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Updated: May 16, 2026

2D and 3D Human Induced Pluripotent Stem Cell-Based Models to Dissect Primary Cilium Involvement during Neocortical Development
Published on: March 25, 2022
[Transcriptional control of ciliogenesis in animal development]
Julie Jerber1, Joëlle Thomas, Bénédicte Durand
1Centre de Genetique et de Physiologie Moleculare et Cellulaire, Universite Lyon, Villeurbanne, Lyon, France.
Abstract:
Cilia and flagella are eukaryotic organelles with a conserved structure and function from unicellular organisms to human. In animals, different types of cilia can be found and cilia assembly during development is a highly dynamic process. Ciliary defects in human lead to a wide spectrum of diseases called ciliopathies. Understanding the molecular mechanisms that govern dynamic cilia assembly during development and in different tissues in metazoans is an important biological challenge. The FOXJ1 (Forkhead Box J1) and RFX (Regulatory Factor X) family of transcription factors have been shown to be important factors in ciliogenesis control. FOXJ1 proteins are required for motile ciliogenesis in vertebrates. By contrast, RFX proteins are essential to assemble both primary and motile cilia through the regulation of specific sets of genes such as those encoding intraflagellar transport components. Recently, new actors with more specific roles in cilia biogenesis and physiology have also been discovered. All these factors are subject to complex regulation, allowing for the dynamic and specific regulation of ciliogenesis in metazoans.
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