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Zebrafish, a popular model for behavioral studies, has six neurexin genes, but their functions in embryogenesis and behavior remain largely unknown. We have previously reported that nrxn2a is aberrantly spliced and specifically dysregulated in motor neurons (MNs) in models of spinal muscular atrophy. In this study, we generated nrxn2aa\u2212\/\u2212 mutants by CRISPR\/Cas9 to understand nrxn2aa function at the zebrafish neuromuscular junction (NMJ) and to determine the effects of its deficiency on adult behavior. Homozygous mutant embryos derived from heterozygous parents did not show obvious defects in axon outgrowth or synaptogenesis of MNs. In contrast, maternal-zygotic (MZ) nrxn2aa\u2212\/\u2212 mutants displayed extensively branched axons and defective MNs, suggesting a cell-autonomous role for maternally provided nrxn2aa in MN development. Analysis of the NMJs revealed enlarged choice points in MNs of mutant larvae and reduced co-localization of pre- and post-synaptic terminals, indicating impaired synapse formation. Severe early NMJ defects partially recovered in late embryos when mutant transcripts became strongly upregulated. Ultimately, however, the induced defects resulted in muscular atrophy symptoms in adult MZ mutants. Zygotic homozygous mutants developed normally but displayed increased anxiety at adult stages. Together, our data demonstrate an essential role for maternal nrxn2aa in NMJ synapse establishment, while zygotic nrxn2aa expression appears dispensable for synapse maintenance. The viable nrxn2aa\u2212\/\u2212 mutant furthermore serves as a novel model to study how an increase in anxiety-like behaviors impacts other deficits.<\/jats:p>","DOI":"10.1093\/hmg\/ddaa260","type":"journal-article","created":{"date-parts":[[2020,12,2]],"date-time":"2020-12-02T14:25:07Z","timestamp":1606919107000},"page":"3765-3780","source":"Crossref","is-referenced-by-count":31,"title":["A Neurexin2aa deficiency results in axon pathfinding defects and increased anxiety in zebrafish"],"prefix":"10.1093","volume":"29","author":[{"given":"Angela","family":"Koh","sequence":"first","affiliation":[{"name":"Department of Biological Sciences and Centre for Bioimaging Sciences, National University of Singapore, Singapore 117543, Singapore"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shijie","family":"Tao","sequence":"additional","affiliation":[{"name":"Department of Biological Sciences and Centre for Bioimaging Sciences, National University of Singapore, Singapore 117543, Singapore"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yun","family":"Jing Goh","sequence":"additional","affiliation":[{"name":"Department of Biological Sciences and Centre for Bioimaging Sciences, National University of Singapore, Singapore 117543, Singapore"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vindhya","family":"Chaganty","sequence":"additional","affiliation":[{"name":"Department of Biological Sciences and Centre for Bioimaging Sciences, National University of Singapore, Singapore 117543, Singapore"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kelvin","family":"See","sequence":"additional","affiliation":[{"name":"Department of Biological Sciences and Centre for Bioimaging Sciences, National University of Singapore, Singapore 117543, Singapore"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kathiresan","family":"Purushothaman","sequence":"additional","affiliation":[{"name":"Reproductive Genomics Group, Temasek Life Sciences Laboratory, Singapore 117604, Singapore"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"L\u00e1szl\u00f3","family":"Orb\u00e1n","sequence":"additional","affiliation":[{"name":"Reproductive Genomics Group, Temasek Life Sciences Laboratory, Singapore 117604, Singapore"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ajay S","family":"Mathuru","sequence":"additional","affiliation":[{"name":"Yale-NUS College, Singapore 138527, Singapore"},{"name":"Institute of Molecular and Cell Biology, Singapore 138673, Singapore"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Thorsten","family":"Wohland","sequence":"additional","affiliation":[{"name":"Department of Biological Sciences and Centre for Bioimaging Sciences, National University of Singapore, Singapore 117543, Singapore"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4688-6241","authenticated-orcid":false,"given":"Christoph","family":"Winkler","sequence":"additional","affiliation":[{"name":"Department of Biological Sciences and Centre for Bioimaging Sciences, National University of Singapore, Singapore 117543, Singapore"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2020,12,4]]},"reference":[{"key":"2021020414452444800_ref1","doi-asserted-by":"crossref","first-page":"50","DOI":"10.1126\/science.1621094","article-title":"Neurexins: synaptic cell surface proteins related to the alpha-latrotoxin receptor and laminin","volume":"257","author":"Ushkaryov","year":"1992","journal-title":"Science"},{"key":"2021020414452444800_ref2","doi-asserted-by":"crossref","first-page":"745","DOI":"10.1016\/j.cell.2017.10.024","article-title":"Synaptic neurexin complexes: a molecular code for the logic of neural circuits","volume":"171","author":"Sudhof","year":"2017","journal-title":"Cell"},{"key":"2021020414452444800_ref3","doi-asserted-by":"crossref","first-page":"11987","DOI":"10.1016\/S0021-9258(17)32671-6","article-title":"Conserved domain structure of beta-neurexins. 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