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AHDS is characterized by severe intellectual deficiency, neuromuscular impairment and brain hypothyroidism. In order to understand the mechanism for TH-dependent hypomyelination, we developed an mct8 mutant (mct8\u2212\/\u2212) zebrafish model. The quantification of genetic markers for oligodendrocyte progenitor cells (OPCs) and mature oligodendrocytes revealed reduced differentiation of OPCs into oligodendrocytes in mct8\u2212\/\u2212 larvae and adults. Live imaging of single glial cells showed that the number of oligodendrocytes and the length of their extensions are reduced, and the number of peripheral Schwann cells is increased, in mct8\u2212\/\u2212 larvae compared with wild type. Pharmacological analysis showed that TH analogs and clemastine partially rescued the hypomyelination in the CNS of mct8\u2212\/\u2212 larvae. Intriguingly, triiodothyronine (T3) treatment rescued hypomyelination in mct8\u2212\/\u2212 embryos before the maturation of the blood\u2013brain barrier (BBB), but did not affect hypomyelination in older larvae. Thus, we expressed Mct8-tagRFP in the endothelial cells of the vascular system and showed that even relatively weak mosaic expression completely rescued hypomyelination in mct8\u2212\/\u2212 larvae. These results suggest potential pharmacological treatments and BBB-targeted gene therapy that can enhance myelination in AHDS and possibly in other TH-dependent brain disorders.<\/jats:p>","DOI":"10.1242\/dmm.027227","type":"journal-article","created":{"date-parts":[[2016,9,23]],"date-time":"2016-09-23T22:08:45Z","timestamp":1474668525000},"page":"1339-1348","source":"Crossref","is-referenced-by-count":62,"title":["Pharmacological treatment and BBB-targeted genetic therapy for MCT8-dependent hypomyelination in zebrafish"],"prefix":"10.1242","volume":"9","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-5064-247X","authenticated-orcid":false,"given":"David","family":"Zada","sequence":"first","affiliation":[{"name":"the Multidisciplinary Brain Research Center, Bar-Ilan University The Faculty of Life Sciences and \u2008 , Ramat-Gan 5290002 , Israel"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6341-0267","authenticated-orcid":false,"given":"Adi","family":"Tovin","sequence":"additional","affiliation":[{"name":"the Multidisciplinary Brain Research Center, Bar-Ilan University The Faculty of Life Sciences and \u2008 , Ramat-Gan 5290002 , Israel"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1476-1081","authenticated-orcid":false,"given":"Tali","family":"Lerer-Goldshtein","sequence":"additional","affiliation":[{"name":"the Multidisciplinary Brain Research Center, Bar-Ilan University The Faculty of Life Sciences and \u2008 , Ramat-Gan 5290002 , Israel"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9248-5919","authenticated-orcid":false,"given":"Lior","family":"Appelbaum","sequence":"additional","affiliation":[{"name":"the Multidisciplinary Brain Research Center, Bar-Ilan University The Faculty of Life Sciences and \u2008 , Ramat-Gan 5290002 , Israel"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","reference":[{"key":"2024061302515560600_DMM027227C1","doi-asserted-by":"publisher","first-page":"5065","DOI":"10.1210\/en.2011-1166","article-title":"Identification and functional characterization of zebrafish solute carrier Slc16a2 (Mct8) as a thyroid hormone membrane transporter","volume":"152","author":"Arjona","year":"2011","journal-title":"Endocrinology"},{"key":"2024061302515560600_DMM027227C2","doi-asserted-by":"publisher","first-page":"254","DOI":"10.1002\/jnr.1218","article-title":"Selective effect of hypothyroidism on expression of myelin markers during development","volume":"66","author":"Barradas","year":"2001","journal-title":"J. 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