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shox2 is required for vestibular statoacoustic neuron development.
Laureano, Alejandra S; Flaherty, Kathleen; Hinman, Anna-Maria; Jadali, Azadeh; Nakamura, Tetsuya; Higashijima, Shin-Ichi; Sabaawy, Hatim E; Kwan, Kelvin Y.
Afiliación
  • Laureano AS; Department of Cell Biology & Neuroscience, Rutgers University, Piscataway, NJ 08854, USA.
  • Flaherty K; Stem Cell Research Center and Keck Center for Collaborative Neuroscience, Rutgers University, NJ 08854, USA.
  • Hinman AM; Department of Comparative Medicine Resources, Rutgers University, Piscataway, NJ 08854, USA.
  • Jadali A; Department of Cell Biology & Neuroscience, Rutgers University, Piscataway, NJ 08854, USA.
  • Nakamura T; Stem Cell Research Center and Keck Center for Collaborative Neuroscience, Rutgers University, NJ 08854, USA.
  • Higashijima SI; Department of Cell Biology & Neuroscience, Rutgers University, Piscataway, NJ 08854, USA.
  • Sabaawy HE; Stem Cell Research Center and Keck Center for Collaborative Neuroscience, Rutgers University, NJ 08854, USA.
  • Kwan KY; Department of Genetics, Rutgers University, Piscataway, NJ 08854, USA.
Biol Open ; 11(12)2022 12 15.
Article en En | MEDLINE | ID: mdl-36594417
Homeobox genes act at the top of genetic hierarchies to regulate cell specification and differentiation during embryonic development. 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.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Pez Cebra / Oído Interno Tipo de estudio: Prognostic_studies Límite: Animals Idioma: En Revista: Biol Open Año: 2022 Tipo del documento: Article País de afiliación: Estados Unidos Pais de publicación: Reino Unido
Texto completo
Añadir a Mi BVS
Imprimir
XML
PubMed Links
Buscar en Google

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Pez Cebra / Oído Interno Tipo de estudio: Prognostic_studies Límite: Animals Idioma: En Revista: Biol Open Año: 2022 Tipo del documento: Article País de afiliación: Estados Unidos Pais de publicación: Reino Unido