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It is unclear whether obstructive apneas are part of the syndrome.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Objectives<\/jats:title>\n                    <jats:p>To determine if Phox2b27Ala\/+ mice, which present the main symptoms of CCHS and die within hours after birth, also express obstructive apneas, and to investigate potential underlying mechanisms.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>Apneas were classified as central, obstructive, or mixed by using a novel system combining pneumotachography and laser detection of abdominal movement immediately after birth. Several respiratory nuclei involved in airway patency were examined by immunohistochemistry and electrophysiology in brainstem\u2013spinal cord preparations.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Measurements and Main Results<\/jats:title>\n                    <jats:p>The median (interquartile range) of obstructive apnea frequency was 2.3 (1.5\u20133.3)\/min in Phox2b27Ala\/+ pups versus 0.6 (0.4\u20131.0)\/min in wild types (P\u2009&amp;lt;\u20090.0001). Obstructive apnea duration was 2.7 seconds (2.3\u20133.9) in Phox2b27Ala\/+ pups versus 1.7 seconds (1.1\u20131.9) in wild types (P\u2009&amp;lt;\u20090.0001). Central and mixed apneas presented similar significant differences. In Phox2b27Ala\/+ preparations, the hypoglossal nucleus had fewer (P\u2009&amp;lt;\u20090.05) and smaller (P\u2009&amp;lt;\u20090.01) neurons, compared with wild-type preparations. Importantly, coordination of phrenic and hypoglossal motor activities was disrupted, as evidenced by the longer and variable delay of hypoglossal activity with respect to phrenic activity onset (P\u2009&amp;lt;\u20090.001).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>The Phox2b27Ala\/+ mutation predisposed pups not only to hypoventilation and central apneas, but also to obstructive and mixed apneas, likely because of hypoglossal dysgenesis. These results thus demand attention toward obstructive events in infants with CCHS.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1164\/rccm.202104-0887oc","type":"journal-article","created":{"date-parts":[[2021,9,3]],"date-time":"2021-09-03T12:38:20Z","timestamp":1630672700000},"page":"1200-1210","update-policy":"https:\/\/doi.org\/10.1164\/crossmarkpolicies","source":"Crossref","is-referenced-by-count":27,"title":["Obstructive Apneas in a Mouse Model of Congenital Central Hypoventilation Syndrome"],"prefix":"10.1093","volume":"204","author":[{"given":"Am\u00e9lia","family":"Madani","sequence":"first","affiliation":[{"name":"NeuroDiderot, FHU I2-D2, Universit\u00e9 de Paris, Inserm, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gabriel","family":"Pitollat","sequence":"additional","affiliation":[{"name":"Institut de Neurosciences Cognitives et Int\u00e9gratives d\u2019Aquitaine, UMR5287, Universit\u00e9 de Bordeaux, CNRS, Bordeaux, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"El\u00e9onore","family":"Sizun","sequence":"additional","affiliation":[{"name":"NeuroDiderot, FHU I2-D2, Universit\u00e9 de Paris, Inserm, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laura","family":"Cardoit","sequence":"additional","affiliation":[{"name":"Institut de Neurosciences Cognitives et Int\u00e9gratives d\u2019Aquitaine, UMR5287, Universit\u00e9 de Bordeaux, CNRS, Bordeaux, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maud","family":"Ringot","sequence":"additional","affiliation":[{"name":"NeuroDiderot, FHU I2-D2, Universit\u00e9 de Paris, Inserm, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Thomas","family":"Bourgeois","sequence":"additional","affiliation":[{"name":"NeuroDiderot, FHU I2-D2, Universit\u00e9 de Paris, Inserm, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nelina","family":"Ramanantsoa","sequence":"additional","affiliation":[{"name":"NeuroDiderot, FHU I2-D2, Universit\u00e9 de Paris, Inserm, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christophe","family":"Delclaux","sequence":"additional","affiliation":[{"name":"NeuroDiderot, FHU I2-D2, Universit\u00e9 de Paris, Inserm, Paris, France"},{"name":"Service d\u2019Explorations Fonctionnelles P\u00e9diatriques and"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"St\u00e9phane","family":"Dauger","sequence":"additional","affiliation":[{"name":"NeuroDiderot, FHU I2-D2, Universit\u00e9 de Paris, Inserm, Paris, France"},{"name":"Service de M\u00e9decine Intensive-R\u00e9animation P\u00e9diatriques, H\u00f4pital Robert Debr\u00e9, AP-HP, Paris, France; and"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marie-Pia","family":"d\u2019Ortho","sequence":"additional","affiliation":[{"name":"NeuroDiderot, FHU I2-D2, Universit\u00e9 de Paris, Inserm, Paris, France"},{"name":"Service de Physiologie-Explorations Fonctionnelles, H\u00f4pital Bichat, AP-HP, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3214-1724","authenticated-orcid":false,"given":"Muriel","family":"Thoby-Brisson","sequence":"additional","affiliation":[{"name":"Institut de Neurosciences Cognitives et Int\u00e9gratives d\u2019Aquitaine, UMR5287, Universit\u00e9 de Bordeaux, CNRS, Bordeaux, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4291-472X","authenticated-orcid":false,"given":"Jorge","family":"Gallego","sequence":"additional","affiliation":[{"name":"NeuroDiderot, FHU I2-D2, Universit\u00e9 de Paris, Inserm, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7845-9618","authenticated-orcid":false,"given":"Boris","family":"Matrot","sequence":"additional","affiliation":[{"name":"NeuroDiderot, FHU I2-D2, Universit\u00e9 de Paris, Inserm, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2021,11,15]]},"reference":[{"key":"2026030321560011200_bib1","doi-asserted-by":"crossref","first-page":"139","DOI":"10.2174\/1573396315666190621103954","article-title":"Neonatal congenital central hypoventilation syndrome: why we should not sleep on it. 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