Phoebe K Yu1, Asitha D L Jayawardena1, Matthew Stenerson1, Margaret B Pulsifer1,2, Julie A Grieco2, Leonard Abbeduto3, Raj C Dedhia4, Ryan J Soose5, Allison Tobey5, Nikhila Raol6, Stacey L Ishman7, Sally R Shott7, Michael S Cohen1, Brian G Skotko8,9, Thomas B Kinane8, Donald G Keamy1, Christopher J Hartnick1. 1. Department of Otolaryngology, Massachusetts Eye and Ear Infirmary, Boston, Massachusetts, U.S.A. 2. Department of Psychiatry, Massachusetts General Hospital, Boston, Massachusetts, U.S.A. 3. MIND Institute and Department of Psychiatry and Behavioral Sciences, University of California Davis, Sacramento, California, U.S.A. 4. CPAP Alternatives Clinic and Division of Sleep Surgery, University of Pennsylvania Perelman School of Medicine, Philadelphia, Pennsylvania, U.S.A. 5. Department of Otolaryngology and Division of Sleep Surgery, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania, U.S.A. 6. Department of Otolaryngology, Children's Healthcare of Atlanta, Atlanta, Georgia, U.S.A. 7. Department of Otolaryngology-Head and Neck Surgery, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, U.S.A. 8. Department of Pediatrics, Massachusetts General Hospital, Boston, Massachusetts, U.S.A. 9. Down Syndrome Program, Division of Medical Genetics and Metabolism, Department of Pediatrics, Massachusetts General Hospital, Boston, Massachusetts, U.S.A.
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.
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.
Authors: Phoebe K Yu; Matthew Stenerson; Stacey L Ishman; Sally R Shott; Nikhila Raol; Ryan J Soose; Allison Tobey; Cristina Baldassari; Raj C Dedhia; Margaret B Pulsifer; Julie A Grieco; Leonard J Abbeduto; Thomas B Kinane; Donald G Keamy; Brian G Skotko; Christopher J Hartnick Journal: JAMA Otolaryngol Head Neck Surg Date: 2022-06-01 Impact factor: 8.961