{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,18]],"date-time":"2026-03-18T07:21:25Z","timestamp":1773818485099,"version":"3.50.1"},"reference-count":154,"publisher":"MDPI AG","issue":"1","license":[{"start":{"date-parts":[[2022,1,4]],"date-time":"2022-01-04T00:00:00Z","timestamp":1641254400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Cells"],"abstract":"<jats:p>Williams syndrome (WS) is a multisystem neurodevelopmental disorder caused by a de novo hemizygous deletion of ~26 genes from chromosome 7q11.23, among them the general transcription factor II-I (GTF2I). By studying a novel murine model for the hypersociability phenotype associated with WS, we previously revealed surprising aberrations in myelination and cell differentiation properties in the cortices of mutant mice compared to controls. These mutant mice had selective deletion of Gtf2i in the excitatory neurons of the forebrain. Here, we applied diffusion magnetic resonance imaging and fiber tracking, which showed a reduction in the number of streamlines in limbic outputs such as the fimbria\/fornix fibers and the stria terminalis, as well as the corpus callosum of these mutant mice compared to controls. Furthermore, we utilized next-generation sequencing (NGS) analysis of cortical small RNAs\u2019 expression (RNA-Seq) levels to identify altered expression of microRNAs (miRNAs), including two from the miR-34 cluster, known to be involved in prominent processes in the developing nervous system. Luciferase reporter assay confirmed the direct binding of miR-34c-5p to the 3\u2019UTR of PTPRU\u2014a gene involved in neural development that was elevated in the cortices of mutant mice relative to controls. Moreover, we found an age-dependent variation in the expression levels of doublecortin (Dcx)\u2014a verified miR-34 target. Thus, we demonstrate the substantial effect a single gene deletion can exert on miRNA regulation and brain structure, and advance our understanding and, hopefully, treatment of WS.<\/jats:p>","DOI":"10.3390\/cells11010158","type":"journal-article","created":{"date-parts":[[2022,1,5]],"date-time":"2022-01-05T20:42:16Z","timestamp":1641415336000},"page":"158","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":13,"title":["Altered White Matter and microRNA Expression in a Murine Model Related to Williams Syndrome Suggests That miR-34b\/c Affects Brain Development via Ptpru and Dcx Modulation"],"prefix":"10.3390","volume":"11","author":[{"given":"Meitar","family":"Grad","sequence":"first","affiliation":[{"name":"Sagol School of Neuroscience, Tel Aviv University, Tel Aviv 6997801, Israel"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ariel","family":"Nir","sequence":"additional","affiliation":[{"name":"Sagol School of Neuroscience, Tel Aviv University, Tel Aviv 6997801, Israel"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Gilad","family":"Levy","sequence":"additional","affiliation":[{"name":"Sagol School of Neuroscience, Tel Aviv University, Tel Aviv 6997801, Israel"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sari Schokoroy","family":"Trangle","sequence":"additional","affiliation":[{"name":"Faculty of Social Sciences, School of Psychological Sciences, Tel Aviv University, Tel Aviv 6997801, Israel"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9376-4955","authenticated-orcid":false,"given":"Guy","family":"Shapira","sequence":"additional","affiliation":[{"name":"Department of Cell and Developmental Biology, Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv 6997801, Israel"},{"name":"Edmond J. Safra Center for Bioinformatics, Tel Aviv University, Tel Aviv 6997801, Israel"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9913-6124","authenticated-orcid":false,"given":"Noam","family":"Shomron","sequence":"additional","affiliation":[{"name":"Sagol School of Neuroscience, Tel Aviv University, Tel Aviv 6997801, Israel"},{"name":"Department of Cell and Developmental Biology, Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv 6997801, Israel"},{"name":"Edmond J. Safra Center for Bioinformatics, Tel Aviv University, Tel Aviv 6997801, Israel"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yaniv","family":"Assaf","sequence":"additional","affiliation":[{"name":"Sagol School of Neuroscience, Tel Aviv University, Tel Aviv 6997801, Israel"},{"name":"Faculty of Life Sciences, School of Neurobiology, Biochemistry & Biophysics, Tel Aviv University, Tel Aviv 6997801, Israel"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9724-4578","authenticated-orcid":false,"given":"Boaz","family":"Barak","sequence":"additional","affiliation":[{"name":"Sagol School of Neuroscience, Tel Aviv University, Tel Aviv 6997801, Israel"},{"name":"Faculty of Social Sciences, School of Psychological Sciences, Tel Aviv University, Tel Aviv 6997801, Israel"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1968","published-online":{"date-parts":[[2022,1,4]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"41","DOI":"10.1016\/j.gde.2021.01.013","article-title":"7q11.23 Deletion and Duplication","volume":"68","author":"Osborne","year":"2021","journal-title":"Curr. 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