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Hypoventilation disproportionate to OSAS severity in children with Prader-Willi syndrome
dc.creator | Abel F., Tan H.-L., Negro V., Bridges N., Carlisle T., Chan E., Laverty A., Miligkos M., Samuels M., Kaditis A.G. | en |
dc.date.accessioned | 2023-01-31T07:30:21Z | |
dc.date.available | 2023-01-31T07:30:21Z | |
dc.date.issued | 2019 | |
dc.identifier | 10.1136/archdischild-2017-314282 | |
dc.identifier.issn | 00039888 | |
dc.identifier.uri | http://hdl.handle.net/11615/70251 | |
dc.description.abstract | Objective 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-hypopnoea index (OAHI). Design Cross-sectional study. Setting Two tertiary care hospitals. Patients 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. Main outcome measure The two groups were compared regarding %TST with transcutaneous CO 2 (PtcCO 2) >50 mm Hg. The interaction between PWS diagnosis and OSAS severity (OAHI <1 episode/h vs 1-5 episodes/h vs >5 episodes/h) regarding %TST with PtcCO 2 >50 mm Hg was tested using multiple linear regression. Results 48 children with PWS and 92 controls were included (median age 2.3 (range 0.2-14.1) years vs 2.2 (0.3-15.1) years; BMI z-score 0.7±1.9 vs 0.8±1.7; median OAHI 0.5 (0-29.5) episodes/h vs 0.5 (0-33.9) episodes/h; p>0.05). The two groups did not differ in %TST with PtcCO 2 >50 mm Hg (median 0% (0-100%) vs 0% (0-81.3%), 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 2 >50 mm Hg between children with PWS and controls for OAHI <1 episode/h, 1-5 episodes/h and >5 episodes/h were +0.2%, +1% and +33%, respectively. Conclusion 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. © Author(s) (or their employer(s)) 2019. No commercial re-use. See rights and permissions. Published by BMJ. | en |
dc.language.iso | en | en |
dc.source | Archives of Disease in Childhood | en |
dc.source.uri | https://www.scopus.com/inward/record.uri?eid=2-s2.0-85050219738&doi=10.1136%2farchdischild-2017-314282&partnerID=40&md5=8bea8b97e885260ed43393f199a38b9f | |
dc.subject | mercury | en |
dc.subject | carbon dioxide | en |
dc.subject | adenotonsillectomy | en |
dc.subject | apnea hypopnea index | en |
dc.subject | apnea index | en |
dc.subject | Article | en |
dc.subject | body mass | en |
dc.subject | capnometry | en |
dc.subject | carbon dioxide tension | en |
dc.subject | child | en |
dc.subject | childhood disease | en |
dc.subject | cross-sectional study | en |
dc.subject | disease severity | en |
dc.subject | female | en |
dc.subject | human | en |
dc.subject | hypercapnia | en |
dc.subject | hypoventilation | en |
dc.subject | major clinical study | en |
dc.subject | male | en |
dc.subject | multicenter study | en |
dc.subject | obesity | en |
dc.subject | Obstructive Apnoea Hypopnoea Index | en |
dc.subject | outcome assessment | en |
dc.subject | positive end expiratory pressure | en |
dc.subject | Prader Willi syndrome | en |
dc.subject | preschool child | en |
dc.subject | prevalence | en |
dc.subject | priority journal | en |
dc.subject | retrospective study | en |
dc.subject | sleep disordered breathing | en |
dc.subject | sleep time | en |
dc.subject | snoring | en |
dc.subject | tertiary care center | en |
dc.subject | upper respiratory tract obstruction | en |
dc.subject | adolescent | en |
dc.subject | blood | en |
dc.subject | case control study | en |
dc.subject | complication | en |
dc.subject | hypoventilation | en |
dc.subject | physiologic monitoring | en |
dc.subject | Prader Willi syndrome | en |
dc.subject | severity of illness index | en |
dc.subject | sleep disordered breathing | en |
dc.subject | Adolescent | en |
dc.subject | Carbon Dioxide | en |
dc.subject | Case-Control Studies | en |
dc.subject | Child | en |
dc.subject | Child, Preschool | en |
dc.subject | Cross-Sectional Studies | en |
dc.subject | Female | en |
dc.subject | Humans | en |
dc.subject | Hypoventilation | en |
dc.subject | Male | en |
dc.subject | Monitoring, Physiologic | en |
dc.subject | Prader-Willi Syndrome | en |
dc.subject | Retrospective Studies | en |
dc.subject | Severity of Illness Index | en |
dc.subject | Sleep Apnea, Obstructive | en |
dc.subject | BMJ Publishing Group | en |
dc.title | Hypoventilation disproportionate to OSAS severity in children with Prader-Willi syndrome | en |
dc.type | journalArticle | en |
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