{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,30]],"date-time":"2026-09-30T17:54:59Z","timestamp":1790790899290,"version":"4.1.0"},"reference-count":37,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2024,1,5]],"date-time":"2024-01-05T00:00:00Z","timestamp":1704412800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Pediatric Pulmonology"],"published-print":{"date-parts":[[2024,4]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Objectives<\/jats:title>\n                    <jats:p>Few data on alveolar hypoventilation in Prader\u2010Willi syndrome (PWS) are available and the respiratory follow\u2010up of these patients is not standardized. The objectives of this study were to evaluate the prevalence of alveolar hypoventilation in children with PWS and identify potential risk factors.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Study Design<\/jats:title>\n                    <jats:p>This retrospective study included children with PWS recorded by polysomnography (PSG) with transcutaneous carbon dioxide pressure (PtcCO2) or end\u2010tidal CO2 (ETCO2) measurements, between 2007 and 2021, in a tertiary hospital center. The primary outcome was the presence of alveolar hypoventilation defined as partial pressure of carbon dioxide (pCO2)\u2009\u2265\u200950\u2009mmHg during \u22652% of total sleep time (TST) or more than five consecutive minutes.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Among the 57 included children (38 boys, median age 4.8 years, range 0.1\u201315.6, 60% treated with growth hormone [GH], 37% obese), 19 (33%) had moderate\u2010to\u2010severe obstructive sleep apnea syndrome (defined as obstructive apnea\u2010hypopnea index \u22655\/h) and 20 (35%) had hypoventilation. The median (range) pCO2 max was 49\u2009mmHg (38\u201369). Among the children with hypoventilation, 25% were asymptomatic. Median age and GH treatment were significantly higher in children with hypoventilation compared to those without. There was no significant difference in terms of sex, BMI, obstructive or central apnea\u2010hypopnea index between both groups.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>The frequency of alveolar hypoventilation in children and adolescents with PWS is of concern and may increase with age and GH treatment. A regular screening by oximetry\u2010capnography appears to be indicated whatever the sex, BMI, and rate of obstructive or central apneas.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/ppul.26852","type":"journal-article","created":{"date-parts":[[2024,1,5]],"date-time":"2024-01-05T05:35:08Z","timestamp":1704432908000},"page":"938-948","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":4,"title":["Hypoventilation in patients with Prader\u2010Willi syndrome across the pediatric age"],"prefix":"10.1002","volume":"59","author":[{"ORCID":"https:\/\/orcid.org\/0009-0003-3354-5760","authenticated-orcid":false,"given":"Catherine","family":"Chen","sequence":"first","affiliation":[{"name":"Service de pneumologie p\u00e9diatrique H\u00f4pital Femme M\u00e8re Enfant, Hospices, Civils de Lyon Bron France"},{"name":"Universit\u00e9 Claude\u2010Bernard Lyon 1 Lyon France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4180-9805","authenticated-orcid":false,"given":"Iulia","family":"Ioan","sequence":"additional","affiliation":[{"name":"Service d'explorations fonctionnelles p\u00e9diatriques, H\u00f4pital d'Enfants, CHRU de Nancy; Unit\u00e9 DevAH Universit\u00e9 de Lorraine Nancy France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marine","family":"Thieux","sequence":"additional","affiliation":[{"name":"Universit\u00e9 Claude\u2010Bernard Lyon 1 Lyon France"},{"name":"Service \u00c9pilepsie\u2010Sommeil\u2010Explorations, Fonctionnelles Neurologiques P\u00e9diatriques H\u00f4pital Femme M\u00e8re Enfant, Hospices Civils de, Lyon Bron France"},{"name":"Unit\u00e9 INSERM U1028 CNRS UMR 5292 Universit\u00e9 Lyon 1 Lyon France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marc","family":"Nicolino","sequence":"additional","affiliation":[{"name":"Universit\u00e9 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