{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,29]],"date-time":"2025-10-29T06:28:24Z","timestamp":1761719304783,"version":"3.41.2"},"reference-count":74,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2024,7,24]],"date-time":"2024-07-24T00:00:00Z","timestamp":1721779200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100005969","name":"Universit\u00e0 di Bologna","doi-asserted-by":"publisher","award":["RFO 2020\u20102022"],"award-info":[{"award-number":["RFO 2020\u20102022"]}],"id":[{"id":"10.13039\/501100005969","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Journal of Sleep Research"],"published-print":{"date-parts":[[2025,4]]},"abstract":"<jats:title>Summary<\/jats:title><jats:p>CDKL5 deficiency disorder is a rare genetic disease caused by mutations in the <jats:italic>CDKL5<\/jats:italic> gene. Central apneas during wakefulness have been reported in patients with CDKL5 deficiency disorder. Studies on CDKL5\u2010knockout mice, a CDKL5 deficiency disorder model, reported sleep apneas, but it is still unclear whether these events are central (central sleep apnea) or obstructive (obstructive sleep apnea) and may be related to alterations of brain circuits that modulate breathing rhythm. This study aimed to discriminate central sleep apnea and obstructive sleep apnea in CDKL5\u2010knockout mice, and explore changes in the somatostatin neurons expressing high levels of neurokinin\u20101 receptors within the preB\u00f6tzinger complex. Ten adult male wild\u2010type and 12 CDKL5\u2010knockout mice underwent electrode implantation for sleep stage discrimination and diaphragmatic activity recording, and were studied using whole\u2010body plethysmography for 7\u2009hr during the light (resting) period. Sleep apneas were categorised as central sleep apnea or obstructive sleep apnea based on the recorded signals. The number of somatostatin neurons in the preB\u00f6tzinger complex and their neurokinin\u20101 receptors expression were assessed through immunohistochemistry in a sub\u2010group of animals. CDKL5\u2010knockout mice exhibited a higher apnea occurrence rate and a greater prevalence of obstructive sleep apnea during rapid eye movement sleep, compared with wild\u2010type, whereas no significant difference was observed for central sleep apnea. Moreover, CDKL5\u2010knockout mice showed a reduced number of somatostatin neurons in the preB\u00f6tzinger complex, and these neurons expressed a lower level of neurokinin\u20101 receptors compared with wild\u2010type controls. These findings underscore the pivotal role of CDKL5 in regulating normal breathing, suggesting its potential involvement in shaping preB\u00f6tzinger complex neural circuitry and controlling respiratory muscles during sleep.<\/jats:p>","DOI":"10.1111\/jsr.14295","type":"journal-article","created":{"date-parts":[[2024,7,25]],"date-time":"2024-07-25T04:55:29Z","timestamp":1721883329000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Characterisation of sleep apneas and respiratory circuitry in mice lacking <scp>CDKL5<\/scp>"],"prefix":"10.1111","volume":"34","author":[{"given":"Gabriele","family":"Matteoli","sequence":"first","affiliation":[{"name":"Department of Biomedical and Neuromotor Sciences Alma Mater Studiorum \u2010 University of Bologna  Bologna Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sara","family":"Alvente","sequence":"additional","affiliation":[{"name":"Department of 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