ftr82 is necessary for hair cell morphogenesis and auditory function during zebrafish development

Yin, Guoli; Qian, Fuping; Jia, Yulin; Wang, Ziyang; Wang, Xin; Liu, Dong; Wang, Cheng

doi:10.1016/j.jgg.2022.11.008

Cited by 4 publications

(5 citation statements)

References 47 publications

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“…The transgenic zebrafish line Tg(Brn3c:mGFP) and wild-type AB strain were bred at 28.5 °C and maintained following the standard procedures as described in previous protocols [ 9 ]. Spawning embryos were raised with E3 medium in a constant temperature incubator at 28.5 °C.…”

Section: Methodsmentioning

confidence: 99%

“…The transgenic zebrafish line Tg(Brn3c:mGFP) and wild-type AB strain were bred at 28.5 • C and maintained following the standard procedures as described in previous protocols [9]. Spawning embryos were raised with E3 medium in a constant temperature incubator at 28.5 • C. To avoid pigment formation, embryo medium was replaced with 0.2 mM PTU (1-phenyl-2-thiourea, Sigma, Saint Louis, MO, USA) solution at 20 h post-fertilization (hpf).…”

Section: Zebrafish Lines and Maintenancementioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

“…In auditory research, the model organisms of birds, chicks, zebrafish, mice, and rats are commonly used [ 4 , 9 ]. Mammalian animals have a typical structure of cochlea similar to humans, however, the complicated anatomy procedures and low throughput of the mammalian animal models bring about certain obstacles in application [ 9 ]. Zebrafish ( Danio rerio ), though devoid of cochlea, has auditory organs in the inner ear, namely the otic vesicle and the lateral line (LL) sensory system, which makes it suitable for using as an economic non-mammalian vertebrate model [ 10 , 11 , 12 ].…”

Section: Introductionmentioning

confidence: 99%

“…Sensory HCs in zebrafish are tightly surrounded by supporting cells which is structurally and functionally similar to the mammalian inner ear. Combined with the innate characteristics of high fecundity, easy genetic manipulation and imaging in vivo, zebrafish has been widely used in drugs ototoxicity assessment, otoprotective agents screening, hearing-related gene functional identification and the mechanism of HC development and regeneration investigation [ 9 , 10 , 11 , 12 , 13 , 14 ].…”

Section: Introductionmentioning

confidence: 99%

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The ndrg2 Gene Regulates Hair Cell Morphogenesis and Auditory Function during Zebrafish Development

Wang

Zheng

et al. 2023

IJMS

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Damages of sensory hair cells (HCs) are mainly responsible for sensorineural hearing loss, however, its pathological mechanism is not yet fully understood due to the fact that many potential deafness genes remain unidentified. N-myc downstream-regulated gene 2 (ndrg2) is commonly regarded as a tumor suppressor and a cell stress-responsive gene extensively involved in cell proliferation, differentiation, apoptosis and invasion, while its roles in zebrafish HC morphogenesis and hearing remains unclear. Results of this study suggested that ndrg2 was highly expressed in the HCs of the otic vesicle and neuromasts via in situ hybridization and single-cell RNA sequencing. Ndrg2 loss-of-function larvae showed decreased crista HCs, shortened cilia, and reduced neuromasts and functional HCs, which could be rescued by the microinjection of ndrg2 mRNA. Moreover, ndrg2 deficiency induced attenuated startle response behaviors to sound vibration stimuli. Mechanistically, there were no detectable HC apoptosis and supporting cell changes in the ndrg2 mutants, and HCs were capable of recovering by blocking the Notch signaling pathway, suggesting that ndrg2 was implicated in HC differentiation mediated by Notch. Overall, our study demonstrates that ndrg2 plays crucial roles in HC development and auditory sensory function utilizing the zebrafish model, which provides new insights into the identification of potential deafness genes and regulation mechanism of HC development.

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Section: Methodsmentioning

confidence: 99%

Section: Zebrafish Lines and Maintenancementioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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The ndrg2 Gene Regulates Hair Cell Morphogenesis and Auditory Function during Zebrafish Development

Wang

Zheng

et al. 2023

IJMS

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Zebrafish in-vivo study reveals deleterious activity of human TBC1D24 genetic variants linked with autosomal dominant hearing loss

Sarosiak,

Jędrychowska,

Oziębło

et al. 2025

Biochimica et Biophysica Acta (BBA) - Molecular Basis of Diseas

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Knockout of dhx38 Causes Inner Ear Developmental Defects in Zebrafish

Ren,

Chen,

Dai

et al. 2024

Biomedicines

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Background: Alternative splicing is essential for the physiological and pathological development of the inner ear. Disruptions in this process can result in both syndromic and non-syndromic forms of hearing loss. DHX38, a DEAH box RNA helicase, is integral to pre-mRNA splicing regulation and plays critical roles in development, cell differentiation, and stem cell maintenance. However, its specific role in inner ear development remains undefined. Here, we utilized a dhx38 knockout zebrafish model to monitor the ear morphology and elucidate a crucial role for DHX38 in the development of the zebrafish inner ear. Methods: Bright-field morphological analysis and in situ hybridization were performed to observe ear morphology changes. Immunofluorescence and semi-quantitative RT-PCR were employed to test apoptotic cells and abnormal splicing. Results: The dhx38-/- mutant zebrafish showed significant inner ear impairments, including decrescent otocysts, absent semicircular canal protrusion, and smaller otoliths. These structural abnormalities were accompanied by substantial DNA damage and p53-dependent apoptosis within the inner ear cells. Alternative splicing analysis showed that genes related to DNA damage repair and inner ear morphogenesis are abnormal in dhx38 knockout mutants. In summary, we suggest that dhx38 promotes cell survival during the inner ear development of zebrafish by ensuring the correct splicing of genes related to DNA damage repair.

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ftr82 is necessary for hair cell morphogenesis and auditory function during zebrafish development

Cited by 4 publications

References 47 publications

The ndrg2 Gene Regulates Hair Cell Morphogenesis and Auditory Function during Zebrafish Development

The ndrg2 Gene Regulates Hair Cell Morphogenesis and Auditory Function during Zebrafish Development

Zebrafish in-vivo study reveals deleterious activity of human TBC1D24 genetic variants linked with autosomal dominant hearing loss

Knockout of dhx38 Causes Inner Ear Developmental Defects in Zebrafish

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