Hypoglossal nerve stimulation in three adults with down syndrome and severe obstructive sleep apnea

Li, Carol; Boon, Maurits; Ishman, Stacey L.; Suurna, Maria V.

doi:10.1002/lary.27723

Cited by 19 publications

(18 citation statements)

References 19 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…However, 55% of patients still had 12‐month post‐operative AHI of 5 or greater. In a small study of three adult patients with severe OSA and Down syndrome, all patients experienced substantial reductions in AHI even though titrated AHI continued to be >5 in two of these patients 21 . In comparison, two large studies in the general adult population have reported therapy response rates of 63% to 69% 15, 22 …”

Section: Discussionmentioning

confidence: 98%

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

Jayawardena

Stenerson

et al. 2020

The Laryngoscope

Self Cite

View full text Add to dashboard Cite

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

show abstract

Section: Discussionmentioning

confidence: 98%

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

Jayawardena

Stenerson

et al. 2020

The Laryngoscope

Self Cite

View full text Add to dashboard Cite

show abstract

“…3 For the adult population, the prevalence of OSA is estimated to be 35% to 42% for adults with DS compared to 12% for adults without DS. 4 Untreated OSA is associated with significant complications including cardiovascular and cerebrovascular disease, pulmonary hypertension, neurocognitive impairment, and death. 4 An effective treatment for refractory OSA is needed to improve the quality of life for these patients.…”

Section: Introductionmentioning

confidence: 99%

“…4 Untreated OSA is associated with significant complications including cardiovascular and cerebrovascular disease, pulmonary hypertension, neurocognitive impairment, and death. 4 An effective treatment for refractory OSA is needed to improve the quality of life for these patients.…”

Section: Introductionmentioning

confidence: 99%

“…2 While data is limited for adults with DS, continuous positive airway pressure (CPAP) is the gold standard of OSA treatment for adults with only 45 to 55% adherence long-term requiring alternative therapies. 4…”

Section: Introductionmentioning

confidence: 99%

“…Hypoglossal nerve stimulation involves implanting a device such as Inspire (Inspire Medical Systems, Inc., Maple Grove, MN) as shown in Figure 1 to electrically stimulate the genioglossus protrusion with successful outcomes reported in pediatric and adult DS patients. 4-6 Two other hypoglossal nerve stimulators available on the market are ImThera® and Genio™, but at this time there are no current studies looking at these devices in DS patients. Midline posterior glossectomy plus lingual tonsillectomy, which involves removing tissue from the surface or base of the tongue as shown in Figure 2, 15 has been shown to improve outcomes for significant OSA in children with DS.…”

Section: Introductionmentioning

confidence: 99%

See 2 more Smart Citations

Tongue-Based Procedures in Treating Refractory Obstructive Sleep Apnea in Down Syndrome Patients: A Systematic Review and Meta-Analysis

Lee

Chung

Johnson

et al. 2021

FACE

View full text Add to dashboard Cite

Objective: There is a high prevalence of obstructive sleep apnea (OSA) in pediatric and adult Down Syndrome (DS) patients that is refractory to adenotonsillectomy and continuous positive airway pressure. Newer treatment modalities have emerged with improved outcomes. The objective is to provide an updated systematic review and meta-analysis to analyze the clinical outcomes of OSA in pediatric and adult DS patients with hypoglossal nerve stimulation using Inspire, midline posterior glossectomy plus lingual tonsillectomy (MPG + LT), and combined genioglossus advancement plus radiofrequency (GGS + RF). Methods: A comprehensive literature search of PubMed and Google Scholar was performed followed by a meta-analysis. Studies with preoperative and post-operative Apnea Hypopnea Index (AHI) values were included with patients serving as their own control. Results: Across 5 studies, 56 patients were analyzed. The mean reduction in AHI was statistically significant before vs. after procedure ( P < .001 for hypoglossal nerve stimulation using Inspire with a paired 2-tailed t-test and P = .031 for MPG + LT). Although individual patient AHI values were unavailable in the GGS + RF study, the standard difference in mean AHI was also significant for GGS + RF with P = .001. Device malfunction was the most common complication for Inspire while postoperative bleeding was observed for MPG + LT and nasopharyngeal obstruction and retropalatal collapse were observed for GGS + RF. Conclusion: This review reveals significant improvement in AHI with Inspire, MPG + LT, and GGS + RF for DS patients with refractory OSA. Further investigation is needed for comparison between these 3 therapies.

show abstract

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

Stenerson

Ishman

et al. 2022

JAMA Otolaryngol Head Neck Surg

Self Cite

View full text Add to dashboard Cite

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.

show abstract

Hypoglossal nerve stimulation in three adults with down syndrome and severe obstructive sleep apnea

Cited by 19 publications

References 19 publications

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

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

Tongue-Based Procedures in Treating Refractory Obstructive Sleep Apnea in Down Syndrome Patients: A Systematic Review and Meta-Analysis

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

Contact Info

Product

Resources

About