{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,22]],"date-time":"2025-10-22T17:28:05Z","timestamp":1761154085433,"version":"3.41.2"},"reference-count":47,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2023,5,19]],"date-time":"2023-05-19T00:00:00Z","timestamp":1684454400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Sleep"],"abstract":"<jats:sec><jats:title>Rationale<\/jats:title><jats:p>Children with Down syndrome (DS) make up a substantial portion of long-term non-invasive ventilation (LT-NIV) users though it is unclear if their unique features alter LT-NIV efficacy or use. The aim of this study is to compare the use and outcomes of LT-NIV for children with DS and a matched comparison (MCG).<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>This is a sub-study of a 10-year retrospective review of children initiated on LT-NIV in Alberta, Canada (<jats:italic>N<\/jats:italic> = 622). Children with DS (<jats:italic>n<\/jats:italic> = 106) were matched in a 1:2 ratio with other children using LT-NIV based on age and therapy start date. Data was collected from medical and sleep laboratory records.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Upper airway disease was the most common indication for LT-NIV in both groups, though was higher in children with DS (DS: 90% vs. MCG: 50%, OR 8.64 [95% CI 4.38\u201317.04]). Sleep and respiratory parameters, at the baseline diagnostic sleep study and the change from baseline to treatment study, did not differ between groups. Nasal masks were the predominant mask type in both children with DS (55%) and the MCG (66%) with more children with DS, compared to the MCG, using full face masks (DS: 45 vs. MCG: 33%, <jats:italic>p<\/jats:italic> &amp;lt; 0.05). Continuous positive airway pressure was used more often in children with DS (93.3% vs. 69.2%, <jats:italic>p<\/jats:italic> &amp;lt; 0.001) while bilevel-positive airway pressure was more common in the MCG (DS: 6.7% vs. MCG 30.8%, <jats:italic>p<\/jats:italic> &amp;lt; 0.001). Children with DS were followed longer than children in the MCG (DS: 2.4 [IQR 2.8] vs. MCG: 1.8 [IQR 2.7] years, <jats:italic>p<\/jats:italic> &amp;lt; 0.05). Adherence was lower in children with DS at both 6\u201312 month follow-up and most recent visit with a similar decrease in adherence in both groups over the follow-up period (0.0 [IQR 1.4] vs. \u22120.3 [IQR 2.0]. Despite this, 66% and 49% of children with DS used LT-NIV for more than 4 h\/night at the 6\u201312 month and most current visit, respectively. Discontinuation of LT-NIV and mortality did not differ between groups.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>LT-NIV is a common and efficacious treatment in children with DS used predominantly for upper airway obstruction. While adherence is lower, the majority of children with DS are successful at using LT-NIV.<\/jats:p><\/jats:sec>","DOI":"10.3389\/frsle.2023.1169236","type":"journal-article","created":{"date-parts":[[2023,5,22]],"date-time":"2023-05-22T08:33:40Z","timestamp":1684744420000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":2,"title":["Comparison of non-invasive ventilation use and outcomes in children with Down syndrome and other children using this technology"],"prefix":"10.3389","volume":"2","author":[{"given":"Rafiaa","family":"Valji","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Maria L.","family":"Castro-Codesal","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Melanie","family":"Lewis","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Joanna E.","family":"MacLean","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1965","published-online":{"date-parts":[[2023,5,19]]},"reference":[{"key":"B1","doi-asserted-by":"publisher","DOI":"10.1183\/13993003.02258-2020","article-title":"Sleep-related breathing disorders and pulmonary hypertension","author":"Adir","year":"2021","journal-title":"Eur. Resp. J"},{"key":"B2","doi-asserted-by":"publisher","first-page":"109","DOI":"10.1513\/AnnalsATS.202009-1089OC","article-title":"Outcomes of long-term noninvasive ventilation use in children with neuromuscular disease: systematic review and meta-analysis","volume":"19","author":"AlBalawi","year":"2022","journal-title":"Ann. Am. Thorac. Soc"},{"key":"B3","doi-asserted-by":"publisher","DOI":"10.1186\/s40510-016-0148-7","article-title":"Craniofacial features as assessed by lateral cephalometric measurements in children with Down syndrome","author":"Allareddy","year":"2016","journal-title":"Prog. Orthod"},{"key":"B4","doi-asserted-by":"publisher","first-page":"1422","DOI":"10.1002\/ppul.24138","article-title":"of long-term continuous positive airway pressure in children","volume":"53","author":"Amaddeo","year":"2018","journal-title":"Pedia. Pulmonol"},{"key":"B5","doi-asserted-by":"publisher","first-page":"866","DOI":"10.1164\/ajrccm.153.2.8564147","article-title":"Standards and indications for cardiopulmonary sleep studies in children. American thoracic society","volume":"153","year":"1996","journal-title":"Am. J. Respirat. Crit. Care Med"},{"year":"1971","author":"Anders","journal-title":"A Manual of Standarized Terminology: Techniques and Criteria for Scoring States of Sleep and Wakefulness in Newborn Infants","key":"B6"},{"key":"B7","doi-asserted-by":"publisher","DOI":"10.3389\/fped.2018.00013","article-title":"Long-term non-invasive ventilation in infants: A systematic review and meta-analysis","author":"Bedi","year":"2018","journal-title":"Front. Pediatr"},{"key":"B8","doi-asserted-by":"publisher","first-page":"e94","DOI":"10.1016\/S2589-7500(19)30214-6","article-title":"Adherence in children using positive airway pressure therapy: a big-data analysis","volume":"2","author":"Bhattacharjee","year":"2020","journal-title":"Lancet Digit. Health"},{"key":"B9","doi-asserted-by":"publisher","first-page":"2344","DOI":"10.1056\/NEJMra1706537","article-title":"Down syndrome","volume":"382","author":"Bull","year":"2020","journal-title":"N. Engl. J. Med"},{"key":"B10","doi-asserted-by":"publisher","first-page":"7010","DOI":"10.1542\/peds.2022-057010","article-title":"Health supervision for children and adolescents with Down syndrome","volume":"149","author":"Bull","year":"2022","journal-title":"Pediatrics"},{"key":"B11","doi-asserted-by":"publisher","first-page":"212","DOI":"10.1016\/j.jpeds.2018.06.031","article-title":"Clinical characteristics and risk factors for developing pulmonary hypertension in children with Down syndrome","volume":"202","author":"Bush","year":"2018","journal-title":"J. Pediat"},{"key":"B12","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0192111","article-title":"Longitudinal changes in clinical characteristics and outcomes for children using long-term non-invasive ventilation","author":"Castro-Codesal","year":"2018","journal-title":"PLoS ONE"},{"key":"B13","doi-asserted-by":"publisher","DOI":"10.1183\/16000617.0098-2016","article-title":"What people with Down syndrome can teach us about cardiopulmonary disease","author":"Colvin","year":""},{"key":"B14","doi-asserted-by":"publisher","first-page":"148","DOI":"10.1016\/j.ijporl.2017.09.017","article-title":"Clinical variables determining the success of adenotonsillectomy in children with Down syndrome","volume":"102","author":"da Rocha","year":"2017","journal-title":"Int. J. Pediatr. Otorhinolaryngol"},{"key":"B15","doi-asserted-by":"publisher","first-page":"L892","DOI":"10.1152\/ajplung.00434.2020","article-title":"Lung disease manifestations in Down syndrome","volume":"321","author":"Danopoulos","year":"2021","journal-title":"Am. J. Physiol. Lung Cell Mol. Physiol"},{"key":"B16","doi-asserted-by":"publisher","first-page":"425","DOI":"10.1161\/CIRCULATIONAHA.122.059706","article-title":"Cardiovascular complications of Down syndrome: Scoping review and expert consensus","volume":"147","author":"Dimopoulos","year":"2023","journal-title":"Circulation"},{"key":"B17","doi-asserted-by":"publisher","first-page":"24","DOI":"10.1002\/ajmg.a.38283","article-title":"Obstructive sleep apnea in Down syndrome: benefits of surgery and noninvasive respiratory support","volume":"24","author":"Dudoignon","year":"2017","journal-title":"Am. J. Med. Genet. A"},{"unstructured":"2022","key":"B18"},{"key":"B19","doi-asserted-by":"publisher","first-page":"377","DOI":"10.1016\/j.jbi.2008.08.010","article-title":"Research electronic data capture (redcap)\u2014A metadata-driven methodology and workflow process for providing translational research informatics support","volume":"42","author":"Harris","year":"2009","journal-title":"J. Biomed. Inform"},{"year":"","key":"B20"},{"key":"B21","doi-asserted-by":"publisher","DOI":"10.3389\/fped.2022.886727","article-title":"Long-term non-invasive ventilation in children with Down syndrome: a systematic review","author":"Hudson","year":"2022","journal-title":"Front. Pediat"},{"year":"2007","author":"Iber","journal-title":"The AASM Manual for the Scoring of Ssleep and Associated Events American Academy of Sleep Medicine","key":"B22"},{"key":"B23","doi-asserted-by":"publisher","first-page":"3013","DOI":"10.1002\/ajmg.a.38399","article-title":"The facial morphology in Down syndrome: a 3D comparison of patients with and without obstructive sleep apnea","volume":"173","author":"Jayaratne","year":"2017","journal-title":"Am. J. Med. Genet. A"},{"key":"B24","doi-asserted-by":"publisher","first-page":"733","DOI":"10.5664\/jcsm.8336","article-title":"Factors related to positive airway pressure therapy adherence in children with obesity and sleep-disordered breathing","volume":"16","author":"Katz","year":"2020","journal-title":"J. Clin. Sleep Med"},{"key":"B25","doi-asserted-by":"publisher","first-page":"18","DOI":"10.1016\/j.sleep.2015.09.014","article-title":"Relationship between obstructive sleep apnea cardiac complications and sleepiness in children with Down syndrome","volume":"17","author":"Konstantinopoulou","year":"2016","journal-title":"Sleep Med"},{"key":"B26","doi-asserted-by":"publisher","first-page":"570","DOI":"10.1378\/chest.14-0266","article-title":"Sleep-disordered breathing in Down syndrome","volume":"147","author":"Lal","year":"2015","journal-title":"Chest"},{"key":"B27","doi-asserted-by":"publisher","first-page":"867","DOI":"10.5664\/jcsm.7126","article-title":"Prevalence of obstructive sleep apnea in children with Down syndrome: a meta-analysis","volume":"14","author":"Lee","year":"2018","journal-title":"J. Clin Sleep Med"},{"key":"B28","doi-asserted-by":"publisher","first-page":"1704","DOI":"10.1002\/ppul.25308","article-title":"Efficacy and adherence of noninvasive ventilation treatment in children with Down syndrome","volume":"56","author":"MacDonagh","year":"2021","journal-title":"Pediatr. Pulmonol"},{"year":"2023","doi-asserted-by":"publisher","key":"B29","DOI":"10.1016\/j.prrv.2023.01.002"},{"key":"B30","doi-asserted-by":"publisher","first-page":"576","DOI":"10.1542\/peds.2012-1671","article-title":"Diagnosis and management of childhood obstructive sleep apnea syndrome","volume":"130","author":"Marcus","year":"2012","journal-title":"Pediatrics"},{"key":"B31","doi-asserted-by":"publisher","first-page":"1235","DOI":"10.1164\/ajrccm\/146.5_Pt_1.1235","article-title":"Normal polysomnographic values for children and adolescents","volume":"146","author":"Marcus","year":"1992","journal-title":"Am. Rev. Respir. Dis"},{"key":"B32","doi-asserted-by":"publisher","first-page":"699","DOI":"10.5665\/sleep.5554","article-title":"Prevalence of obstructive sleep apnea in children with Down syndrome","volume":"39","author":"Maris","year":"2016","journal-title":"Sleep"},{"key":"B33","doi-asserted-by":"publisher","first-page":"331","DOI":"10.1136\/archdischild-2015-310351","article-title":"Outcome of adenotonsillectomy in children with Down syndrome and obstructive sleep apnoea","volume":"102","author":"Maris","year":"2017","journal-title":"Arch. Dis. Child."},{"key":"B34","doi-asserted-by":"publisher","first-page":"1145","DOI":"10.5664\/jcsm.9836","article-title":"Symptom care approach to noninvasive ventilatory support in children with complex neural disability","volume":"18","author":"Morrison","year":"2022","journal-title":"J. Clin. Sleep Med"},{"key":"B35","doi-asserted-by":"publisher","first-page":"500","DOI":"10.1002\/ppul.23569","article-title":"Thirteen years of invasive and noninvasive home ventilation for children in a developing country: a retrospective study","volume":"52","author":"Nathan","year":"2017","journal-title":"Pediat. Pulmonol"},{"key":"B36","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijporl.2020.109968","article-title":"Obstructive sleep apnea in children with Down syndrome\u2014Prevalence and evaluation of surgical treatment","author":"Nerfeldt","year":"2020","journal-title":"Int. J. Pediatr. Otorhinolaryngol"},{"key":"B37","doi-asserted-by":"publisher","first-page":"651","DOI":"10.1093\/sleep\/29.5.651","article-title":"Compliance rates in children using noninvasive continuous positive airway pressure","volume":"29","author":"O'Donnell","year":"2006","journal-title":"Sleep"},{"key":"B38","doi-asserted-by":"publisher","first-page":"1963","DOI":"10.1002\/lary.27063","article-title":"Down syndrome and pediatric obstructive sleep apnea surgery: a national cohort","volume":"128","author":"Ong","year":"2018","journal-title":"Laryngoscope"},{"key":"B39","doi-asserted-by":"publisher","first-page":"2035","DOI":"10.1002\/ppul.24484","article-title":"Understanding adherence to non-invasive ventilation in youth with duchenne muscular dystrophy","volume":"54","author":"Pascoe","year":"2019","journal-title":"Pediatr. Pulmonol"},{"key":"B40","doi-asserted-by":"publisher","first-page":"575","DOI":"10.1007\/s11325-018-01775-2","article-title":"Long-term adherence to ambulatory initiated continuous positive airway pressure in non-syndromic osa children","volume":"23","author":"Perriol","year":"2019","journal-title":"Sleep Breath"},{"key":"B41","doi-asserted-by":"publisher","DOI":"10.1038\/s41598-022-26918-0","article-title":"Evaluation of congenital and acquired heart diseases in a spanish cohort of adults with Down syndrome","author":"Rabes","year":"2022","journal-title":"Sci. Rep"},{"year":"1968","author":"Rechtschaffen","journal-title":"A Manual of Standardized Terminology, Techniques and Scoring System for Sleep Stages of Human Subjects","key":"B42"},{"key":"B43","doi-asserted-by":"publisher","first-page":"241","DOI":"10.1016\/j.ijporl.2009.11.006","article-title":"Effects of adeno-tonsillectomy on polysomnography patterns in Down syndrome children with obstructive sleep apnea: a comparative study with children without Down syndrome","volume":"74","author":"Shete","year":"2010","journal-title":"Int. J. Pediatr. Otorhinolaryngol"},{"key":"B44","doi-asserted-by":"publisher","first-page":"795","DOI":"10.1111\/jch.14488","article-title":"The impact of continuous positive airway pressure on cardiac mechanics: Findings from a meta-analysis of echocardiographic studies","volume":"24","author":"Tadic","year":"2022","journal-title":"J. Clin. Hyperten"},{"key":"B45","doi-asserted-by":"publisher","first-page":"177","DOI":"10.1177\/1055665621998181","article-title":"Three-dimensional assessment of craniofacial features in patients with Down syndrome during the mixed dentition period: a case-control study","volume":"59","author":"Takizawa","year":"2022","journal-title":"Cleft. Palate. Craniofac. J"},{"key":"B46","doi-asserted-by":"publisher","DOI":"10.1016\/j.jns.2020.116765","article-title":"Changes in right-to-left cardiac shunting by continuous positive airway pressure: a word of caution","author":"Tanaka","year":"2020","journal-title":"J. Neurol. Sci"},{"key":"B47","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijporl.2019.109763","article-title":"The spectrum of obstructive sleep apnea in infants and children with Down syndrome","author":"Waters","year":"2020","journal-title":"Int. J. Pediatr. Otorhinolaryngol"}],"container-title":["Frontiers in Sleep"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/frsle.2023.1169236\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,5,22]],"date-time":"2023-05-22T08:33:49Z","timestamp":1684744429000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/frsle.2023.1169236\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,5,19]]},"references-count":47,"alternative-id":["10.3389\/frsle.2023.1169236"],"URL":"https:\/\/doi.org\/10.3389\/frsle.2023.1169236","relation":{},"ISSN":["2813-2890"],"issn-type":[{"type":"electronic","value":"2813-2890"}],"subject":[],"published":{"date-parts":[[2023,5,19]]},"article-number":"1169236"}}