{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,7]],"date-time":"2026-10-07T18:39:01Z","timestamp":1791398341468,"version":"4.3.3"},"reference-count":39,"publisher":"Oxford University Press (OUP)","issue":"2","license":[{"start":{"date-parts":[[2005,7,1]],"date-time":"2005-07-01T00:00:00Z","timestamp":1120176000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/pages\/standard-publication-reuse-rights"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2005,7,15]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Rationale: Central congenital hypoventilation syndrome (CCHS) is a rare autosomal dominant syndrome present from birth, and characterized by depressed ventilation during sleep. Heterozygous mutations of the homeobox gene Phox2b were recently found in a very high proportion of patients. Objectives: To determine whether newborn mice with heterozygous targeted deletion of the transcription factor Phox2b would display sleep-disordered breathing. Methods: We measured breathing pattern using whole-body plethysmography in wild-type and mutant 5-day-old mice, and we classified sleep\u2013wake states using nuchal EMG and behavioral scores. Results: We found that sleep apnea total time was approximately six times longer (8.9 \u00b1 12 vs. 1.5 \u00b1 2.2 seconds, p &amp;lt; 0.0015), and ventilation during active sleep was 21% lower (18.4 \u00b1 5.1 vs. 23.3 \u00b1 5.5 ml\/g\/second, p &amp;lt; 0.006) in mutant than in wild-type pups. During wakefulness, apnea time and ventilation were not significantly different between mutant and wild-type pups. Mutant and wild-type pups showed highly similar sleep\u2013wake states. Conclusion: Although their respiratory phenotype was much less severe than CCHS, the Phox2b+\/\u2212 mutant mice showed sleep-disordered breathing, which partially modeled the key feature of CCHS.<\/jats:p>","DOI":"10.1164\/rccm.200411-1528oc","type":"journal-article","created":{"date-parts":[[2005,4,28]],"date-time":"2005-04-28T20:35:17Z","timestamp":1114720517000},"page":"238-243","source":"Crossref","is-referenced-by-count":66,"title":["Sleep-disordered Breathing in Newborn Mice Heterozygous for the Transcription Factor Phox2b"],"prefix":"10.1093","volume":"172","author":[{"given":"Estelle","family":"Durand","sequence":"first","affiliation":[{"name":"INSERM U676, and Service de Re\u0301animation, Ho\u0302pital Robert-Debre\u0301; and CNRS UMR 8542, Ecole Normale Supe\u0301rieure, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ste\u0301phane","family":"Dauger","sequence":"additional","affiliation":[{"name":"INSERM U676, and Service de 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France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jorge","family":"Gallego","sequence":"additional","affiliation":[{"name":"INSERM U676, and Service de Re\u0301animation, Ho\u0302pital Robert-Debre\u0301; and CNRS UMR 8542, Ecole Normale Supe\u0301rieure, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2005,7,15]]},"reference":[{"key":"2026032606004153800_BIB1","first-page":"273","volume-title":"Pediatr Pulmonol","year":"1998"},{"key":"2026032606004153800_BIB2","first-page":"267","volume-title":"Am J Med Genet","year":"2003"},{"key":"2026032606004153800_BIB3","first-page":"16","volume-title":"Am J Respir Critl Care Med","year":"2004"},{"key":"2026032606004153800_BIB4","first-page":"953","volume-title":"Pediatr Res","year":"2004"},{"key":"2026032606004153800_BIB5","first-page":"958","volume-title":"J Appl 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