Zebrafish <i>jam‐b2</i> Gal4‐enhancer trap line recapitulates endogenous <i>jam‐b2</i> expression in extraocular muscles

Matsui, Hirokazu; Dorigo, Alessandro; Buchberger, Astrid; Hocking, Jennifer C.; Distel, Martin; Köster, Reinhard W.

doi:10.1002/dvdy.24347

Cited by 3 publications

(3 citation statements)

References 34 publications

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“…Whole-mount in situ hybridization on zebrafish embryos and larvae was performed as described previously with slight modifications ( 69 ). Briefly, embryos and larvae were fixed in 4% paraformaldehyde (PFA)/phospahte buffered saline (PBS) overnight, dehydrated in 25%, 50%, and 75% methanol/PBS and stored in 100% methanol at −20 °C overnight or until use.…”

Section: Methodsmentioning

confidence: 99%

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A novel inhibitor rescues cerebellar defects in a zebrafish model of Down syndrome–associated kinase Dyrk1A overexpression

Buchberger

Schepergerdes

Flaßhoff

et al. 2021

Journal of Biological Chemistry

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The highly conserved dual-specificity tyrosine phosphorylation–regulated kinase 1A (Dyrk1A) plays crucial roles during central nervous system development and homeostasis. Furthermore, its hyperactivity is considered responsible for some neurological defects in individuals with Down syndrome. We set out to establish a zebrafish model expressing human Dyrk1A that could be further used to characterize the interaction between Dyrk1A and neurological phenotypes. First, we revealed the prominent expression of dyrk1a homologs in cerebellar neurons in the zebrafish larval and adult brains. Overexpression of human dyrk1a in postmitotic cerebellar Purkinje neurons resulted in a structural misorganization of the Purkinje cells in cerebellar hemispheres and a compaction of this cell population. This impaired Purkinje cell organization was progressive, leading to an age-dependent dispersal of Purkinje neurons throughout the cerebellar molecular layer with larval swim deficits resulting in miscoordination of swimming and reduced exploratory behavior in aged adults. We also found that the structural misorganization of the larval Purkinje cell layer could be rescued by pharmacological treatment with Dyrk1A inhibitors. We further reveal the in vivo efficiency of a novel selective Dyrk1A inhibitor, KuFal194. These findings demonstrate that the zebrafish is a well-suited vertebrate organism to genetically model severe neurological diseases with single cell type specificity. Such models can be used to relate molecular malfunction to cellular deficits, impaired tissue formation, and organismal behavior and can also be used for pharmacological compound testing and validation.

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Section: Methodsmentioning

confidence: 99%

“…Immunohistochemistry of zebrafish embryos or brain frozen sections were processed using standard protocols described previously ( 43 , 69 ).…”

Section: Methodsmentioning

confidence: 99%

A novel inhibitor rescues cerebellar defects in a zebrafish model of Down syndrome–associated kinase Dyrk1A overexpression

Buchberger

Schepergerdes

Flaßhoff

et al. 2021

Journal of Biological Chemistry

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“…The OKR assay set-up was detailed in 89.9% of studies, with a further 5.5% referencing another paper's methodology. A total of 3.7% do not mention OKR methodology [58,[64][65][66].…”

Section: Okr Assay Set-upmentioning

confidence: 99%

Zebrafish Optokinetic Reflex: Minimal Reporting Guidelines and Recommendations

Rodwell,

Patil,

Kuht

et al. 2023

Biology

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Optokinetic reflex (OKR) assays in zebrafish models are a valuable tool for studying a diverse range of ophthalmological and neurological conditions. Despite its increasing popularity in recent years, there are no clear reporting guidelines for the assay. Following reporting guidelines in research enhances reproducibility, reduces bias, and mitigates underreporting and poor methodologies in published works. To better understand optimal reporting standards for an OKR assay in zebrafish, we performed a systematic literature review exploring the animal, environmental, and technical factors that should be considered. Using search criteria from three online databases, a total of 109 research papers were selected for review. Multiple crucial factors were identified, including larval characteristics, sample size, fixing method, OKR set-up, distance of stimulus, detailed stimulus parameters, eye recording, and eye movement analysis. The outcome of the literature analysis highlighted the insufficient information provided in past research papers and the lack of a systematic way to present the parameters related to each of the experimental factors. To circumvent any future errors and champion robust transparent research, we have created the zebrafish optokinetic (ZOK) reflex minimal reporting guideline.

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The use of fish models to study human neurological disorders

Matsui

2017

Neuroscience Research

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Zebrafish jam‐b2 Gal4‐enhancer trap line recapitulates endogenous jam‐b2 expression in extraocular muscles

Cited by 3 publications

References 34 publications

A novel inhibitor rescues cerebellar defects in a zebrafish model of Down syndrome–associated kinase Dyrk1A overexpression

A novel inhibitor rescues cerebellar defects in a zebrafish model of Down syndrome–associated kinase Dyrk1A overexpression

Zebrafish Optokinetic Reflex: Minimal Reporting Guidelines and Recommendations

The use of fish models to study human neurological disorders

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