{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,10]],"date-time":"2026-06-10T04:02:41Z","timestamp":1781064161561,"version":"3.54.1"},"reference-count":86,"publisher":"Elsevier BV","license":[{"start":{"date-parts":[[2026,6,1]],"date-time":"2026-06-01T00:00:00Z","timestamp":1780272000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2026,6,1]],"date-time":"2026-06-01T00:00:00Z","timestamp":1780272000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2026,4,15]],"date-time":"2026-04-15T00:00:00Z","timestamp":1776211200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100001819","name":"International Rett Syndrome Foundation","doi-asserted-by":"publisher","award":["3511"],"award-info":[{"award-number":["3511"]}],"id":[{"id":"10.13039\/100001819","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000065","name":"National Institute of Neurological Disorders and Stroke","doi-asserted-by":"publisher","award":["R01NS112171"],"award-info":[{"award-number":["R01NS112171"]}],"id":[{"id":"10.13039\/100000065","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100007086","name":"Pitt Hopkins Research Foundation","doi-asserted-by":"publisher","award":["23"],"award-info":[{"award-number":["23"]}],"id":[{"id":"10.13039\/100007086","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000025","name":"National Institute of Mental Health","doi-asserted-by":"publisher","award":["K01MH112983"],"award-info":[{"award-number":["K01MH112983"]}],"id":[{"id":"10.13039\/100000025","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000025","name":"National Institute of Mental Health","doi-asserted-by":"publisher","award":["F31MH119699"],"award-info":[{"award-number":["F31MH119699"]}],"id":[{"id":"10.13039\/100000025","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000057","name":"NIH NIGMS","doi-asserted-by":"publisher","award":["T32 GM007628"],"award-info":[{"award-number":["T32 GM007628"]}],"id":[{"id":"10.13039\/100000057","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["Molecular and Cellular Neuroscience"],"published-print":{"date-parts":[[2026,6]]},"DOI":"10.1016\/j.mcn.2026.104094","type":"journal-article","created":{"date-parts":[[2026,4,15]],"date-time":"2026-04-15T20:32:49Z","timestamp":1776285169000},"page":"104094","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":0,"special_numbering":"C","title":["Increasing MeCP2 protein in Pitt-Hopkins syndrome model (Tcf4+\/\u2212) mice does not affect abnormal myelination but induces the generation of astrocytes"],"prefix":"10.1016","volume":"137","author":[{"given":"Sheryl Anne D.","family":"Vermudez","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Geanne A.","family":"Freitas","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aditi","family":"Buch","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yuta","family":"Moxley","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hemangi","family":"Rajpal","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rocco G.","family":"Gogliotti","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Colleen M.","family":"Niswender","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"issue":"5","key":"10.1016\/j.mcn.2026.104094_bb3000","doi-asserted-by":"crossref","first-page":"668","DOI":"10.1002\/mus.27187","article-title":"Safety and efficacy of nusinersen in spinal muscular atrophy: the EMBRACE study","volume":"63","author":"Acsadi","year":"2021","journal-title":"Muscle Nerve"},{"issue":"3","key":"10.1016\/j.mcn.2026.104094_bb0005","doi-asserted-by":"crossref","DOI":"10.1016\/j.isci.2024.109296","article-title":"Mecp2 knock-out astrocytes affect synaptogenesis by interleukin 6 dependent mechanisms","volume":"27","author":"Albizzati","year":"2024","journal-title":"iScience"},{"issue":"5","key":"10.1016\/j.mcn.2026.104094_bb0010","doi-asserted-by":"crossref","first-page":"988","DOI":"10.1086\/515582","article-title":"Mutations in TCF4, encoding a class I basic helix-loop-helix transcription factor, are responsible for Pitt-Hopkins syndrome, a severe epileptic encephalopathy associated with autonomic dysfunction","volume":"80","author":"Amiel","year":"2007","journal-title":"Am. J. Hum. Genet."},{"issue":"2","key":"10.1016\/j.mcn.2026.104094_bb0015","doi-asserted-by":"crossref","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. Genet."},{"issue":"8","key":"10.1016\/j.mcn.2026.104094_bb0020","doi-asserted-by":"crossref","first-page":"3331","DOI":"10.1093\/brain\/awad057","article-title":"Promyelinating drugs promote functional recovery in an autism spectrum disorder mouse model of Pitt-Hopkins syndrome","volume":"146","author":"Bohlen","year":"2023","journal-title":"Brain"},{"key":"10.1016\/j.mcn.2026.104094_bb3020","doi-asserted-by":"crossref","first-page":"33","DOI":"10.1016\/j.biopsych.2010.03.015","article-title":"Cognitive and sensorimotor gating impairments in transgenic mice overexpressing the schizophrenia susceptibility gene Tcf4 in the brain","volume":"68","author":"Brz\u00f3zka","year":"2010","journal-title":"Biol. Psychiatry"},{"issue":"11","key":"10.1016\/j.mcn.2026.104094_bb0025","doi-asserted-by":"crossref","first-page":"3015","DOI":"10.1007\/s00221-018-5415-z","article-title":"Role of astrocytic MeCP2 in regulation of CNS myelination by affecting oligodendrocyte and neuronal physiology and axo-glial interactions","volume":"236","author":"Buch","year":"2018","journal-title":"Exp. Brain Res."},{"issue":"10","key":"10.1016\/j.mcn.2026.104094_bb0030","doi-asserted-by":"crossref","first-page":"1887","DOI":"10.1242\/dev.02847","article-title":"A crucial role for Olig2 in white matter astrocyte development","volume":"134","author":"Cai","year":"2007","journal-title":"Development"},{"key":"10.1016\/j.mcn.2026.104094_bb0035","doi-asserted-by":"crossref","first-page":"94","DOI":"10.3389\/fnmol.2016.00094","article-title":"Tcf4 controls neuronal migration of the cerebral cortex through regulation of Bmp7","volume":"9","author":"Chen","year":"2016","journal-title":"Front. Mol. Neurosci."},{"key":"10.1016\/j.mcn.2026.104094_bb0040","doi-asserted-by":"crossref","first-page":"2813","DOI":"10.2147\/NDT.S371483","article-title":"Rett syndrome and MECP2 duplication syndrome: disorders of MeCP2 dosage","volume":"18","author":"Collins","year":"2022","journal-title":"Neuropsychiatr. Dis. Treat."},{"issue":"21","key":"10.1016\/j.mcn.2026.104094_bb0045","doi-asserted-by":"crossref","first-page":"2679","DOI":"10.1093\/hmg\/ddh282","article-title":"Mild overexpression of MeCP2 causes a progressive neurological disorder in mice","volume":"13","author":"Collins","year":"2004","journal-title":"Hum. Mol. Genet."},{"issue":"1","key":"10.1016\/j.mcn.2026.104094_bb0050","doi-asserted-by":"crossref","DOI":"10.1111\/gbb.12739","article-title":"Safety and efficacy of genetic MECP2 supplementation in the R294X mouse model of Rett syndrome","volume":"21","author":"Collins","year":"2022","journal-title":"Genes Brain Behav."},{"issue":"1","key":"10.1016\/j.mcn.2026.104094_bb0055","doi-asserted-by":"crossref","DOI":"10.1016\/j.ejmg.2020.104102","article-title":"CDKL5 mutations may mimic Pitt-Hopkins syndrome phenotype","volume":"64","author":"Curro","year":"2021","journal-title":"Eur. J. Med. Genet."},{"issue":"5","key":"10.1016\/j.mcn.2026.104094_bb0060","doi-asserted-by":"crossref","DOI":"10.1016\/j.neurot.2024.e00376","article-title":"MeCP2 gene therapy ameliorates disease phenotype in mouse model for Pitt Hopkins syndrome","volume":"21","author":"Dennys","year":"2024","journal-title":"Neurotherapeutics"},{"issue":"3","key":"10.1016\/j.mcn.2026.104094_bb0065","doi-asserted-by":"crossref","first-page":"681","DOI":"10.1084\/jem.20160446","article-title":"Neuronal CTGF\/CCN2 negatively regulates myelination in a mouse model of tuberous sclerosis complex","volume":"214","author":"Ercan","year":"2017","journal-title":"J. Exp. Med."},{"issue":"18","key":"10.1016\/j.mcn.2026.104094_bb3005","doi-asserted-by":"crossref","first-page":"1723","DOI":"10.1056\/NEJMoa1702752","article-title":"Nusinersen versus sham control in infantile-onset spinal muscular atrophy","volume":"377","author":"Finkel","year":"2017","journal-title":"N. Engl. J. Med."},{"issue":"7","key":"10.1016\/j.mcn.2026.104094_bb3010","doi-asserted-by":"crossref","first-page":"491","DOI":"10.1016\/S2352-4642(21)00100-0","article-title":"Treatment of infantile-onset spinal muscular atrophy with nusinersen: final report of a phase 2, open-label, multicentre, dose-escalation study","volume":"5","author":"Finkel","year":"2021","journal-title":"Lancet Child Adolesc. Health"},{"issue":"9","key":"10.1016\/j.mcn.2026.104094_bb0070","doi-asserted-by":"crossref","first-page":"2210","DOI":"10.1021\/acschemneuro.7b00414","article-title":"Genetic reduction or negative modulation of mGlu7 does not impact anxiety and fear learning phenotypes in a mouse model of MECP2 duplication syndrome","volume":"9","author":"Fisher","year":"2018","journal-title":"ACS Chem. Neurosci."},{"issue":"12","key":"10.1016\/j.mcn.2026.104094_bb0075","doi-asserted-by":"crossref","first-page":"1676","DOI":"10.1002\/humu.22160","article-title":"Functional analysis of TCF4 missense mutations that cause Pitt-Hopkins syndrome","volume":"33","author":"Forrest","year":"2012","journal-title":"Hum. Mutat."},{"issue":"6","key":"10.1016\/j.mcn.2026.104094_bb0080","doi-asserted-by":"crossref","DOI":"10.26508\/lsa.202403102","article-title":"A novel human organoid model system reveals requirement of TCF4 for oligodendroglial differentiation","volume":"8","author":"Furlanetto","year":"2025","journal-title":"Life Sci Alliance"},{"issue":"11","key":"10.1016\/j.mcn.2026.104094_bb0085","doi-asserted-by":"crossref","first-page":"E242","DOI":"10.1002\/humu.20859","article-title":"TCF4 deletions in Pitt-Hopkins Syndrome","volume":"29","author":"Giurgea","year":"2008","journal-title":"Hum. Mutat."},{"issue":"403","key":"10.1016\/j.mcn.2026.104094_bb0090","doi-asserted-by":"crossref","DOI":"10.1126\/scitranslmed.aai7459","article-title":"mGlu7 potentiation rescues cognitive, social, and respiratory phenotypes in a mouse model of Rett syndrome","volume":"9","author":"Gogliotti","year":"2017","journal-title":"Sci. Transl. Med."},{"issue":"1","key":"10.1016\/j.mcn.2026.104094_bb0095","doi-asserted-by":"crossref","DOI":"10.1038\/s41572-024-00568-0","article-title":"Rett syndrome","volume":"10","author":"Gold","year":"2024","journal-title":"Nat. Rev. Dis. Primers"},{"issue":"3","key":"10.1016\/j.mcn.2026.104094_bb0100","doi-asserted-by":"crossref","first-page":"233","DOI":"10.1177\/0883073817750490","article-title":"Pitt-Hopkins Syndrome: a review of current literature, clinical approach, and 23-patient case series","volume":"33","author":"Goodspeed","year":"2018","journal-title":"J. Child Neurol."},{"issue":"3","key":"10.1016\/j.mcn.2026.104094_bb0105","doi-asserted-by":"crossref","first-page":"322","DOI":"10.1038\/85899","article-title":"A mouse Mecp2-null mutation causes neurological symptoms that mimic Rett syndrome","volume":"27","author":"Guy","year":"2001","journal-title":"Nat. Genet."},{"issue":"5815","key":"10.1016\/j.mcn.2026.104094_bb0110","doi-asserted-by":"crossref","first-page":"1143","DOI":"10.1126\/science.1138389","article-title":"Reversal of neurological defects in a mouse model of Rett syndrome","volume":"315","author":"Guy","year":"2007","journal-title":"Science"},{"issue":"2","key":"10.1016\/j.mcn.2026.104094_bb0115","first-page":"147","article-title":"Preclinical milestones in MECP2 gene transfer for treating Rett syndrome","volume":"47","author":"Jagadeeswaran","year":"2025","journal-title":"Dev. Neurosci."},{"key":"10.1016\/j.mcn.2026.104094_bb0120","doi-asserted-by":"crossref","first-page":"20","DOI":"10.1186\/s13229-018-0200-1","article-title":"Analysis of the expression pattern of the schizophrenia-risk and intellectual disability gene TCF4 in the developing and adult brain suggests a role in development and plasticity of cortical and hippocampal neurons","volume":"9","author":"Jung","year":"2018","journal-title":"Mol. Autism."},{"key":"10.1016\/j.mcn.2026.104094_bb0125","doi-asserted-by":"crossref","DOI":"10.7554\/eLife.72290","article-title":"Rescue of behavioral and electrophysiological phenotypes in a Pitt-Hopkins syndrome mouse model by genetic restoration of Tcf4 expression","volume":"11","author":"Kim","year":"2022","journal-title":"Elife"},{"issue":"2\u20133","key":"10.1016\/j.mcn.2026.104094_bb0130","doi-asserted-by":"crossref","first-page":"191","DOI":"10.1016\/j.neuint.2003.08.015","article-title":"The problem of astrocyte identity","volume":"45","author":"Kimelberg","year":"2004","journal-title":"Neurochem. Int."},{"issue":"7","key":"10.1016\/j.mcn.2026.104094_bb0135","doi-asserted-by":"crossref","first-page":"1685","DOI":"10.1002\/ajmg.a.34055","article-title":"Fetal pads as a clue to the diagnosis of Pitt-Hopkins syndrome","volume":"155A","author":"Lehalle","year":"2011","journal-title":"Am. J. Med. Genet. A"},{"issue":"8","key":"10.1016\/j.mcn.2026.104094_bb0140","doi-asserted-by":"crossref","first-page":"1235","DOI":"10.1038\/s41380-019-0353-0","article-title":"Disruption of TCF4 regulatory networks leads to abnormal cortical development and mental disabilities","volume":"24","author":"Li","year":"2019","journal-title":"Mol. Psychiatry"},{"issue":"1","key":"10.1016\/j.mcn.2026.104094_bb0145","doi-asserted-by":"crossref","DOI":"10.1038\/s41467-025-61092-7","article-title":"Targeting microRNA-dependent control of X chromosome inactivation improves the Rett Syndrome phenotype","volume":"16","author":"Lou","year":"2025","journal-title":"Nat. Commun."},{"issue":"3","key":"10.1016\/j.mcn.2026.104094_bb0150","doi-asserted-by":"crossref","first-page":"168","DOI":"10.1055\/s-0035-1564570","article-title":"Pitt-Hopkins syndrome and differential diagnosis: A molecular and clinical challenge","volume":"4","author":"Marangi","year":"2015","journal-title":"J. Pediatr. Genet."},{"issue":"7","key":"10.1016\/j.mcn.2026.104094_bb0155","doi-asserted-by":"crossref","first-page":"1536","DOI":"10.1002\/ajmg.a.34070","article-title":"The Pitt-Hopkins syndrome: report of 16 new patients and clinical diagnostic criteria","volume":"155A","author":"Marangi","year":"2011","journal-title":"Am. J. Med. Genet. A"},{"issue":"32","key":"10.1016\/j.mcn.2026.104094_bb0160","doi-asserted-by":"crossref","first-page":"7289","DOI":"10.1523\/JNEUROSCI.1924-05.2005","article-title":"Olig2 directs astrocyte and oligodendrocyte formation in postnatal subventricular zone cells","volume":"25","author":"Marshall","year":"2005","journal-title":"J. Neurosci."},{"key":"10.1016\/j.mcn.2026.104094_bb0165","doi-asserted-by":"crossref","first-page":"27","DOI":"10.1007\/978-3-540-46006-0_2","article-title":"Mechanisms regulating lineage diversity during mammalian cerebral cortical neurogenesis and gliogenesis","volume":"39","author":"Mehler","year":"2002","journal-title":"Results Probl. Cell Differ."},{"issue":"8","key":"10.1016\/j.mcn.2026.104094_bb0170","doi-asserted-by":"crossref","first-page":"954","DOI":"10.1038\/nm.3618","article-title":"Micropillar arrays as a high-throughput screening platform for therapeutics in multiple sclerosis","volume":"20","author":"Mei","year":"2014","journal-title":"Nat. Med."},{"issue":"21","key":"10.1016\/j.mcn.2026.104094_bb0175","doi-asserted-by":"crossref","first-page":"5546","DOI":"10.1523\/JNEUROSCI.5540-06.2007","article-title":"A mouse model of tuberous sclerosis: neuronal loss of Tsc1 causes dysplastic and ectopic neurons, reduced myelination, seizure activity, and limited survival","volume":"27","author":"Meikle","year":"2007","journal-title":"J. Neurosci."},{"issue":"7","key":"10.1016\/j.mcn.2026.104094_bb3015","doi-asserted-by":"crossref","first-page":"625","DOI":"10.1056\/NEJMoa1710504","article-title":"Nusinersen versus sham control in later-onset spinal muscular atrophy","volume":"378","author":"Mercuri","year":"2018","journal-title":"N. Engl. J. Med."},{"issue":"9","key":"10.1016\/j.mcn.2026.104094_bb0180","doi-asserted-by":"crossref","first-page":"3109","DOI":"10.1523\/JNEUROSCI.6000-11.2012","article-title":"A mouse model for MeCP2 duplication syndrome: MeCP2 overexpression impairs learning and memory and synaptic transmission","volume":"32","author":"Na","year":"2012","journal-title":"J. Neurosci."},{"issue":"48","key":"10.1016\/j.mcn.2026.104094_bb0185","doi-asserted-by":"crossref","first-page":"18764","DOI":"10.1523\/JNEUROSCI.2657-13.2013","article-title":"Oligodendrocyte lineage cells contribute unique features to Rett syndrome neuropathology","volume":"33","author":"Nguyen","year":"2013","journal-title":"J. Neurosci."},{"issue":"5","key":"10.1016\/j.mcn.2026.104094_bb0190","doi-asserted-by":"crossref","DOI":"10.1523\/ENEURO.0197-21.2021","article-title":"Isoform-specific reduction of the basic Helix-loop-Helix transcription factor TCF4 levels in Huntington\u2019s disease","volume":"8","author":"Nurm","year":"2021","journal-title":"eNeuro"},{"issue":"2","key":"10.1016\/j.mcn.2026.104094_bb3025","doi-asserted-by":"crossref","first-page":"304","DOI":"10.1038\/mp.2017.37","article-title":"The schizophrenia- and autism-associated gene, transcription factor 4 regulates the columnar distribution of layer 2\/3 prefrontal pyramidal neurons in an activity-dependent manner","volume":"23","author":"Page","year":"2018","journal-title":"Mol. Psychiatry"},{"issue":"11","key":"10.1016\/j.mcn.2026.104094_bb0195","doi-asserted-by":"crossref","first-page":"851","DOI":"10.1007\/s40263-024-01106-y","article-title":"Rett syndrome: the emerging landscape of treatment strategies","volume":"38","author":"Percy","year":"2024","journal-title":"CNS Drugs"},{"issue":"1","key":"10.1016\/j.mcn.2026.104094_bb0200","doi-asserted-by":"crossref","first-page":"42","DOI":"10.1002\/aur.1262","article-title":"The behavioral phenotype in MECP2 duplication syndrome: a comparison with idiopathic autism","volume":"6","author":"Peters","year":"2013","journal-title":"Autism Res."},{"key":"10.1016\/j.mcn.2026.104094_bb0205","doi-asserted-by":"crossref","DOI":"10.1038\/s41593-019-0578-x","article-title":"A myelin-related transcriptomic profile is shared by Pitt-Hopkins syndrome models and human autism spectrum disorder","author":"Phan","year":"2020","journal-title":"Nat. Neurosci."},{"issue":"2","key":"10.1016\/j.mcn.2026.104094_bb0210","doi-asserted-by":"crossref","first-page":"195","DOI":"10.1038\/ejhg.2012.140","article-title":"NF-kappaB signalling requirement for brain myelin formation is shown by genotype\/MRI phenotype correlations in patients with Xq28 duplications","volume":"21","author":"Philippe","year":"2013","journal-title":"Eur. J. Human Genet."},{"issue":"9","key":"10.1016\/j.mcn.2026.104094_bb0215","doi-asserted-by":"crossref","first-page":"2662","DOI":"10.1093\/hmg\/ddv030","article-title":"Rett syndrome like phenotypes in the R255X Mecp2 mutant mouse are rescued by MECP2 transgene","volume":"24","author":"Pitcher","year":"2015","journal-title":"Hum. Mol. Genet."},{"issue":"3","key":"10.1016\/j.mcn.2026.104094_bb0220","first-page":"182","article-title":"A syndrome of mental retardation, wide mouth and intermittent overbreathing","volume":"14","author":"Pitt","year":"1978","journal-title":"Aust. Paediatr. J."},{"issue":"4","key":"10.1016\/j.mcn.2026.104094_bb0225","doi-asserted-by":"crossref","first-page":"669","DOI":"10.1002\/humu.20935","article-title":"Mutational, functional, and expression studies of the TCF4 gene in Pitt-Hopkins syndrome","volume":"30","author":"de Pontual","year":"2009","journal-title":"Hum. Mutat."},{"issue":"5","key":"10.1016\/j.mcn.2026.104094_bb0230","doi-asserted-by":"crossref","first-page":"1079","DOI":"10.1002\/ajmg.a.33184","article-title":"The MECP2 duplication syndrome","volume":"152A","author":"Ramocki","year":"2010","journal-title":"Am. J. Med. Genet. A"},{"issue":"11","key":"10.1016\/j.mcn.2026.104094_bb0235","doi-asserted-by":"crossref","first-page":"797","DOI":"10.1097\/GIM.0b013e3181bd38a9","article-title":"Genotype-phenotype analysis of TCF4 mutations causing Pitt-Hopkins syndrome shows increased seizure activity with missense mutations","volume":"11","author":"Rosenfeld","year":"2009","journal-title":"Genet. Med."},{"issue":"1","key":"10.1016\/j.mcn.2026.104094_bb0240","doi-asserted-by":"crossref","first-page":"529","DOI":"10.1186\/s12859-017-1934-z","article-title":"ImageJ2: ImageJ for the next generation of scientific image data","volume":"18","author":"Rueden","year":"2017","journal-title":"BMC Bioinform."},{"key":"10.1016\/j.mcn.2026.104094_bb0245","series-title":"Investigating the Effects of Genetically Increasing MECP2 Dosage in Mouse Models of Typical and Atypical Rett Syndrome","author":"S.A.D.","year":"2021"},{"issue":"3","key":"10.1016\/j.mcn.2026.104094_bb0250","doi-asserted-by":"crossref","first-page":"244","DOI":"10.1016\/S0959-4388(02)00322-7","article-title":"Molecular mechanisms controlling cortical gliogenesis","volume":"12","author":"Sauvageot","year":"2002","journal-title":"Curr. Opin. Neurobiol."},{"issue":"7","key":"10.1016\/j.mcn.2026.104094_bb0255","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0022138","article-title":"Functional diversity of human basic helix-loop-helix transcription factor TCF4 isoforms generated by alternative 5\u2032 exon usage and splicing","volume":"6","author":"Sepp","year":"2011","journal-title":"PLoS One"},{"issue":"13","key":"10.1016\/j.mcn.2026.104094_bb0260","doi-asserted-by":"crossref","first-page":"2873","DOI":"10.1093\/hmg\/dds112","article-title":"Pitt-Hopkins syndrome-associated mutations in TCF4 lead to variable impairment of the transcription factor function ranging from hypomorphic to dominant-negative effects","volume":"21","author":"Sepp","year":"2012","journal-title":"Hum. Mol. Genet."},{"issue":"2","key":"10.1016\/j.mcn.2026.104094_bb0265","doi-asserted-by":"crossref","first-page":"176","DOI":"10.1007\/s12031-015-0597-3","article-title":"Involvement of MeCP2 in regulation of myelin-related gene expression in cultured rat oligodendrocytes","volume":"57","author":"Sharma","year":"2015","journal-title":"J. Mol. Neurosci."},{"issue":"6","key":"10.1016\/j.mcn.2026.104094_bb0270","doi-asserted-by":"crossref","DOI":"10.1016\/j.jbc.2021.101381","article-title":"Functional consequences of TCF4 missense substitutions associated with Pitt-Hopkins syndrome, mild intellectual disability, and schizophrenia","volume":"297","author":"Sirp","year":"2021","journal-title":"J. Biol. Chem."},{"key":"10.1016\/j.mcn.2026.104094_bb0275","doi-asserted-by":"crossref","DOI":"10.3389\/fnmol.2022.1033224","article-title":"Expression of alternative transcription factor 4 mRNAs and protein isoforms in the developing and adult rodent and human tissues","volume":"15","author":"Sirp","year":"2022","journal-title":"Front. Mol. Neurosci."},{"issue":"12","key":"10.1016\/j.mcn.2026.104094_bb0280","doi-asserted-by":"crossref","first-page":"4291","DOI":"10.1093\/cercor\/bhx282","article-title":"Contextual fear extinction induces hippocampal Metaplasticity mediated by metabotropic glutamate receptor 5","volume":"28","author":"Stansley","year":"2018","journal-title":"Cereb. Cortex"},{"key":"10.1016\/j.mcn.2026.104094_bb0285","article-title":"Characterization of astrocyte density in the Pitt-Hopkins syndrome mouse model of ASD","author":"Stump","year":"2025","journal-title":"bioRxiv"},{"issue":"3","key":"10.1016\/j.mcn.2026.104094_bb0290","doi-asserted-by":"crossref","first-page":"161","DOI":"10.1016\/S1084-9521(03)00007-7","article-title":"Making and repairing the mammalian brain--signaling toward neurogenesis and gliogenesis","volume":"14","author":"Sun","year":"2003","journal-title":"Semin. Cell Dev. Biol."},{"issue":"1","key":"10.1016\/j.mcn.2026.104094_bb0295","doi-asserted-by":"crossref","DOI":"10.1016\/j.celrep.2022.111942","article-title":"Mutations in the transcriptional regulator MeCP2 severely impact key cellular and molecular signatures of human astrocytes during maturation","volume":"42","author":"Sun","year":"2023","journal-title":"Cell Rep."},{"issue":"1","key":"10.1016\/j.mcn.2026.104094_bb3030","doi-asserted-by":"crossref","first-page":"14133","DOI":"10.1038\/s41598-017-14207-0","article-title":"Differentiation of oligodendrocyte precursor cells from Sox10-venus mice to oligodendrocytes and astrocytes","volume":"7","author":"Suzuki","year":"2017","journal-title":"Sci. Rep."},{"key":"10.1016\/j.mcn.2026.104094_bb0300","doi-asserted-by":"crossref","DOI":"10.1038\/emm.2013.32","article-title":"Pitt-Hopkins syndrome: intellectual disability due to loss of TCF4-regulated gene transcription","volume":"45","author":"Sweatt","year":"2013","journal-title":"Exp. Mol. Med."},{"issue":"4","key":"10.1016\/j.mcn.2026.104094_bb0305","doi-asserted-by":"crossref","first-page":"975","DOI":"10.1002\/ajmg.a.36416","article-title":"If not Angelman, what is it? A review of Angelman-like syndromes","volume":"164A","author":"Tan","year":"2014","journal-title":"Am. J. Med. Genet. A"},{"key":"10.1016\/j.mcn.2026.104094_bb0310","doi-asserted-by":"crossref","first-page":"8","DOI":"10.3389\/fnana.2018.00008","article-title":"Olig2-lineage astrocytes: a distinct subtype of astrocytes that differs from GFAP astrocytes","volume":"12","author":"Tatsumi","year":"2018","journal-title":"Front. Neuroanat."},{"issue":"4","key":"10.1016\/j.mcn.2026.104094_bb0315","doi-asserted-by":"crossref","first-page":"1989","DOI":"10.1007\/s00429-016-1316-8","article-title":"Employing an open-source tool to assess astrocyte tridimensional structure","volume":"222","author":"Tavares","year":"2017","journal-title":"Brain Struct. Funct."},{"issue":"1","key":"10.1016\/j.mcn.2026.104094_bb0320","doi-asserted-by":"crossref","first-page":"20565","DOI":"10.1038\/s41598-024-71040-y","article-title":"Mitochondrial dysfunction and increased reactive oxygen species production in MECP2 mutant astrocytes and their impact on neurons","volume":"14","author":"Tomasello","year":"2024","journal-title":"Sci. Rep."},{"issue":"3","key":"10.1016\/j.mcn.2026.104094_bb0325","doi-asserted-by":"crossref","first-page":"442","DOI":"10.1086\/444549","article-title":"Duplication of the MECP2 region is a frequent cause of severe mental retardation and progressive neurological symptoms in males","volume":"77","author":"Van Esch","year":"2005","journal-title":"Am. J. Hum. Genet."},{"key":"10.1016\/j.mcn.2026.104094_bb0330","doi-asserted-by":"crossref","DOI":"10.1016\/j.neuropharm.2022.109022","article-title":"Exploration of group II metabotropic glutamate receptor modulation in mouse models of Rett syndrome and MECP2 duplication syndrome","volume":"209","author":"Vermudez","year":"2022","journal-title":"Neuropharmacology"},{"issue":"1","key":"10.1016\/j.mcn.2026.104094_bb0335","doi-asserted-by":"crossref","DOI":"10.1111\/gbb.12752","article-title":"Profiling beneficial and potential adverse effects of MeCP2 overexpression in a hypomorphic Rett syndrome mouse model","volume":"21","author":"Vermudez","year":"2022","journal-title":"Genes Brain Behav."},{"issue":"2","key":"10.1016\/j.mcn.2026.104094_bb0340","doi-asserted-by":"crossref","first-page":"95","DOI":"10.1002\/1098-1136(200008)31:2<95::AID-GLIA10>3.0.CO;2-6","article-title":"Controversy surrounding the existence of discrete functional classes of astrocytes in adult gray matter","volume":"31","author":"Walz","year":"2000","journal-title":"Glia"},{"issue":"1","key":"10.1016\/j.mcn.2026.104094_bb0345","doi-asserted-by":"crossref","first-page":"36","DOI":"10.1186\/s13041-021-00747-0","article-title":"Region-specific distribution of Olig2-expressing astrocytes in adult mouse brain and spinal cord","volume":"14","author":"Wang","year":"2021","journal-title":"Mol. Brain"},{"issue":"9","key":"10.1016\/j.mcn.2026.104094_bb0350","doi-asserted-by":"crossref","first-page":"4839","DOI":"10.1093\/nar\/gkaa218","article-title":"Transcription factor Tcf4 is the preferred heterodimerization partner for Olig2 in oligodendrocytes and required for differentiation","volume":"48","author":"Wedel","year":"2020","journal-title":"Nucleic Acids Res."},{"issue":"1","key":"10.1016\/j.mcn.2026.104094_bb0355","doi-asserted-by":"crossref","first-page":"64","DOI":"10.1002\/humu.21639","article-title":"Novel comprehensive diagnostic strategy in Pitt-Hopkins syndrome: clinical score and further delineation of the TCF4 mutational spectrum","volume":"33","author":"Whalen","year":"2012","journal-title":"Hum. Mutat."},{"issue":"1","key":"10.1016\/j.mcn.2026.104094_bb0360","doi-asserted-by":"crossref","first-page":"3","DOI":"10.1186\/2040-2392-4-3","article-title":"MeCP2 modulates gene expression pathways in astrocytes","volume":"4","author":"Yasui","year":"2013","journal-title":"Mol. Autism."},{"key":"10.1016\/j.mcn.2026.104094_bb0365","doi-asserted-by":"crossref","DOI":"10.1016\/j.jneumeth.2020.108798","article-title":"Establishment of a simple one-step method for oligodendrocyte progenitor cell preparation from rodent brains","volume":"342","author":"Yoshida","year":"2020","journal-title":"J. Neurosci. Methods"},{"issue":"11","key":"10.1016\/j.mcn.2026.104094_bb0370","doi-asserted-by":"crossref","first-page":"6562","DOI":"10.1038\/s41380-021-01119-9","article-title":"Transcription factor 4 controls positioning of cortical projection neurons through regulation of cell adhesion","volume":"26","author":"Zhang","year":"2021","journal-title":"Mol. Psychiatry"},{"issue":"21","key":"10.1016\/j.mcn.2026.104094_bb0375","doi-asserted-by":"crossref","first-page":"5114","DOI":"10.1038\/s44318-024-00218-x","article-title":"Astrocyte allocation during brain development is controlled by Tcf4-mediated fate restriction","volume":"43","author":"Zhang","year":"2024","journal-title":"EMBO J."},{"issue":"6","key":"10.1016\/j.mcn.2026.104094_bb0380","doi-asserted-by":"crossref","first-page":"2898","DOI":"10.1128\/MCB.16.6.2898","article-title":"B-lymphocyte development is regulated by the combined dosage of three basic helix-loop-helix genes, E2A, E2-2, and HEB","volume":"16","author":"Zhuang","year":"1996","journal-title":"Mol. Cell. Biol."},{"issue":"4","key":"10.1016\/j.mcn.2026.104094_bb0385","doi-asserted-by":"crossref","first-page":"462","DOI":"10.1111\/cge.13506","article-title":"Diagnosis and management in Pitt-Hopkins syndrome: first international consensus statement","volume":"95","author":"Zollino","year":"2019","journal-title":"Clin. Genet."},{"issue":"5","key":"10.1016\/j.mcn.2026.104094_bb0390","doi-asserted-by":"crossref","first-page":"994","DOI":"10.1086\/515583","article-title":"Haploinsufficiency of TCF4 causes syndromal mental retardation with intermittent hyperventilation (Pitt-Hopkins syndrome)","volume":"80","author":"Zweier","year":"2007","journal-title":"Am. J. Hum. Genet."},{"issue":"11","key":"10.1016\/j.mcn.2026.104094_bb0395","doi-asserted-by":"crossref","first-page":"738","DOI":"10.1136\/jmg.2008.060129","article-title":"Further delineation of Pitt-Hopkins syndrome: phenotypic and genotypic description of 16 novel patients","volume":"45","author":"Zweier","year":"2008","journal-title":"J. Med. Genet."}],"container-title":["Molecular and Cellular Neuroscience"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S1044743126000242?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S1044743126000242?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2026,6,10]],"date-time":"2026-06-10T03:35:42Z","timestamp":1781062542000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S1044743126000242"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,6]]},"references-count":86,"alternative-id":["S1044743126000242"],"URL":"https:\/\/doi.org\/10.1016\/j.mcn.2026.104094","relation":{},"ISSN":["1044-7431"],"issn-type":[{"value":"1044-7431","type":"print"}],"subject":[],"published":{"date-parts":[[2026,6]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"Increasing MeCP2 protein in Pitt-Hopkins syndrome model (Tcf4+\/\u2212) mice does not affect abnormal myelination but induces the generation of astrocytes","name":"articletitle","label":"Article Title"},{"value":"Molecular and Cellular Neuroscience","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.mcn.2026.104094","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2026 The Authors. Published by Elsevier Inc.","name":"copyright","label":"Copyright"}],"article-number":"104094"}}