Abstract

Primary cilia are present on almost all vertebrate cells, and they have diverse functions in distinct tissues. Cilia are important for sensation in multiple capacities in contexts as different as the retina, kidney, and inner ear. In addition to these roles, cilia play a critical part in various developmental processes. Of particular importance is the development of the neural tube, where cilia are essential for the transduction of the Sonic Hedgehog (Shh) signaling pathway that specifies neuronal cell fates. This relationship is well established and is the most recognizable function for cilia in the neural tube, but it may be part of a larger picture. Here, we discuss the links between cilia and Shh signaling, as well as suggesting additional roles for cilia, and mechanisms for their placement, in the neural tube.

Highlights

  • Primary cilia are present on almost all vertebrate cells, and they have diverse functions in distinct tissues

  • Cilia are required for Sonic Hedgehog signaling and neural tube patterning Patterning of the neural tube and correct cell fate specification are integral parts of proper development, and tight control of the Shh signaling pathway is required for appropriate ventral patterning and specification of motor neurons and interneurons [29,30]

  • Intraflagellar transport mutants connect cilia transport and Sonic Hedgehog signaling Cilia and Shh were initially linked through forward genetics in the mouse, since mutants defective in intraflagellar transport (IFT) proteins showed improper neural tube patterning, and IFT is necessary for ciliogenesis and cilia maintenance [1,2]

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Summary

Introduction

Primary cilia are present on almost all vertebrate cells, and they have diverse functions in distinct tissues. Proper positioning of cilia in several developmental contexts is linked to the planar cell polarity (PCP) pathway, raising the possibility that the placement of cilia in the neural tube may be critical [16,17,18,19,20,21]. Intraflagellar transport mutants connect cilia transport and Sonic Hedgehog signaling Cilia and Shh were initially linked through forward genetics in the mouse, since mutants defective in intraflagellar transport (IFT) proteins showed improper neural tube patterning, and IFT is necessary for ciliogenesis and cilia maintenance [1,2].

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