International Journal of Marine Science, 2024, Vol.14, No.5, 332-340 http://www.aquapublisher.com/index.php/ijms 336 fluorescent vital dyes and transgenic cell type-specific fluorescent reporters have been used to identify different cell types and visualize subcellular structures in primary cell cultures of zebrafish embryos (Sassen et al., 2017). These techniques facilitate the study of ciliary function and related phenotypes in live zebrafish embryos, contributing to our understanding of ciliopathies and other cilia-related disorders. Figure 2 Three-dimensional imaging analysis helps to orient and localize the OP of zebrafish for TEM studies (Adopted from Pinto et al., 2021) Image caption: (A, B) Immunofluorescent labelling with anti-acetylated α-tubulin shows the distribution of multiciliated cells in the OP of 4 dpf larvae. (C, D) 3D surface reconstructions from the respective OPs revealing the concave morphology of the organ when rotated. Anti-Acetylated α-tubulin immunofluorescence in magenta and DAPI in cyan. Scale bar 20 μm (Adopted from Pinto et al., 2021) 6 Case Study: Zebrafish in the Study of Human Ciliopathies 6.1 Application of zebrafish in understanding joubert syndrome Joubert Syndrome (JS) is a neurodevelopmental disorder characterized by a distinctive brain malformation known as the "molar tooth sign" on axial MRI, along with symptoms such as hypotonia, ataxia, and abnormal eye movements. Zebrafish models have been instrumental in understanding the genetic and molecular mechanisms underlying JS. For instance, zebrafish models have been used to study the role of ARMC9, a basal body protein, in JS. Mutations in ARMC9 have been shown to cause typical ciliopathy phenotypes in zebrafish, such as curved body shape and retinal dystrophy, thereby supporting its role in JS (Weghe et al., 2017). Zebrafish models have helped elucidate the tissue-specific roles of proteins implicated in JS, revealing that different genes can cause distinct phenotypes in various organs (Rusterholz et al., 2022). These models are invaluable for exploring the pathomechanisms of JS and developing potential therapeutic strategies.
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