Christen L Caloway1,2, Gillian R Diercks1,2, Donald Keamy1,2,3, Vanessa de Guzman1, Ryan Soose4, Nikhila Raol5, Sally R Shott6, Stacey L Ishman6, Christopher J Hartnick1,2. 1. Department of Otolaryngology, Massachusetts Eye and Ear Infirmary, Boston, Massachusetts. 2. Department of Otology and Laryngology, Harvard Medical School, Boston, Massachusetts. 3. Pediatric Sleep Associates, Massachusetts General Hospital for Children, Boston, Massachusetts. 4. University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania. 5. Department of Otolaryngology, Emory University School of Medicine, Atlanta, Georgia. 6. Department of Otolaryngology, Cincinnati Children's Hospital, Cincinnati, Ohio, U.S.A.
Abstract
OBJECTIVES/HYPOTHESIS: Hypoglossal nerve (HGN) stimulation is a novel therapy for obstructive sleep apnea (OSA) in adults. Its efficacy and safety in children with Down syndrome (DS) was previously reported in a preliminary case series of six adolescents. STUDY DESIGN: Case series. METHODS: Twenty nonobese children and adolescents (aged 10-21 years) with DS and severe OSA (apnea-hypopnea index [AHI] >10 and <50 events/hr) despite prior adenotonsillectomy were enrolled. Participants had failed a trial of continuous positive airway pressure therapy and underwent sleep endoscopy confirming surgical candidacy. The primary outcome was to assess safety and monitor for adverse events. Secondary outcomes included efficacy in reducing AHI (% reduction in AHI), adherence to therapy, and change in a validated quality-of-life instrument, the OSA-18 survey. RESULTS: All 20 children (median age = 16.0 years [interquartile range = 13-17 years], 13 male) were implanted with no long-term complications. We report two interval adverse events, both of which were corrected with revision surgery. Twenty participants completed the 2-month polysomnogram, with median percent reduction in titration AHI of 85% (interquartile range = 75%-92%). The median nightly usage for these children was 9.21 hours/night. There was a median change in the OSA-18 score of 1.15, indicating a moderate, yet significant, clinical change. CONCLUSIONS: HGN stimulation was safe and effective in the study population. Two minor surgical complications were corrected surgically. Overall, these data suggest that pediatric HGN stimulation appears to be a safe and effective therapy for children with DS and refractory severe OSA. LEVEL OF EVIDENCE: 4 Laryngoscope, 130:E263-E267, 2020.
OBJECTIVES/HYPOTHESIS: Hypoglossal nerve (HGN) stimulation is a novel therapy for obstructive sleep apnea (OSA) in adults. Its efficacy and safety in children with Down syndrome (DS) was previously reported in a preliminary case series of six adolescents. STUDY DESIGN: Case series. METHODS: Twenty nonobese children and adolescents (aged 10-21 years) with DS and severe OSA (apnea-hypopnea index [AHI] >10 and <50 events/hr) despite prior adenotonsillectomy were enrolled. Participants had failed a trial of continuous positive airway pressure therapy and underwent sleep endoscopy confirming surgical candidacy. The primary outcome was to assess safety and monitor for adverse events. Secondary outcomes included efficacy in reducing AHI (% reduction in AHI), adherence to therapy, and change in a validated quality-of-life instrument, the OSA-18 survey. RESULTS: All 20 children (median age = 16.0 years [interquartile range = 13-17 years], 13 male) were implanted with no long-term complications. We report two interval adverse events, both of which were corrected with revision surgery. Twenty participants completed the 2-month polysomnogram, with median percent reduction in titration AHI of 85% (interquartile range = 75%-92%). The median nightly usage for these children was 9.21 hours/night. There was a median change in the OSA-18 score of 1.15, indicating a moderate, yet significant, clinical change. CONCLUSIONS: HGN stimulation was safe and effective in the study population. Two minor surgical complications were corrected surgically. Overall, these data suggest that pediatric HGN stimulation appears to be a safe and effective therapy for children with DS and refractory severe OSA. LEVEL OF EVIDENCE: 4 Laryngoscope, 130:E263-E267, 2020.
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
Authors: Saif Mashaqi; Salma Imran Patel; Daniel Combs; Lauren Estep; Sonia Helmick; Joan Machamer; Sairam Parthasarathy Journal: Int J Environ Res Public Health Date: 2021-02-09 Impact factor: 3.390
Authors: Maria V Suurna; Ofer Jacobowitz; Jolie Chang; Ioannis Koutsourelakis; David Smith; Uri Alkan; Mark D'Agostino; Maurits Boon; Clemens Heiser; Paul Hoff; Colin Huntley; David Kent; Alan Kominsky; Richard Lewis; Joachim T Maurer; Madeline J Ravesloot; Ryan Soose; Armin Steffen; Edward M Weaver; Amy M Williams; Tucker Woodson; Kathleen Yaremchuk; Stacey L Ishman Journal: J Clin Sleep Med Date: 2021-12-01 Impact factor: 4.062