drugset / Trial / NCT06043440
Down Syndrome Obstructive Sleep Apnea
Phase 2
Recruiting
230 enrolled
Brigham and Women's Hospital
Children's Hospital Los Angeles · collabChildren's Hospital Medical Center, Cincinnati · collabChildren's Hospital of Philadelphia · collabChildren's Hospital of The King's Daughters · collabRainbow Babies and Children's Hospital · collabSeattle Children's Hospital · collabUniversity of Michigan · collabUniversity of Southern California · collab
RandomizedParallel-groupTriple-blindTreatment
Summary
The purpose of this study is to assess whether oxygen supplementation during sleep improves working memory and other clinical and patient-reported outcomes among children who have Down Syndrome (DS) with moderate to severe Obstructive Sleep Apnea (OSA).
Timeline
- Start
- 2023-10-24
- Primary completion
- 2027-11-30
- Completion
- 2027-12-30
Publications
- Background Das D, Medina B, Baktir MA, Mojabi FS, Fahimi A, Ponnusamy R, Salehi A. Increased incidence of intermittent hypoxemia in the Ts65Dn mouse model of Down syndrome. Neurosci Lett. 2015 Sep 14;604:91-6. doi: 10.1016/j.neulet.2015.07.040. Epub 2015 Aug 1.
- Background Schloo BL, Vawter GF, Reid LM. Down syndrome: patterns of disturbed lung growth. Hum Pathol. 1991 Sep;22(9):919-23. doi: 10.1016/0046-8177(91)90183-p.
- Background Bush D, Galambos C, Ivy DD, Abman SH, Wolter-Warmerdam K, Hickey F. Clinical Characteristics and Risk Factors for Developing Pulmonary Hypertension in Children with Down Syndrome. J Pediatr. 2018 Nov;202:212-219.e2. doi: 10.1016/j.jpeds.2018.06.031. Epub 2018 Jul 17.
- Background Cooney TP, Wentworth PJ, Thurlbeck WM. Diminished radial count is found only postnatally in Down's syndrome. Pediatr Pulmonol. 1988;5(4):204-9. doi: 10.1002/ppul.1950050405.
- Background Gonzalez OR, Gomez IG, Recalde AL, Landing BH. Postnatal development of the cystic lung lesion of Down syndrome: suggestion that the cause is reduced formation of peripheral air spaces. Pediatr Pathol. 1991 Jul-Aug;11(4):623-33. doi: 10.3109/15513819109064794.
- Background O'Neill AC, Richter GT. Pharyngeal dysphagia in children with Down syndrome. Otolaryngol Head Neck Surg. 2013 Jul;149(1):146-50. doi: 10.1177/0194599813483445. Epub 2013 Mar 22.
- Background Jackson A, Maybee J, Moran MK, Wolter-Warmerdam K, Hickey F. Clinical Characteristics of Dysphagia in Children with Down Syndrome. Dysphagia. 2016 Oct;31(5):663-71. doi: 10.1007/s00455-016-9725-7. Epub 2016 Jul 12.
- Background Khoo MCK, Hu WH, Amin R. Effects of Ventilation-Perfusion Mismatch on Severity of Obstructive Sleep Apnea: A Modeling Study. Annu Int Conf IEEE Eng Med Biol Soc. 2020 Jul;2020:2792-2795. doi: 10.1109/EMBC44109.2020.9175297.
- Background Alex RM, Panza GS, Hakim H, Badr MS, Edwards BA, Sands SA, Mateika JH. Exposure to mild intermittent hypoxia increases loop gain and the arousal threshold in participants with obstructive sleep apnoea. J Physiol. 2019 Jul;597(14):3697-3711. doi: 10.1113/JP277711. Epub 2019 May 9.
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- Background Rueda N, Vidal V, Garcia-Cerro S, Narcis JO, Llorens-Martin M, Corrales A, Lantigua S, Iglesias M, Merino J, Merino R, Martinez-Cue C. Anti-IL17 treatment ameliorates Down syndrome phenotypes in mice. Brain Behav Immun. 2018 Oct;73:235-251. doi: 10.1016/j.bbi.2018.05.008. Epub 2018 May 31.
- Background Rueda Revilla N, Martinez-Cue C. Antioxidants in Down Syndrome: From Preclinical Studies to Clinical Trials. Antioxidants (Basel). 2020 Aug 3;9(8):692. doi: 10.3390/antiox9080692.
- Background Zhang P, Wang Y, Wang H, Cao J. Sesamol alleviates chronic intermittent hypoxia-induced cognitive deficits via inhibiting oxidative stress and inflammation in rats. Neuroreport. 2021 Jan 13;32(2):105-111. doi: 10.1097/WNR.0000000000001564.
- Background Sapin E, Peyron C, Roche F, Gay N, Carcenac C, Savasta M, Levy P, Dematteis M. Chronic Intermittent Hypoxia Induces Chronic Low-Grade Neuroinflammation in the Dorsal Hippocampus of Mice. Sleep. 2015 Oct 1;38(10):1537-46. doi: 10.5665/sleep.5042.
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- Background Churchill SS, Kieckhefer GM, Bjornson KF, Herting JR. Relationship between sleep disturbance and functional outcomes in daily life habits of children with Down syndrome. Sleep. 2015 Jan 1;38(1):61-71. doi: 10.5665/sleep.4326.
- Background Edgin JO, Tooley U, Demara B, Nyhuis C, Anand P, Spano G. Sleep Disturbance and Expressive Language Development in Preschool-Age Children With Down Syndrome. Child Dev. 2015 Nov-Dec;86(6):1984-98. doi: 10.1111/cdev.12443. Epub 2015 Oct 5.
- Background Hoffmire CA, Magyar CI, Connolly HV, Fernandez ID, van Wijngaarden E. High prevalence of sleep disorders and associated comorbidities in a community sample of children with Down syndrome. J Clin Sleep Med. 2014 Apr 15;10(4):411-9. doi: 10.5664/jcsm.3618.
- Background Kuroda H, Sawatari H, Ando S, Ohkusa T, Rahmawati A, Ono J, Nishizaka M, Hashiguchi N, Matsuoka F, Chishaki A. A nationwide, cross-sectional survey on unusual sleep postures and sleep-disordered breathing-related symptoms in people with Down syndrome. J Intellect Disabil Res. 2017 Jul;61(7):656-667. doi: 10.1111/jir.12379. Epub 2017 Apr 5.
- Background Pittaras E, Colas D, Chuluun B, Allocca G, Heller C. Enhancing sleep after training improves memory in down syndrome model mice. Sleep. 2022 Apr 11;45(4):zsab247. doi: 10.1093/sleep/zsab247.
- Background Hawkins A, Langton-Hewer S, Henderson J, Tulloh RM. Management of pulmonary hypertension in Down syndrome. Eur J Pediatr. 2011 Jul;170(7):915-21. doi: 10.1007/s00431-010-1378-1. Epub 2011 Jan 4.
- Background Bush D, Abman SH, Galambos C. Prominent Intrapulmonary Bronchopulmonary Anastomoses and Abnormal Lung Development in Infants and Children with Down Syndrome. J Pediatr. 2017 Jan;180:156-162.e1. doi: 10.1016/j.jpeds.2016.08.063. Epub 2016 Sep 22.
- Background Chi TPL?Krovetz J. The pulmonary vascular bed in children with Down syndrome. J Pediatr. 1975 Apr;86(4):533-8. doi: 10.1016/s0022-3476(75)80142-9.
- Background Iwaya Y, Muneuchi J, Inoue Y, Watanabe M, Okada S, Ochiai Y. Relationship Between Pulmonary Arterial Resistance and Compliance in Patients with Down Syndrome. Pediatr Cardiol. 2019 Apr;40(4):841-847. doi: 10.1007/s00246-019-02080-9. Epub 2019 Mar 4.
- Background Bush D, Galambos C, Dunbar Ivy D. Pulmonary hypertension in children with Down syndrome. Pediatr Pulmonol. 2021 Mar;56(3):621-629. doi: 10.1002/ppul.24687. Epub 2020 Feb 12.
- Background Amin RS, Kimball TR, Kalra M, Jeffries JL, Carroll JL, Bean JA, Witt SA, Glascock BJ, Daniels SR. Left ventricular function in children with sleep-disordered breathing. Am J Cardiol. 2005 Mar 15;95(6):801-4. doi: 10.1016/j.amjcard.2004.11.044.
- Background Domany KA, Huang G, Hossain MM, Schuler CL, Somers VK, Daniels SR, Amin R. Effect of Adenotonsillectomy on Cardiac Function in Children Age 5-13 Years With Obstructive Sleep Apnea. Am J Cardiol. 2021 Feb 15;141:120-126. doi: 10.1016/j.amjcard.2020.11.019. Epub 2020 Nov 19.
- Background Amin RS, Kimball TR, Bean JA, Jeffries JL, Willging JP, Cotton RT, Witt SA, Glascock BJ, Daniels SR. Left ventricular hypertrophy and abnormal ventricular geometry in children and adolescents with obstructive sleep apnea. Am J Respir Crit Care Med. 2002 May 15;165(10):1395-9. doi: 10.1164/rccm.2105118.
- Background Konstantinopoulou S, Tapia IE, Kim JY, Xanthopoulos MS, Radcliffe J, Cohen MS, Hanna BD, Pipan M, Cielo C, Thomas AJ, Zemel B, Amin R, Bradford R, Traylor J, Shults J, Marcus CL. Relationship between obstructive sleep apnea cardiac complications and sleepiness in children with Down syndrome. Sleep Med. 2016 Jan;17:18-24. doi: 10.1016/j.sleep.2015.09.014. Epub 2015 Oct 23.
- Background Bittles AH, Glasson EJ. Clinical, social, and ethical implications of changing life expectancy in Down syndrome. Dev Med Child Neurol. 2004 Apr;46(4):282-6. doi: 10.1017/s0012162204000441. No abstract available.
- Background Presson AP, Partyka G, Jensen KM, Devine OJ, Rasmussen SA, McCabe LL, McCabe ER. Current estimate of Down Syndrome population prevalence in the United States. J Pediatr. 2013 Oct;163(4):1163-8. doi: 10.1016/j.jpeds.2013.06.013. Epub 2013 Jul 23.
- Background Chamseddin BH, Johnson RF, Mitchell RB. Obstructive Sleep Apnea in Children with Down Syndrome: Demographic, Clinical, and Polysomnographic Features. Otolaryngol Head Neck Surg. 2019 Jan;160(1):150-157. doi: 10.1177/0194599818797308. Epub 2018 Aug 28.
- Background Lee CF, Lee CH, Hsueh WY, Lin MT, Kang KT. Prevalence of Obstructive Sleep Apnea in Children With Down Syndrome: A Meta-Analysis. J Clin Sleep Med. 2018 May 15;14(5):867-875. doi: 10.5664/jcsm.7126.
- Background Cornacchia M, Sethness J, Alapat P, Lin YH, Peacock C. The Prevalence of OSA Among an Adult Population With Down Syndrome Referred to a Medical Clinic. Am J Intellect Dev Disabil. 2019 Jan;124(1):4-10. doi: 10.1352/1944-7558-124.1.4.
- Background Gimenez S, Videla L, Romero S, Benejam B, Clos S, Fernandez S, Martinez M, Carmona-Iragui M, Antonijoan RM, Mayos M, Fortuna A, Penacoba P, Plaza V, Osorio RS, Sharma RA, Bardes I, Rebillat AS, Lleo A, Blesa R, Videla S, Fortea J. Prevalence of Sleep Disorders in Adults With Down Syndrome: A Comparative Study of Self-Reported, Actigraphic, and Polysomnographic Findings. J Clin Sleep Med. 2018 Oct 15;14(10):1725-1733. doi: 10.5664/jcsm.7382.
- Background Gimenez S, Altuna M, Blessing E, Osorio RM, Fortea J. Sleep Disorders in Adults with Down Syndrome. J Clin Med. 2021 Jul 6;10(14):3012. doi: 10.3390/jcm10143012.
- Background Tarasiuk A, Greenberg-Dotan S, Simon-Tuval T, Freidman B, Goldbart AD, Tal A, Reuveni H. Elevated morbidity and health care use in children with obstructive sleep apnea syndrome. Am J Respir Crit Care Med. 2007 Jan 1;175(1):55-61. doi: 10.1164/rccm.200604-577OC. Epub 2006 Oct 12.
- Background Reuveni H, Simon T, Tal A, Elhayany A, Tarasiuk A. Health care services utilization in children with obstructive sleep apnea syndrome. Pediatrics. 2002 Jul;110(1 Pt 1):68-72. doi: 10.1542/peds.110.1.68.
- Background Tarasiuk A, Simon T, Tal A, Reuveni H. Adenotonsillectomy in children with obstructive sleep apnea syndrome reduces health care utilization. Pediatrics. 2004 Feb;113(2):351-6. doi: 10.1542/peds.113.2.351.
- Background Bergeron M, Duggins AL, Cohen AP, Leader BA, Ishman SL. The impact of persistent pediatric obstructive sleep apnea on the Quality of Life of Patients' families. Int J Pediatr Otorhinolaryngol. 2020 Feb;129:109723. doi: 10.1016/j.ijporl.2019.109723. Epub 2019 Oct 12.
- Background Jensen KM, Sevick CJ, Seewald LAS, Halbower AC, Davis MM, McCabe ERB, Kempe A, Abman SH. Greater risk of hospitalization in children with Down syndrome and OSA at higher elevation. Chest. 2015 May;147(5):1344-1351. doi: 10.1378/chest.14-1883.
- Background Boulet SL, Molinari NA, Grosse SD, Honein MA, Correa-Villasenor A. Health care expenditures for infants and young children with Down syndrome in a privately insured population. J Pediatr. 2008 Aug;153(2):241-6. doi: 10.1016/j.jpeds.2008.02.046. Epub 2008 Apr 23.
- Background Kong AM, Hurley D, Evans KA, Brixner D, Csoboth C, Visootsak J. A Retrospective, Longitudinal, Claims-Based Comparison of Concomitant Diagnoses Between Individuals with and Without Down Syndrome. J Manag Care Spec Pharm. 2017 Jul;23(7):761-770. doi: 10.18553/jmcp.2017.23.7.761.
- Background Baker AB, Farhood Z, Brandstetter KA, Teufel RJ 2nd, LaRosa A, White DR. Tonsillectomy in Children with Down Syndrome: A National Cohort of Inpatients. Otolaryngol Head Neck Surg. 2017 Sep;157(3):499-503. doi: 10.1177/0194599817711377. Epub 2017 Aug 1.
- Background Nation J, Brigger M. The Efficacy of Adenotonsillectomy for Obstructive Sleep Apnea in Children with Down Syndrome: A Systematic Review. Otolaryngol Head Neck Surg. 2017 Sep;157(3):401-408. doi: 10.1177/0194599817703921. Epub 2017 May 9.
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- Background Merrell JA, Shott SR. OSAS in Down syndrome: T&A versus T&A plus lateral pharyngoplasty. Int J Pediatr Otorhinolaryngol. 2007 Aug;71(8):1197-203. doi: 10.1016/j.ijporl.2007.04.009. Epub 2007 May 29.
- Background Thottam PJ, Trivedi S, Siegel B, Williams K, Mehta D. Comparative outcomes of severe obstructive sleep apnea in pediatric patients with Trisomy 21. Int J Pediatr Otorhinolaryngol. 2015 Jul;79(7):1013-6. doi: 10.1016/j.ijporl.2015.04.015. Epub 2015 Apr 28.
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Drugs
| Evaluation | Drug | Modality | Dose | Route |
|---|---|---|---|---|
| Comparator | Oxygen | Other / unclassified | — | — |