to research, AHI decreases as stimulation voltage increases, with 21.1 events/h at 1.3 V, 3.7 at 1.4 V, and 3.4 at 1.5 V ( 15 ).…”

Section: Resultsmentioning

confidence: 99%

“…Most researchers followed patients with DS and OSA for a short duration. Only one study had a follow-up duration of 44 to 58 months ( 23 ), two studies had <6 months ( 15 , 25 ), five studies had 6 to 12 months ( 18 , 20 – 22 , 26 ), and one study provided no details regarding follow-up duration ( 24 ). Noticeably, two participants had a persistently moderate OSA after 44–58 months of follow-up, postoperatively ( 23 ).…”

Section: Resultsmentioning

confidence: 99%

“…Eight studies stated that HNS treatment should consider the influence of body mass index (BMI) factors ( 15 , 18 , 20 , 21 , 23 – 26 ), and five advised that patients should have a BMI of <32 kg/m 2 ( 15 , 18 , 21 , 23 , 26 ). However, only two studies performed BMI data analysis.…”

Section: Resultsmentioning

confidence: 99%

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Hypoglossal nerve stimulation in adolescents with down syndrome and obstructive sleep apnea: A systematic review and meta-analysis

et al. 2022

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ObjectiveTo evaluate the efficacy and adverse effects of hypoglossal nerve stimulation in adolescents with down syndrome and obstructive sleep apnea.MethodsA systematic search was conducted using PubMed, Web of Science, Embase, and Scopus databases. The systematic review followed the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines. A comprehensive search strategy used a combination of Medical Subject Headings and free words with “OR” and “AND.” Articles were screened to extract data reporting apnea-hypopnea index, quality of life, voltage, follow-up duration, and complications. All included participants were adolescents with down syndrome and obstructive sleep apnea.ResultsA total of 92 articles were identified, of which 9 articles met the inclusion criteria. A total of 106 patients were included. All the studies showed that patients receiving hypoglossal nerve stimulation experienced a significant decrease in apnea-hypopnea index (at least 50%). The pooled AHI was significantly lower in patients following treatment (mean AHI reduction 17.43 events/h, 95% confidence interval 13.98–20.88 events/h, P < 0.001) after 2 case reports were excluded. The pooled OSA-18 were significantly decreased in 88 patients after treatment (mean OSA-18 reduction 1.67, 95% confidence interval 1.27–2.08, P < 0.001) after excluding 5 studies. Four investigations examined the necessity to optimize stimulation voltage for arousal during treatment. The most common complication was pain or discomfort in the tongue or mouth. Most studies had relatively short patient follow-up periods, with the most extended follow-up being 44–58 months.ConclusionHypoglossal nerve stimulation significantly reduces apnea-hypopnea index and improves the quality of life; and thus, could be a potential alternative therapy for obstructive sleep apnea in adolescents with down syndrome. The adolescent's age, potential complications, adverse events, long-term efficacy, and comfort, needs to be considered while performing hypoglossal nerve stimulation.

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Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

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Hypoglossal nerve stimulation in adolescents with down syndrome and obstructive sleep apnea: A systematic review and meta-analysis

et al. 2022

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Evaluation of Upper Airway Stimulation for Adolescents With Down Syndrome and Obstructive Sleep Apnea

Stenerson

Ishman

et al. 2022

JAMA Otolaryngol Head Neck Surg

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IMPORTANCEPatients with Down syndrome have a high incidence of persistent obstructive sleep apnea (OSA) and limited treatment options. Upper airway hypoglossal stimulation has been shown to be effective for adults with OSA but has not yet been evaluated for pediatric populations.OBJECTIVE To evaluate the safety and effectiveness of upper airway stimulation for adolescent patients with Down syndrome and severe OSA.DESIGN, SETTING, AND PARTICIPANTS This prospective single-group multicenter cohort study with 1-year follow-up was conducted between April 1, 2015, and July 31, 2021, among a referred sample of 42 consecutive adolescent patients with Down syndrome and persistent severe OSA after adenotonsillectomy.INTERVENTION Upper airway stimulation. MAIN OUTCOMES AND MEASURESThe prespecified primary outcomes were safety and the change in apnea-hypopnea index (AHI) from baseline to 12 months postoperatively. Polysomnographic and quality of life outcomes were assessed at 1, 2, 6, and 12 months postoperatively. RESULTS Among the 42 patients (28 male patients [66.7%]; mean [SD] age, 15.1 [3.0] years), there was a mean (SD) decrease in AHI of 12.9 (13.2) events/h (95% CI, -17.0 to -8.7 events/h). With the use of a therapy response definition of a 50% decrease in AHI, the 12-month response rate was 65.9% (27 of 41), and 73.2% of patients (30 of 41) had a 12-month AHI of less than 10 events/h. The most common complication was temporary tongue or oral discomfort, which occurred in 5 patients (11.9%). The reoperation rate was 4.8% (n = 2). The mean (SD) improvement in the OSA-18 total score was 34.8 (20.3) (95% CI, -42.1 to -27.5), and the mean (SD) improvement in the Epworth Sleepiness Scale score was 5.1 (6.9) (95% CI, -7.4 to -2.8). The mean (SD) duration of nightly therapy was 9.0 (1.8) hours, with 40 patients (95.2%) using the device at least 4 hours a night.CONCLUSIONS AND RELEVANCE Upper airway stimulation was able to be safely performed for 42 adolescents who had Down syndrome and persistent severe OSA after adenotonsillectomy with positive airway pressure intolerance. There was an acceptable adverse event profile with high rates of therapy response and quality of life improvement.

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Down Syndrome for the Otolaryngologist

Zalzal

Lawlor

2023

JAMA Otolaryngol Head Neck Surg

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ImportanceThere are many features of Down syndrome that prompt referral to an otolaryngologist. As the lifetime prevalence and life expectancy of individuals with Down syndrome increase, it is increasingly likely that otolaryngologists will have the opportunity to care for patients with Down syndrome.ObservationsA confluence of characteristics common to Down syndrome may be associated with issues in the head and neck, from infancy through adulthood. Hearing concerns range from narrow ear canals and cerumen impactions to eustachian tube dysfunction, middle ear effusion, cochlear malformations, and conductive, sensorineural, and/or mixed hearing loss. Immune deficiency, hypertrophy of Waldeyer ring, and hypoplastic sinuses may complicate and develop into chronic rhinosinusitis. Speech delay, obstructive sleep apnea, dysphagia, and airway anomalies are also common among this patient population. Because these concerns may necessitate otolaryngologic surgery, it is vital for otolaryngologists to familiarize themselves with anesthetic concerns, including cervical spine instability, in patients with Down syndrome. Comorbid cardiac disease, hypothyroidism, and obesity may also affect these patients and otolaryngologic care.Conclusions and RelevanceIndividuals with Down syndrome may visit otolaryngology practices at all ages. Otolaryngologists that familiarize themselves with the head and neck manifestations that are common among patients with Down syndrome and know when to order screening tests will be able to provide comprehensive care.

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Long-term stability of hypoglossal nerve stimulation for the treatment of obstructive sleep apnea in children with Down syndrome

Cited by 13 publications

References 22 publications

Hypoglossal nerve stimulation in adolescents with down syndrome and obstructive sleep apnea: A systematic review and meta-analysis

Hypoglossal nerve stimulation in adolescents with down syndrome and obstructive sleep apnea: A systematic review and meta-analysis

Evaluation of Upper Airway Stimulation for Adolescents With Down Syndrome and Obstructive Sleep Apnea

Down Syndrome for the Otolaryngologist

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