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We identified the short stature homeobox domain 2 (shox2) transcription factor that is required for vestibular neuron development. shox2 transcripts are initially localized to the otic placode of the developing inner ear where neurosensory progenitors reside. To study shox2 function, we generated CRISPR-mediated mutant shox2 fish. Mutant embryos display behaviors associated with vestibular deficits and showed reduced number of anterior statoacoustic ganglion neurons that innervate the utricle, the vestibular organ in zebrafish. Moreover, a shox2-reporter fish showed labeling of developing statoacoustic ganglion neurons in the anterior macula of the otic vesicle. Single cell RNA-sequencing of cells from the developing otic vesicle of shox2 mutants revealed altered otic progenitor profiles, while single molecule in situ assays showed deregulated levels of transcripts in developing neurons. This study implicates a role for shox2 in development of vestibular but not auditory statoacoustic ganglion neurons.<\/jats:p>","DOI":"10.1242\/bio.059599","type":"journal-article","created":{"date-parts":[[2023,1,3]],"date-time":"2023-01-03T09:16:08Z","timestamp":1672737368000},"source":"Crossref","is-referenced-by-count":7,"title":["<i>shox2<\/i> is required for vestibular statoacoustic neuron development"],"prefix":"10.1242","volume":"11","author":[{"given":"Alejandra S.","family":"Laureano","sequence":"first","affiliation":[{"name":"Rutgers University 1 Department of Cell Biology & Neuroscience , , Piscataway, NJ 08854 , USA"},{"name":"Stem Cell Research Center and Keck Center for Collaborative Neuroscience, Rutgers University 2 , NJ 08854 , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kathleen","family":"Flaherty","sequence":"additional","affiliation":[{"name":"Rutgers University 3 Department of Comparative Medicine Resources , , Piscataway, NJ 08854 , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Anna-Maria","family":"Hinman","sequence":"additional","affiliation":[{"name":"Rutgers University 1 Department of Cell Biology & Neuroscience , , Piscataway, NJ 08854 , USA"},{"name":"Stem Cell Research Center and Keck Center for Collaborative Neuroscience, Rutgers University 2 , NJ 08854 , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Azadeh","family":"Jadali","sequence":"additional","affiliation":[{"name":"Rutgers University 1 Department of Cell Biology & Neuroscience , , Piscataway, NJ 08854 , USA"},{"name":"Stem Cell Research Center and Keck Center for Collaborative Neuroscience, Rutgers University 2 , NJ 08854 , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0183-1685","authenticated-orcid":false,"given":"Tetsuya","family":"Nakamura","sequence":"additional","affiliation":[{"name":"Rutgers University 4 Department of Genetics , , Piscataway, NJ 08854 , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shin-ichi","family":"Higashijima","sequence":"additional","affiliation":[{"name":"Institutes of Natural Sciences, Exploratory Research Center on Life and Living Systems 5 , Okazaki, Aichi 444-8787 , Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7976-303X","authenticated-orcid":false,"given":"Hatim E.","family":"Sabaawy","sequence":"additional","affiliation":[{"name":"University of Colorado Anschutz Medical Campus 6 Department of Medicine, Division of Medical Oncology , , Aurora, CO 80045 , USA"},{"name":"Department of Medicine RBHS-Robert Wood Johnson Medical School, Rutgers University 7 , Piscataway, NJ 08854 , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8617-5543","authenticated-orcid":false,"given":"Kelvin Y.","family":"Kwan","sequence":"additional","affiliation":[{"name":"Rutgers University 1 Department of Cell Biology & Neuroscience , , Piscataway, NJ 08854 , USA"},{"name":"Stem Cell Research Center and Keck Center for Collaborative Neuroscience, Rutgers University 2 , NJ 08854 , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"237","published-online":{"date-parts":[[2022,12,21]]},"reference":[{"key":"2023010309112027000_BIO059599C1","doi-asserted-by":"publisher","first-page":"483","DOI":"10.1387\/ijdb.072343ga","article-title":"Establishment of a proneural field in the inner ear","volume":"51","author":"Abello","year":"2007","journal-title":"Int. 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