{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,13]],"date-time":"2026-10-13T22:03:03Z","timestamp":1791928983229,"version":"4.3.4"},"reference-count":60,"publisher":"Oxford University Press (OUP)","issue":"8","license":[{"start":{"date-parts":[[2023,4,18]],"date-time":"2023-04-18T00:00:00Z","timestamp":1681776000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"funder":[{"DOI":"10.13039\/100007086","name":"Pitt\u2013Hopkins Research Foundation","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100007086","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100000025","name":"NIMH","doi-asserted-by":"publisher","award":["R01MH110487"],"award-info":[{"award-number":["R01MH110487"]}],"id":[{"id":"10.13039\/100000025","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2023,8,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>Pitt\u2013Hopkins syndrome is an autism spectrum disorder caused by autosomal dominant mutations in the human transcription factor 4 gene (TCF4). One pathobiological process caused by murine Tcf4 mutation is a cell autonomous reduction in oligodendrocytes and myelination. In this study, we show that the promyelinating compounds, clemastine, sobetirome and Sob-AM2 are effective at restoring myelination defects in a Pitt\u2013Hopkins syndrome mouse model. In vitro, clemastine treatment reduced excess oligodendrocyte precursor cells and normalized oligodendrocyte density. In vivo, 2-week intraperitoneal administration of clemastine also normalized oligodendrocyte precursor cell and oligodendrocyte density in the cortex of Tcf4 mutant mice and appeared to increase the number of axons undergoing myelination, as EM imaging of the corpus callosum showed a significant increase in the proportion of uncompacted myelin and an overall reduction in the g-ratio. Importantly, this treatment paradigm resulted in functional rescue by improving electrophysiology and behaviour. To confirm behavioural rescue was achieved via enhancing myelination, we show that treatment with the thyroid hormone receptor agonist sobetirome or its brain penetrating prodrug Sob-AM2, was also effective at normalizing oligodendrocyte precursor cell and oligodendrocyte densities and behaviour in the Pitt\u2013Hopkins syndrome mouse model. Together, these results provide preclinical evidence that promyelinating therapies may be beneficial in Pitt\u2013Hopkins syndrome and potentially other neurodevelopmental disorders characterized by dysmyelination.<\/jats:p>","DOI":"10.1093\/brain\/awad057","type":"journal-article","created":{"date-parts":[[2023,4,18]],"date-time":"2023-04-18T00:58:05Z","timestamp":1681779485000},"page":"3331-3346","source":"Crossref","is-referenced-by-count":42,"title":["Promyelinating drugs promote functional recovery in an autism spectrum disorder mouse model of Pitt\u2013Hopkins syndrome"],"prefix":"10.1093","volume":"146","author":[{"given":"Joseph F","family":"Bohlen","sequence":"first","affiliation":[{"name":"Lieber Institute for Brain Development, Johns Hopkins Medical Campus , Baltimore, MD 21205 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Colin M","family":"Cleary","sequence":"additional","affiliation":[{"name":"Department of Physiology and Neurobiology, University of Connecticut , Storrs, CT 06269 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Debamitra","family":"Das","sequence":"additional","affiliation":[{"name":"Lieber Institute for Brain Development, Johns Hopkins Medical Campus , Baltimore, MD 21205 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6236-6396","authenticated-orcid":false,"given":"Srinidhi Rao","family":"Sripathy","sequence":"additional","affiliation":[{"name":"Lieber Institute for Brain Development, Johns Hopkins Medical Campus , Baltimore, MD 21205 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Norah","family":"Sadowski","sequence":"additional","affiliation":[{"name":"Lieber Institute for Brain Development, Johns Hopkins Medical Campus , Baltimore, MD 21205 , USA"},{"name":"Solomon H. Snyder Department of Neuroscience, Johns Hopkins University School of Medicine , Baltimore, MD 21205 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gina","family":"Shim","sequence":"additional","affiliation":[{"name":"Lieber Institute for Brain Development, Johns Hopkins Medical Campus , Baltimore, MD 21205 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rakaia F","family":"Kenney","sequence":"additional","affiliation":[{"name":"Lieber Institute for Brain Development, Johns Hopkins Medical Campus , Baltimore, MD 21205 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ingrid P","family":"Buchler","sequence":"additional","affiliation":[{"name":"Lieber Institute for Brain Development, Johns Hopkins Medical Campus , Baltimore, MD 21205 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tapasree","family":"Banerji","sequence":"additional","affiliation":[{"name":"Department of Physiology & Pharmacology and Program in Chemical Biology, Oregon Health & Science University , Portland, OR 97239 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Thomas S","family":"Scanlan","sequence":"additional","affiliation":[{"name":"Department of Physiology & Pharmacology and Program in Chemical Biology, Oregon Health & Science University , Portland, OR 97239 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Daniel K","family":"Mulkey","sequence":"additional","affiliation":[{"name":"Department of Physiology and Neurobiology, University of Connecticut , Storrs, CT 06269 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Brady J","family":"Maher","sequence":"additional","affiliation":[{"name":"Lieber Institute for Brain Development, Johns Hopkins Medical Campus , Baltimore, MD 21205 , USA"},{"name":"Solomon H. 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