Drosophila chibby is required for basal body formation and ciliogenesis but not for Wg signaling

Abstract

In contrast to vertebrate CBY, which functions in WNT signaling, Drosophila CBY is essential for normal basal body structure and function but dispensable for Wg signaling.

BAR Domain-Containing FAM92 Proteins Interact with Chibby1 To Facilitate Ciliogenesis

“…Cby1 knockout (Cby1-KO) mice display several hallmarks of ciliary defects, including chronic upper airway infection (12), subfertility, and polycystic kidneys (15) as well as polydactyly and hydrocephalus at low frequencies. In Drosophila melanogaster, Cby1 is expressed in sensory neurons and male germ cells, the only ciliated cell types in this organism, and is required for the proper formation of neuronal cilia and sperm flagella, respectively (16). Similarly, in Xenopus laevis, Cby1 is indispensable for ciliogenesis in multiciliated cells of the embryonic epidermis (17).…”

“…Chibby1 (Cby1) is a conserved 15-kDa coiled-coil protein that predominantly localizes to mother centrioles/basal bodies and plays a pivotal role in the formation and function of cilia (6,(12)(13)(14)(15)(16)(17)(18)(19). Cby1 knockout (Cby1-KO) mice display several hallmarks of ciliary defects, including chronic upper airway infection (12), subfertility, and polycystic kidneys (15) as well as polydactyly and hydrocephalus at low frequencies.…”

Does Unc–GFP uncover ciliary structures in the rhabdomeric eye of Drosophila?

“…We therefore assessed the localization of Chibby, a protein selectively expressed in the transition zone of the Drosophila ciliary structures (Enjolras et al, 2012). Although distinct Unc dots were evident, we did not find Chibby signals (data not shown).…”

“…We therefore asked whether the Drosophila ommatidia contained ciliary structures by looking at the distribution of an Uncoordinated (Unc) GFP fusion protein, a good marker for centriole-to-basal body conversion in Drosophila Enjolras et al, 2012). The Unc protein has, until now, been found in the cell types in which the centrioles nucleate a ciliary axoneme, namely sensory neurons and male germ cells, but it is absent in other Drosophila tissues that lack ciliary structures.…”

Contrôle transcriptionnel des gènes ciliaires

“…[31], est nécessaire à la motilité des cils via le contrôle de l'assemblage des bras de dynéine chez le poisson zèbre [43]. Il en est de même pour le gène chibby, également identifié comme cible de RFX, et qui joue un rôle crucial dans la biogenèse de la zone de transition chez la drosophile et pour la mise en place des cils mobiles chez la souris [44,45]. De la même manière, une étude visant à trouver les cibles de RFX2 dans les cellules multiciliées de la peau de xénope a conduit à l'identification de deux nouveaux gènes ciliaires.…”