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More than thirty Joubert syndrome genes have been identified, including<jats:italic>ARL13B<\/jats:italic>. However, its role in cerebellar development remains unexplored. We found that knockdown or knockout of<jats:italic>arl13b<\/jats:italic>impaired balance and locomotion in zebrafish larvae. Granule cells were selectively reduced in the corpus cerebelli, a structure homologous to the mammalian vermis. Purkinje cell progenitors were also selectively disturbed dorsomedially. The expression of<jats:italic>atoh1<\/jats:italic>and<jats:italic>ptf1<\/jats:italic>, proneural genes of granule and Purkinje cells, respectively, were selectively down-regulated along the dorsal midline of the cerebellum. Moreover,<jats:italic>wnt1<\/jats:italic>, which is transiently expressed early in cerebellar development, was selectively reduced. Intriguingly, activating Wnt signaling partially rescued the granule cell defects in<jats:italic>arl13b<\/jats:italic>mutants. These findings suggested that Arl13b is necessary for the early development of cerebellar granule and Purkinje cells. The<jats:italic>arl13b<\/jats:italic>-deficient zebrafish can serve as a model organism for studying Joubert syndrome.<\/jats:p>","DOI":"10.1007\/s12264-020-00554-y","type":"journal-article","created":{"date-parts":[[2020,8,18]],"date-time":"2020-08-18T18:03:33Z","timestamp":1597773813000},"page":"1023-1034","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":21,"title":["The Joubert Syndrome Gene arl13b is Critical for Early Cerebellar Development in Zebrafish"],"prefix":"10.1007","volume":"36","author":[{"given":"Jian","family":"Zhu","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Han-Tsing","family":"Wang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yu-Rong","family":"Chen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ling-Ya","family":"Yan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ying-Ying","family":"Han","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ling-Yan","family":"Liu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ying","family":"Cao","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zhi-Zhi","family":"Liu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4938-0203","authenticated-orcid":false,"given":"Hong A.","family":"Xu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2020,8,18]]},"reference":[{"key":"554_CR1","doi-asserted-by":"crossref","first-page":"894","DOI":"10.1016\/S1474-4422(13)70136-4","volume":"12","author":"M Romani","year":"2013","unstructured":"Romani M, Micalizzi A, Valente EM. 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