International Journal of Marine Science, 2024, Vol.14, No.5, 332-340 http://www.aquapublisher.com/index.php/ijms 332 Feature Review Open Access Zebrafish as a Model for Studying Ciliary Development and Disease Fan Wang, Fei Zhao Aquatic Biology Research Center, Cuixi Academy of Biotechnology, Zhuji, 311800, Zhejiang, China Corresponding author: fei.zhao@cuixi.org International Journal of Marine Science, 2024, Vol.14, No.5, doi: 10.5376/ijms.2024.14.0037 Received: 18 Aug., 2024 Accepted: 28 Sep., 2024 Published: 26 Oct., 2024 Copyright © 2024 Wang and Zhao, This is an open access article published under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproductio4n in any medium, provided the original work is properly cited. Preferred citation for this article: Wang F., and Zhao F., 2024, Zebrafish as a model for studying ciliary development and disease, International Journal of Marine Science, 14(5): 332-340 (doi: 10.5376/ijms.2024.14.0037) Abstract Cilia play crucial roles in numerous biological processes, from cell signaling to tissue homeostasis, and their dysfunction can lead to a group of disorders known as ciliopathies. Zebrafish (Danio rerio), due to its genetic tractability and transparency during early development, has become an important model organism for studying ciliary development and related diseases. This study analyzes the stages of ciliary development in zebrafish, including tissue-specific processes and the role of key signaling pathways, and explores how zebrafish models contribute to understanding various ciliopathies. It emphasizes genetic manipulation to induce ciliary defects and phenotypic analysis, and describes key observational techniques in zebrafish ciliary research, including high-resolution imaging, genetic markers, and fluorescent reporters. Case studies demonstrate the application of zebrafish in studying human ciliopathies, such as Joubert syndrome, Bardet-Biedl syndrome, and nephronophthisis, as well as kidney and liver ciliopathies. It is expected that this study will provide reference value for future research on ciliary related diseases, promote the understanding of the pathological mechanisms of fibrotic disorders, and develop treatment strategies. Keywords Ciliarydevelopment; Zebrafishmodel; Ciliopathies; Geneticmanipulation; Observationaltechniques 1 Introduction Cilia are microtubule-based organelles that extend from the surface of eukaryotic cells and play crucial roles in cellular signaling, motility, and sensory functions. They are involved in various physiological processes, including signal transduction, organ development, and tissue homeostasis. Abnormalities in ciliary structure and function can lead to a group of human diseases known as ciliopathies, which affect multiple organs and systems (Blacque et al., 2017; Zhang et al., 2022). Cilia are essential for the proper functioning of sensory structures such as the eye, ear, and nose, and are also involved in developmental processes like left-right asymmetry formation and limb morphogenesis (Leventea et al., 2016). Zebrafish (Danio rerio) have emerged as a powerful model organism for studying ciliary development and related diseases due to their genetic similarity to humans and the conservation of ciliary structure and function across vertebrates. Zebrafish offer several advantages for ciliary research, including their rapid development, transparency during embryonic stages, and the availability of sophisticated genetic and imaging techniques (Sedykh et al., 2016). These features make zebrafish an ideal model for investigating the roles of cilia in organogenesis and for modeling human ciliopathies (Pinto et al., 2021; Liu et al., 2023). This study will discuss the various types of cilia present in zebrafish, their roles in organogenesis, and the advantages of using zebrafish for ciliary research. It will also highlight recent advances in the field, including the development of transgenic zebrafish lines for in vivo visualization of cilia and the identification of new roles for ciliary proteins. This study emphasizes the importance of zebrafish as a model organism in cilia research, promoting our understanding of fibrosis. 2 Ciliary Development in Zebrafish 2.1 Early stages of ciliary formation Ciliary formation in zebrafish begins during early embryogenesis, where cilia play a crucial role in cellular signaling, tissue morphogenesis, and body patterning. The Nlz1 protein has been identified as a key player in this process. Knockdown of nlz1 in zebrafish results in abnormal cell specification in Kupffer's vesicle (KV) and a
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