{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"institution":[{"name":"bioRxiv"}],"indexed":{"date-parts":[[2026,7,1]],"date-time":"2026-07-01T21:07:01Z","timestamp":1782940021871,"version":"3.54.5"},"posted":{"date-parts":[[2023,7,18]]},"group-title":"Neuroscience","reference-count":116,"publisher":"openRxiv","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"accepted":{"date-parts":[[2023,7,18]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                <jats:p>\n                  Fragile X syndrome (FXS) is a highly-prevalent genetic cause of intellectual disability, associated with disrupted cognition and sleep abnormalities. Sleep loss itself negatively impacts cognitive function, yet the contribution of sleep loss to impaired cognition in FXS is vastly understudied. One untested possibility is that disrupted cognition in FXS is exacerbated by abnormal sleep. We hypothesized that restoration of sleep-dependent mechanisms could improve functions such as memory consolidation in FXS. We examined whether administration of ML297, a hypnotic drug acting on G-protein-activated inward-rectifying potassium channels, could restore sleep phenotypes and improve disrupted memory consolidation in\n                  <jats:italic>\n                    Fmr1\n                    <jats:sup>-\/y<\/jats:sup>\n                  <\/jats:italic>\n                  mice. Using 24-h polysomnographic recordings, we found that\n                  <jats:italic>\n                    Fmr1\n                    <jats:sup>-\/y<\/jats:sup>\n                  <\/jats:italic>\n                  mice exhibit reduced non-rapid eye movement (NREM) sleep and fragmented NREM sleep architecture, alterations in NREM EEG spectral power (including reductions in sleep spindles), and reduced EEG coherence between cortical areas. These alterations were reversed in the hours following ML297 administration. Hypnotic treatment following contextual fear or spatial learning also ameliorated disrupted memory consolidation in\n                  <jats:italic>\n                    Fmr1\n                    <jats:sup>-\/y<\/jats:sup>\n                  <\/jats:italic>\n                  mice. Hippocampal activation patterns during memory recall was altered in\n                  <jats:italic>\n                    Fmr1\n                    <jats:sup>-\/y<\/jats:sup>\n                  <\/jats:italic>\n                  mice, reflecting an altered balance of activity among principal neurons vs. parvalbumin-expressing (PV+) interneurons. This phenotype was partially reversed by post-learning ML297 administration. These studies suggest that sleep disruption could have a major impact on neurophysiological and behavioral phenotypes in FXS, and that hypnotic therapy may significantly improve disrupted cognition in this disorder.\n                <\/jats:p>","DOI":"10.1101\/2023.07.14.549070","type":"posted-content","created":{"date-parts":[[2023,7,18]],"date-time":"2023-07-18T12:55:55Z","timestamp":1689684955000},"source":"Crossref","is-referenced-by-count":3,"title":["Hypnotic treatment reverses NREM sleep disruption and EEG desynchronization in a mouse model of Fragile X syndrome to rescue memory consolidation deficits"],"prefix":"10.64898","author":[{"given":"Jessy D.","family":"Martinez","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lydia G.","family":"Wilson","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"William P.","family":"Brancaleone","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kathryn G.","family":"Peterson","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Donald S.","family":"Popke","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Valentina Caicedo","family":"Garzon","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Roxanne E.","family":"Perez Tremble","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marcus J.","family":"Donnelly","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stephany L.","family":"Mendez Ortega","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Daniel","family":"Torres","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"James J.","family":"Shaver","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Brittany C.","family":"Clawson","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sha","family":"Jiang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zhongying","family":"Yang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sara J.","family":"Aton","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"54368","reference":[{"key":"2023072023200892000_2023.07.14.549070v1.1","doi-asserted-by":"publisher","DOI":"10.1097\/00004703-200112000-00008"},{"key":"2023072023200892000_2023.07.14.549070v1.2","first-page":"41","volume-title":"Frontiers in Molecular Neuroscience","volume":"11","year":"2018"},{"key":"2023072023200892000_2023.07.14.549070v1.3","doi-asserted-by":"crossref","first-page":"744","DOI":"10.1542\/peds.95.5.744","article-title":"Specification of the neurobehavioral phenotype in males with fragile X syndrome","volume":"95","year":"1995","journal-title":"Pediatrics"},{"key":"2023072023200892000_2023.07.14.549070v1.4","doi-asserted-by":"publisher","DOI":"10.1038\/ejhg.2008.61"},{"key":"2023072023200892000_2023.07.14.549070v1.5","doi-asserted-by":"publisher","DOI":"10.1352\/1944-7558-116.1.16"},{"key":"2023072023200892000_2023.07.14.549070v1.6","doi-asserted-by":"crossref","first-page":"679","DOI":"10.1093\/sleep\/33.5.679","article-title":"Prevalence, nature, and correlates of sleep problems among children with fragile X syndrome based on a large scale parent survey","volume":"33","year":"2010","journal-title":"Sleep"},{"key":"2023072023200892000_2023.07.14.549070v1.7","doi-asserted-by":"publisher","DOI":"10.1352\/1944-7588-114.6.383"},{"key":"2023072023200892000_2023.07.14.549070v1.8","doi-asserted-by":"publisher","DOI":"10.1542\/peds.2013-4301"},{"key":"2023072023200892000_2023.07.14.549070v1.9","doi-asserted-by":"publisher","DOI":"10.1017\/S0012162205000186"},{"key":"2023072023200892000_2023.07.14.549070v1.10","unstructured":"Colten, H. 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