The role of cilia during organogenesis in zebrafish

Liu, Junjun; Xie, Haibo; Wu, Mengfan; Hu, Yidan; Kang, Yunsi

doi:10.1098/rsob.230228

Ciliary length regulation by intraflagellar transport in zebrafish

Sun,

Chen,

Jin

et al. 2024

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How cells regulate the size of their organelles remains a fundamental question in cell biology. Cilia, with their simple structure and surface localization, provide an ideal model for investigating organelle size control. However, most studies on cilia length regulation are primarily performed on several single-celled organisms. In contrast, the mechanism of length regulation in cilia across diverse cell types within multicellular organisms remains a mystery. Similar to humans, zebrafish contain diverse types of cilia with variable lengths. Taking advantage of the transparency of zebrafish embryos, we conducted a comprehensive investigation into intraflagellar transport (IFT), an essential process for ciliogeneis. We observed IFT in multiple types of cilia with varying lengths. Remarkably, cilia exhibited variable IFT speeds in different cell types, with longer cilia exhibiting faster IFT speeds. The increased IFT speed in longer cilia was not due to changes in common factors that regulate IFT, such as motor selection, BBS proteins, or tubulin modification. Instead, longer cilia can organize larger IFT particles for faster transportation. Reducing the size of IFT particles can slow down IFT speed, resulting in shorter cilia. Our study presents an intriguing model of cilia length regulation via controlling IFT speed through the modulation of the size of the IFT complex. This discovery may provide further insights into our understanding of how organelle size is regulated in higher vertebrates.

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Using Zebrafish to Study Multiciliated Cell Development and Disease States

Nguyen,

Baker,

Rodriguez

et al. 2024

Cells

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Multiciliated cells (MCCs) serve many important functions, including fluid propulsion and chemo- and mechanosensing. Diseases ranging from rare conditions to the recent COVID-19 global health pandemic have been linked to MCC defects. In recent years, the zebrafish has emerged as a model to investigate the biology of MCCs. Here, we review the major events in MCC formation including centriole biogenesis and basal body docking. Then, we discuss studies on the role of MCCs in diseases of the brain, respiratory, kidney and reproductive systems, as well as recent findings about the link between MCCs and SARS-CoV-2. Next, we explore why the zebrafish is a useful model to study MCCs and provide a comprehensive overview of previous studies of genetic components essential for MCC development and motility across three major tissues in the zebrafish: the pronephros, brain ependymal cells and nasal placode. Taken together, here we provide a cohesive summary of MCC research using the zebrafish and its future potential for expanding our understanding of MCC-related disease states.

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Ciliary length regulation by intraflagellar transport in zebrafish

Sun,

Chen,

Jin

et al. 2024

eLife

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How cells regulate the size of their organelles remains a fundamental question in cell biology. Cilia, with their simple structure and surface localization, provide an ideal model for investigating organelle size control. However, most studies on cilia length regulation are primarily performed on several single-celled organisms. In contrast, the mechanism of length regulation in cilia across diverse cell types within multicellular organisms remains a mystery. Similar to humans, zebrafish contain diverse types of cilia with variable lengths. Taking advantage of the transparency of zebrafish embryos, we conducted a comprehensive investigation into intraflagellar transport (IFT), an essential process for ciliogeneis. We observed IFT in multiple types of cilia with varying lengths. Remarkably, cilia exhibited variable IFT speeds in different cell types, with longer cilia exhibiting faster IFT speeds. The increased IFT speed in longer cilia was not due to changes in common factors that regulate IFT, such as motor selection, BBS proteins, or tubulin modification. Instead, longer cilia can organize larger IFT particles for faster transportation. Reducing the size of IFT particles can slow down IFT speed, resulting in shorter cilia. Our study presents an intriguing model of cilia length regulation via controlling IFT speed through the modulation of the size of the IFT complex. This discovery may provide further insights into our understanding of how organelle size is regulated in higher vertebrates.

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The role of cilia during organogenesis in zebrafish

Cited by 5 publications

References 192 publications

Ciliary length regulation by intraflagellar transport in zebrafish

Ciliary length regulation by intraflagellar transport in zebrafish

Using Zebrafish to Study Multiciliated Cell Development and Disease States

Ciliary length regulation by intraflagellar transport in zebrafish

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