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Genetic analyses in mouse\n                    <jats:italic>Mecp2<\/jats:italic>\n                    mutants, which exhibit key features of human RTT, have been essential for deciphering the mechanisms of MeCP2 function; nonetheless, our understanding of these complex mechanisms is incomplete. Zebrafish\n                    <jats:italic>mecp2<\/jats:italic>\n                    mutants exhibit mild behavioral deficits but have not been analyzed in depth. Here, we combine transcriptomic and behavioral assays to assess baseline and stimulus\u2010evoked motor responses and sensory filtering in zebrafish\n                    <jats:italic>mecp2<\/jats:italic>\n                    mutants from 5 to 7\u2009days post\u2010fertilization (dpf). We show that zebrafish\n                    <jats:italic>mecp2<\/jats:italic>\n                    function is required for normal thigmotaxis but is dispensable for gross movement, acoustic startle response, and sensory filtering (habituation and sensorimotor gating), and reveal a previously unknown role for\n                    <jats:italic>mecp2<\/jats:italic>\n                    in behavioral responses to visual stimuli. RNA\u2010seq analysis identified a large gene set that requires\n                    <jats:italic>mecp2<\/jats:italic>\n                    function for correct transcription at 4 dpf, and pathway analysis revealed several pathways that require MeCP2 function in both zebrafish and mammals. These findings show that MeCP2's function as a transcriptional regulator is conserved across vertebrates and supports using zebrafish to complement mouse modeling in elucidating these conserved mechanisms.\n                  <\/jats:p>","DOI":"10.1002\/ajmg.b.32981","type":"journal-article","created":{"date-parts":[[2024,3,29]],"date-time":"2024-03-29T06:10:30Z","timestamp":1711692630000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":7,"title":["Behavioral and transcriptomic analyses of\n                    <scp>\n                      <i>mecp<\/i>\n                      2\n                    <\/scp>\n                    function in zebrafish"],"prefix":"10.1002","volume":"195","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-4657-6860","authenticated-orcid":false,"given":"Nicholas J.","family":"Santistevan","sequence":"first","affiliation":[{"name":"Departments of Integrative Biology and Neuroscience University of Wisconsin  Madison Wisconsin USA"},{"name":"Genetics Ph.D. Training Program University of Wisconsin  Madison Wisconsin USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7859-3622","authenticated-orcid":false,"given":"Colby T.","family":"Ford","sequence":"additional","affiliation":[{"name":"School of Data Science University of North Carolina  Charlotte North Carolina USA"},{"name":"Department of Bioinformatics and Genomics University of North Carolina  Charlotte North Carolina USA"},{"name":"Tuple, LLC  Charlotte North Carolina USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0009-0004-5455-919X","authenticated-orcid":false,"given":"Cole S.","family":"Gilsdorf","sequence":"additional","affiliation":[{"name":"Departments of Integrative Biology and Neuroscience University of Wisconsin  Madison Wisconsin USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3131-7572","authenticated-orcid":false,"given":"Yevgenya","family":"Grinblat","sequence":"additional","affiliation":[{"name":"Departments of Integrative Biology and Neuroscience University of Wisconsin  Madison Wisconsin 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