{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,30]],"date-time":"2026-09-30T17:55:00Z","timestamp":1790790900343,"version":"4.1.0"},"reference-count":25,"publisher":"BMJ","issue":"2","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["Arch Dis Child"],"published-print":{"date-parts":[[2019,2]]},"abstract":"\n Objective<\/jats:title>\n To test the hypothesis that children with Prader-Willi syndrome (PWS) and obstructive sleep apnoea syndrome (OSAS) have hypercapnia for higher proportion of total sleep time (TST) than non-syndromic children with similar obstructive apnoea\u2013hypopnoea index (OAHI).<\/jats:p>\n <\/jats:sec>\n \n Design<\/jats:title>\n Cross-sectional study.<\/jats:p>\n <\/jats:sec>\n \n Setting<\/jats:title>\n Two tertiary care hospitals.<\/jats:p>\n <\/jats:sec>\n \n Patients<\/jats:title>\n Patients with PWS and non-syndromic children with snoring who underwent polygraphy and were of similar age, body mass index (BMI) z-score and OAHI.<\/jats:p>\n <\/jats:sec>\n \n Main outcome measure<\/jats:title>\n \n The two groups were compared regarding %TST with transcutaneous CO\n 2<\/jats:sub>\n (PtcCO\n 2<\/jats:sub>\n ) >50\u2009mm Hg. The interaction between PWS diagnosis and OSAS severity (OAHI <1\u2009episode\/h vs 1\u20135 episodes\/h vs >5\u2009episodes\/h) regarding %TST with PtcCO\n 2<\/jats:sub>\n >50\u2009mm Hg was tested using multiple linear regression.\n <\/jats:p>\n <\/jats:sec>\n \n Results<\/jats:title>\n \n 48 children with PWS and 92 controls were included (median age 2.3 (range 0.2\u201314.1) years vs 2.2 (0.3\u201315.1) years; BMI z-score 0.7\u00b11.9 vs 0.8\u00b11.7; median OAHI 0.5 (0\u201329.5) episodes\/h vs 0.5 (0\u201333.9) episodes\/h; p>0.05). The two groups did not differ in %TST with PtcCO\n 2<\/jats:sub>\n >50\u2009mm Hg (median 0% (0\u2013100%) vs 0% (0\u201381.3%), respectively; p>0.05). However, the interaction between PWS and OSAS severity with respect to duration of hypoventilation was significant (p<0.01); the estimated mean differences of %TST with PtcCO\n 2<\/jats:sub>\n >50\u2009mm Hg between children with PWS and controls for OAHI <1\u2009episode\/h, 1\u20135 episodes\/h and >5\u2009episodes\/h were +0.2%, +1%\u2009and +33%, respectively.\n <\/jats:p>\n <\/jats:sec>\n \n Conclusion<\/jats:title>\n Increasing severity of upper airway obstruction during sleep in children with PWS is accompanied by disproportionately longer periods of hypoventilation when compared with non-syndromic children with similar frequency of obstructive events.<\/jats:p>\n <\/jats:sec>","DOI":"10.1136\/archdischild-2017-314282","type":"journal-article","created":{"date-parts":[[2018,7,14]],"date-time":"2018-07-14T12:26:46Z","timestamp":1531571206000},"page":"166-171","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":30,"title":["Hypoventilation disproportionate to OSAS severity in children with Prader-Willi syndrome"],"prefix":"10.1136","volume":"104","author":[{"given":"Francois","family":"Abel","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hui-Leng","family":"Tan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Valentina","family":"Negro","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nicola","family":"Bridges","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Thomas","family":"Carlisle","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elaine","family":"Chan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aidan","family":"Laverty","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael","family":"Miligkos","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Martin","family":"Samuels","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3592-0037","authenticated-orcid":false,"given":"Athanasios G","family":"Kaditis","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"239","published-online":{"date-parts":[[2018,7,14]]},"reference":[{"key":"2020060122150658000_104.2.166.1","doi-asserted-by":"crossref","first-page":"403","DOI":"10.5664\/jcsm.3616","article-title":"Prader Willi syndrome and obstructive sleep apnea: co-occurrence in the pediatric population","volume":"10","author":"Sedky","year":"2014","journal-title":"J Clin Sleep Med"},{"key":"2020060122150658000_104.2.166.2","doi-asserted-by":"publisher","DOI":"10.1111\/jpc.12109"},{"key":"2020060122150658000_104.2.166.3","first-page":"52","article-title":"Respiratory complications in children with Prader Willi syndrome","volume":"22","author":"Tan","year":"2017","journal-title":"Paediatr Respir Rev"},{"key":"2020060122150658000_104.2.166.4","doi-asserted-by":"publisher","DOI":"10.1136\/archdischild-2012-303441"},{"key":"2020060122150658000_104.2.166.5","doi-asserted-by":"crossref","unstructured":"Cohen M , Hamilton J , Narang I . 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