Using zebrafish as a model to study the role of epigenetics in hearing loss

He, Yingzi; Bao, Beier; Li, Huawei

doi:10.1080/17460441.2017.1340270

Cited by 19 publications

(17 citation statements)

References 80 publications

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“…Moreover, numerous genes as ATOH1, POU4F3, GFI1 and other genes, are expressed in the sensory cells of zebrafish, mainly during embryonic development, also expressed in mammals [20]. It is well known that zebrafish is able to regenerate, within a few days, the hair cells damaged or lost by acoustic trauma, chemical exposure or genetics [21][22][23][24][25]. Therefore, based on the peculiar ability of zebrafish to regenerate new hair cells, this study was undertaken to analyze the cellular localization of BDNF and its specific receptor TrkB location in the inner ear of the zebrafish.…”

Section: Introductionmentioning

confidence: 99%

Expression and Localization of BDNF/TrkB System in the Zebrafish Inner Ear

Germanà¹,

Guerrera²,

Laurà³

et al. 2020

IJMS

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Brain-derived neurotrophic factor (BDNF), a member of the neurotrophin family, is involved in multiple and fundamental functions of the central and peripheral nervous systems including sensory organs. Despite recent advances in knowledge on the functional significance of BDNF and TrkB in the regulation of the acoustic system of mammals, the localization of BDNF/TrkB system in the inner ear of zebrafish during development, is not well known. Therefore, the goal of the present study is to analyze the age-dependent changes using RT-PCR, Western Blot and single and double immunofluorescence of the BDNF and its specific receptor in the zebrafish inner ear. The results showed the mRNA expression and the cell localization of BDNF and TrkB in the hair cells of the crista ampullaris and in the neuroepithelium of the utricle, saccule and macula lagena, analyzed at different ages. Our results demonstrate that the BDNF/TrkB system is present in the sensory cells of the inner ear, during whole life. Therefore, this system might play a key role in the development and maintenance of the hair cells in adults, suggesting that the zebrafish inner ear represents an interesting model to study the involvement of the neurotrophins in the biology of sensory cells

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Section: Introductionmentioning

confidence: 99%

Expression and Localization of BDNF/TrkB System in the Zebrafish Inner Ear

Germanà¹,

Guerrera²,

Laurà³

et al. 2020

IJMS

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“…Several studies have shown the presence of similar LCs in zebrafish ( He et al, 2017 ; Lin et al, 2019 ). It is still unclear whether these cells are actually LCs, as the ontogenesis of Langerin/CD207 in zebrafish is not known.…”

Section: Langerin/cd207mentioning

confidence: 99%

Marking vertebrates langerhans cells, from fish to mammals

et al. 2020

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Langerhans cells (LCs) are specialized dendritic cells (DCs) that play a defense role in recognizing foreign antigens, in tissue where antigenic exposures occur, as in the skin and mucous membranes. LCs are able to continuously move within the tissues thanks to dendritic contraction and distension performing their surveillance and/or phagocytosis role. These cells are characterized by the presence of Birbeck granules in their cytoplasm, involved in endocytosis. LCs have been characterized in several classes of vertebrates, from fish to mammals using different histological and molecular techniques. The aim of the present review is to define the state of art and the need of information about immunohistochemical markers of LCs in different classes of vertebrates. The most used immunohistochemical (IHC) markers are Langerin/CD207, CD1a, S-100 and TLR. These IHC markers are described in relation to their finding in different vertebrate classes with phylogenetical considerations. Among the four markers, Langerin/CD207 and TLR have the widest spectrum of cross reactivity in LCs.

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“…Furthermore, zebrafish shares high genetic homology with human in terms of the genes and signalling pathways involved in the formation and maintenance of the auditory systems of zebrafish and mammals. Therefore, zebrafish is an appropriate model for exploring human hearing loss …”

Section: Introductionmentioning

confidence: 99%

“…Therefore, zebrafish is an appropriate model for exploring human hearing loss. 11 2 | MATERIALS AND METHODS…”

mentioning

confidence: 99%

Novel TRRAP mutation causes autosomal dominant non‐syndromic hearing loss

Xia

et al. 2019

Clinical Genetics

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Hereditary non‐syndromic hearing loss is the most common inherited sensory defect in humans. More than 40 genes have been identified as causative genes for autosomal dominant non‐syndromic hearing loss (ADNSHL), but there are many other candidate genes that remain to be discovered. We aimed to identify the causative gene mutation for post‐lingual progressive ADNSHL in a Chinese family. Whole‐exome sequencing, bioinformatic analysis, and Sanger sequencing were used to verify the co‐segregation of a novel pathogenic variant (NM_ 001244580, c.511C>T, p.Arg171Cys) in the TRansformation/tRanscription domain‐Associated Protein gene associated with hearing loss in a three‐generation Chinese family with ADNSHL). Additionally, three more novel variants of transformation/transcription domain associated protein (TRRAP) were detected in 66 sporadic cases of hearing loss. Morpholino oligonucleotides knockdown and clustered regularly interspaced short palindromic repeats/Cas9 knockout zebrafish were constructed to validate the genetic findings. Knockdown or knockout of TRRAP resulted in significant defects in the inner ear of zebrafish, indicating that TRRAP plays an important role in inner ear development. In conclusion, TRRAP (NM_ 001244580, c.511C>T, p.Arg171Cys) co‐segregated with hearing loss in a Chinese family with ADNSHL, and TRRAP deficiency caused hearing disability in zebrafish, suggesting TRRAP is a gene associated with ADNSHL.

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Using zebrafish as a model to study the role of epigenetics in hearing loss

Cited by 19 publications

References 80 publications

Expression and Localization of BDNF/TrkB System in the Zebrafish Inner Ear

Expression and Localization of BDNF/TrkB System in the Zebrafish Inner Ear

Marking vertebrates langerhans cells, from fish to mammals

Novel TRRAP mutation causes autosomal dominant non‐syndromic hearing loss

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