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Of the deleted genes, <jats:italic>GTF2I<\/jats:italic> has been linked to hypersociability in WS. We have recently shown that <jats:italic>Gtf2i<\/jats:italic> deletion from forebrain excitatory neurons, referred to as <jats:italic>Gtf2i<\/jats:italic> conditional knockout (cKO) mice leads to multi\u2010faceted myelination deficits associated with the social behaviors affected in WS. These deficits were potentially mediated also by microglia, as they present a close relationship with oligodendrocytes. To study the impact of altered myelination, we characterized these mice in terms of microglia over the course of development. In postnatal day 30 (P30) <jats:italic>Gtf2i<\/jats:italic> cKO mice, cortical microglia displayed a more ramified state, as compared with wild type (controls). However, postnatal day 4 (P4) microglia exhibited high proliferation rates and an elevated activation state, demonstrating altered properties related to activation and inflammation in <jats:italic>Gtf2i<\/jats:italic> cKO mice compared with control. Intriguingly, P4 <jats:italic>Gtf2i<\/jats:italic> cKO\u2010derived microglial cells exhibited significantly elevated myelin phagocytosis <jats:italic>in\u00a0vitro<\/jats:italic> compared to control mice. Lastly, systemic injection of clemastine to P4 <jats:italic>Gtf2i<\/jats:italic> cKO and control mice until P30, led to a significant interaction between genotypes and treatments on the expression levels of the phagocytic marker CD68, and a significant reduction of the macrophage\/microglial marker <jats:italic>Iba1<\/jats:italic> transcript levels in the cortex of the <jats:italic>Gtf2i<\/jats:italic> cKO treated mice. Our data thus implicate microglia as important players in WS, and that early postnatal manipulation of microglia might be beneficial in treating inflammatory and myelin\u2010related pathologies.<\/jats:p>","DOI":"10.1002\/glia.24519","type":"journal-article","created":{"date-parts":[[2024,3,7]],"date-time":"2024-03-07T12:12:02Z","timestamp":1709813522000},"page":"1117-1135","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["Neuronal deletion of <i>Gtf2i<\/i> results in developmental microglial alterations in a mouse model related to Williams syndrome"],"prefix":"10.1002","volume":"72","author":[{"given":"Ela","family":"Bar","sequence":"first","affiliation":[{"name":"The School of Psychological Sciences, Faculty of Social Sciences Tel Aviv University  Tel Aviv Israel"},{"name":"The School of Neurobiology, Biochemistry &amp; Biophysics, Faculty of Life Sciences Tel Aviv University  Tel Aviv 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