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Individuals with WS exhibit friendly, impulsive, and hyper-social behaviours, often coupled with anxiety. WS is caused by a hemizygous microdeletion on chromosome 7q11.23. Among the affected genes,\n                    <jats:italic>FZD9<\/jats:italic>\n                    is implicated in neural development and synaptic function. Thus, we postulated that disruption of\n                    <jats:italic>FZD9<\/jats:italic>\n                    might contribute to the behavioural features of WS, and that pharmacological modulation of Wnt signalling, particularly the canonical \u03b2-catenin pathway, could offer therapeutic potential. We generated two zebrafish lines carrying predicted loss-of-function alleles of\n                    <jats:italic>fzd9b<\/jats:italic>\n                    , a zebrafish orthologue of\n                    <jats:italic>FZD9<\/jats:italic>\n                    . Our results demonstrated that mutant fish exhibited altered expression of hypothalamic\u2013pituitary\u2013interrenal axis-related genes, increased neuronal branching, impaired habituation to acoustic startle, and increased anxiety-like behaviour in adulthood. Canonical Wnt\/\u03b2-catenin pathway gene expression was also altered, together with the expression of other frizzled receptors; however, acute GSK-3 inhibition failed to rescue behavioural phenotypes under the conditions tested. These findings indicate that the selected acute treatment paradigm was insufficient to reverse the phenotype but do not exclude a contribution of canonical Wnt signalling. Expression of\n                    <jats:italic>fzd9b<\/jats:italic>\n                    ,\n                    <jats:italic>wnt5b<\/jats:italic>\n                    , and\n                    <jats:italic>tafa5l<\/jats:italic>\n                    was upregulated, further supporting transcriptional dysregulation. Together, our results highlight a conserved role for Fzd9b in modulating stress reactivity and anxiety-like behaviour in zebrafish and support FZD9 as a contributor to WS-associated neurobehavioural alterations.\n                  <\/jats:p>","DOI":"10.1007\/s12035-026-06127-w","type":"journal-article","created":{"date-parts":[[2026,8,24]],"date-time":"2026-08-24T20:29:05Z","timestamp":1787603345000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Disrupting fzd9b in Zebrafish Recapitulates Stress- and Anxiety-Like Behaviours Relevant to Williams Syndrome"],"prefix":"10.1007","volume":"63","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-6390-9690","authenticated-orcid":false,"given":"Jose V.","family":"Torres-Perez","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0449-9665","authenticated-orcid":false,"given":"Adele","family":"Leggieri","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xian","family":"Wang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aine","family":"Kehoe","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lianri","family":"Van Schalkwyk","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0511-6048","authenticated-orcid":false,"given":"Aleksandra M.","family":"Mech","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5784-582X","authenticated-orcid":false,"given":"Sofia","family":"Anagianni","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1965-5894","authenticated-orcid":false,"given":"William","family":"Havelange","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4169-4083","authenticated-orcid":false,"given":"Caroline H.","family":"Brennan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2026,8,24]]},"reference":[{"key":"6127_CR1","doi-asserted-by":"publisher","unstructured":"Martens MA, Wilson SJ, Reutens DC (2008) Research review: Williams syndrome: A critical review of the cognitive, behavioral, and neuroanatomical phenotype. 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