PUBLICATION
shox2 is required for vestibular statoacoustic neuron development
- Authors
- Laureano, A.S., Flaherty, K., Hinman, A.M., Jadali, A., Nakamura, T., Higashijima, S.I., Sabaawy, H.E., Kwan, K.Y.
- ID
- ZDB-PUB-230104-4
- Date
- 2022
- Source
- Biology Open 11(12): (Journal)
- Registered Authors
- Higashijima, Shin-ichi
- Keywords
- Inner ear, Statoacoustic ganglion neuron, Vestibular neuron
- Datasets
- GEO:GSE197270
- MeSH Terms
-
- Animals
- Ear, Inner*/innervation
- Neurogenesis
- Neurons
- Transcription Factors
- Zebrafish*/genetics
- Zebrafish Proteins/genetics
- PubMed
- 36594417 Full text @ Biol. Open
Citation
Laureano, A.S., Flaherty, K., Hinman, A.M., Jadali, A., Nakamura, T., Higashijima, S.I., Sabaawy, H.E., Kwan, K.Y. (2022) shox2 is required for vestibular statoacoustic neuron development. Biology Open. 11(12):.
Abstract
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.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping