{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,3]],"date-time":"2026-10-03T01:53:06Z","timestamp":1790992386109,"version":"4.1.0"},"reference-count":32,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2026,10,2]],"date-time":"2026-10-02T00:00:00Z","timestamp":1790899200000},"content-version":"vor","delay-in-days":1,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2026,10,1]],"date-time":"2026-10-01T00:00:00Z","timestamp":1790812800000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Intl J of Devlp Neuroscience"],"published-print":{"date-parts":[[2026,10]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>\n                    The zinc finger transcription factor\n                    <jats:italic>PLAG1<\/jats:italic>\n                    has been implicated in multiple diverse cellular processes, yet its role in the onset of vertebrate neurodevelopment and behaviour remains largely unexplored. Notably, dysregulation of\n                    <jats:italic>PLAG1<\/jats:italic>\n                    has been associated with Silver\u2013Russell syndrome (SRS) in humans, a growth disorder often presenting with neurodevelopmental delay and craniofacial abnormalities. Here, we investigate the function of\n                    <jats:italic>plag1<\/jats:italic>\n                    in zebrafish embryogenesis, focusing on behavioural phenotypes, neural development and craniofacial morphogenesis, as a potential novel model of SRS. Using antisense morpholino\u2010mediated knockdown, we assessed the impact of\n                    <jats:italic>plag1<\/jats:italic>\n                    loss on brain architecture, motor neuron development, craniofacial patterning and stimulus\u2010evoked behavioural responses. While\n                    <jats:italic>plag1<\/jats:italic>\n                    \u2010deficient embryos displayed pronounced locomotor defects when housed in groups, these phenotypes were not recapitulated under isolated, individual tracking conditions, suggesting a role for\n                    <jats:italic>plag1<\/jats:italic>\n                    in modulating specific behavioural outputs. Morphological analyses revealed no overt disruptions to gross brain structure or primary motor neuron patterning. However, quantitation of craniofacial skeleton formation identified subtle abnormalities in jaw and cranial cartilage development, consistent with features observed in SRS. Furthermore,\n                    <jats:italic>plag1<\/jats:italic>\n                    morphants exhibited significantly altered responses to sensory stimuli, implicating\n                    <jats:italic>plag1<\/jats:italic>\n                    in the regulation of sensorimotor integration during early development. Collectively, these findings uncover a novel role for\n                    <jats:italic>plag1<\/jats:italic>\n                    in early neurobehavioural modulation and craniofacial patterning, phenocopying multiple features of SRS, and highlight the importance of environmental and social context in the phenotypic interpretation of neurodevelopmental gene function.\n                  <\/jats:p>","DOI":"10.1002\/jdn.70185","type":"journal-article","created":{"date-parts":[[2026,10,3]],"date-time":"2026-10-03T00:45:27Z","timestamp":1790988327000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Plag1 Regulates Sensorimotor Modulation in Zebrafish"],"prefix":"10.1002","volume":"86","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-8133-3242","authenticated-orcid":false,"given":"Jemma G.","family":"Gasperoni","sequence":"first","affiliation":[{"name":"Department of Microbiology, Anatomy, Physiology and Pharmacology, and La Trobe Institute for Molecular Science La Trobe University  Bundoora Victoria Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0001-0070-9736","authenticated-orcid":false,"given":"Jarrad N.","family":"Fuller","sequence":"additional","affiliation":[{"name":"Department of Microbiology, Anatomy, Physiology and Pharmacology, and La Trobe Institute for Molecular Science La Trobe University  Bundoora Victoria Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0000-8799-0526","authenticated-orcid":false,"given":"April L.","family":"Lewis","sequence":"additional","affiliation":[{"name":"Department of Biochemistry and Chemistry, and La Trobe Institute for Molecular Science La Trobe University  Bundoora Victoria Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6846-9313","authenticated-orcid":false,"given":"Travis K.","family":"Johnson","sequence":"additional","affiliation":[{"name":"Department of Biochemistry and Chemistry, and La Trobe Institute for Molecular Science La Trobe University  Bundoora Victoria Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3191-852X","authenticated-orcid":false,"given":"Bert","family":"De Groef","sequence":"additional","affiliation":[{"name":"Department of Microbiology, Anatomy, Physiology and Pharmacology, and La Trobe Institute for Molecular Science La Trobe University  Bundoora Victoria Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5828-9992","authenticated-orcid":false,"given":"Sebastian","family":"Dworkin","sequence":"additional","affiliation":[{"name":"Department of Microbiology, Anatomy, Physiology and Pharmacology, and La Trobe Institute for Molecular Science La Trobe University  Bundoora Victoria Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,10,2]]},"reference":[{"key":"e_1_2_11_2_1","doi-asserted-by":"publisher","DOI":"10.1242\/bio.038661"},{"key":"e_1_2_11_3_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41598\u2010020\u201079923\u20106"},{"key":"e_1_2_11_4_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0279745"},{"key":"e_1_2_11_5_1","volume-title":"MIC\u2010Drop\u2010seq: Scalable Single\u2010Cell Phenotyping of Mutant Vertebrate Embryos","author":"Carey C. 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