{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,22]],"date-time":"2026-07-22T15:41:32Z","timestamp":1784734892441,"version":"3.55.0"},"reference-count":83,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2022,9,20]],"date-time":"2022-09-20T00:00:00Z","timestamp":1663632000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Neurol."],"abstract":"<jats:p>Rett Syndrome (RTT, OMIM 312750), a unique rare neurodevelopmental disorder, mostly affects females and causes severe multi-disabilities including poor sleep. This meta-analysis systematically reviewed the polysomnographic (PSG) data of individuals with RTT on both sleep macrostructure and sleep respiratory indexes and compared them to literature normative values. Studies were collected from PubMed, Web of Science, PsycINFO, Ebsco, Scopus, and Cochrane Library till 26 April 2022. Across 13 included studies, the 134 selected RTT cases were mostly females being <jats:italic>MECP2<\/jats:italic> (<jats:italic>n<\/jats:italic> = 41) and <jats:italic>CDKL5<\/jats:italic> (<jats:italic>n<\/jats:italic> = 4) positive. They were further stratified by gene, age, and clinical features. Findings of comparison with literature normative values suggested shorter total sleep time (TST) and sleep onset latency (SOL), twice as long wake after sleep onset (WASO) with lower sleep efficiency (SEI) in RTT, as well as increased non-rapid eye movement stage 3 (stage N3) and decreased rapid eye movement sleep. Based on limited data per stratifications, we found in RTT cases &amp;lt;5 years old lower stage N3, and in RTT cases &amp;gt;5 years old less WASO and more WASO in the epileptic strata. However, meta-results generated from studies designed with comparison groups only showed lower stage N1 in RTT than in healthy comparison, together with similar SEI and stage N3 to primary snoring subjects. For sleep respiratory indexes, severe disordered sleep breathing was confirmed across roughly all RTT strata. We are the first study to meta-analyze PSG data of subjects with RTT, illustrating shorter TST and aberrant sleep staging in RTT that may vary with age or the presence of epilepsy. Severe nocturnal hypoxemia with apneic events was also demonstrated. More studies are needed to explore and elucidate the pathophysiological mechanisms of these sleep findings in the future.<\/jats:p><jats:sec><jats:title>Systematic review registration<\/jats:title><jats:p><jats:ext-link>https:\/\/www.crd.york.ac.uk\/prospero\/display_record.php?RecordID=198099<\/jats:ext-link>, identifier: CRD 42020198099.<\/jats:p><\/jats:sec>","DOI":"10.3389\/fneur.2022.963626","type":"journal-article","created":{"date-parts":[[2022,9,20]],"date-time":"2022-09-20T07:14:57Z","timestamp":1663658097000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":4,"title":["A meta-review of standard polysomnography parameters in Rett Syndrome"],"prefix":"10.3389","volume":"13","author":[{"given":"Xin-Yan","family":"Zhang","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Karen","family":"Spruyt","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2022,9,20]]},"reference":[{"key":"B1","doi-asserted-by":"publisher","first-page":"944","DOI":"10.1002\/ana.22124","article-title":"Rett syndrome: revised diagnostic criteria and nomenclature","volume":"68","author":"Neul","year":"2010","journal-title":"Ann Neurol."},{"key":"B2","doi-asserted-by":"publisher","first-page":"372","DOI":"10.1016\/S0387-7604(85)80048-6","article-title":"Rett syndrome: criteria for inclusion and exclusion","volume":"7","author":"Hagberg","year":"1985","journal-title":"Brain Dev."},{"key":"B3","doi-asserted-by":"publisher","first-page":"47","DOI":"10.1002\/ajmg.1320250506","article-title":"Rett syndrome: a suggested staging system for describing impairment profile with increasing age towards adolescence","volume":"1","author":"Hagberg","year":"1986","journal-title":"Am J Med Genet Suppl."},{"key":"B4","doi-asserted-by":"publisher","first-page":"208","DOI":"10.1016\/S0387-7604(01)00193-0","article-title":"Guidelines for reporting clinical features in cases with Mecp2 mutations","volume":"23","author":"Kerr","year":"2001","journal-title":"Brain Dev."},{"key":"B5","doi-asserted-by":"publisher","first-page":"185","DOI":"10.1038\/13810","article-title":"Rett syndrome is caused by mutations in X-linked Mecp2, encoding methyl-Cpg-binding protein 2","volume":"23","author":"Amir","year":"1999","journal-title":"Nat Gen."},{"key":"B6","doi-asserted-by":"publisher","first-page":"89","DOI":"10.1016\/j.ajhg.2008.05.015","article-title":"Foxg1 is responsible for the congenital variant of rett syndrome","volume":"83","author":"Ariani","year":"2008","journal-title":"Am J Hum Genet."},{"key":"B7","doi-asserted-by":"publisher","first-page":"1149","DOI":"10.1086\/426460","article-title":"Mutations in the X-Linked Cyclin-Dependent Kinase-Like 5 (Cdkl5\/Stk9) Gene Are Associated with Severe Neurodevelopmental Retardation","volume":"75","author":"Tao","year":"2004","journal-title":"Am J Hum Genet."},{"key":"B8","doi-asserted-by":"publisher","first-page":"1079","DOI":"10.1086\/426462","article-title":"Mutations of Cdkl5 cause a severe neurodevelopmental disorder with infantile spasms and mental retardation","volume":"75","author":"Weaving","year":"2004","journal-title":"Am J Hum Genet."},{"key":"B9","doi-asserted-by":"publisher","first-page":"293","DOI":"10.1053\/ejpn.2002.0612","article-title":"An update on clinically applicable diagnostic criteria in rett syndrome. 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