Abstract

Primary cilia play a role in the sensation of and response to the surrounding environment. Caenorhabditis elegans (C. elegans) have primary cilia only on the distal tips of some dendrites. In order to better understand the relationship between receptor localization to cilia, cilia structure and cilia function, we have characterized a mutation originally identified in a forward genetic screen for mutants with defective PKD-2 ciliary localization. Through behavioral assays and examination of the structure of cilia in the cil-5 (my13) mutant animals, we have found that my13 disrupts not only receptor localization, but also some cilia-mediated sensory behaviors and cilia structural integrity. We have identified the my13 lesion and found that it is a missense mutation in bbs-7, an ortholog of human BBS-7, a gene known to affect human cilia and to be involved in Bardet-Biedl syndrome. Finally, we show that bbs-7(my13) also affects the glia cells which support the cilia.

Highlights

  • Primary cilia play a role in sensation of and response to the surrounding environment

  • C. elegans homozygous for bbs-7(my13) exhibit defects in male-specific ciliated neurons and some ciliated neurons common to both males and hermaphrodites

  • There is increased accumulation of PKD-2::GFP at the ciliary base in the mutant animals compared to wild-type

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Summary

Introduction

Primary cilia play a role in sensation of and response to the surrounding environment. They do so for many different cell types. The structure of primary cilia and the process by which they are formed are well-conserved across multiple cell types and organisms. December 8, 2014 bbs-7 (my13) Affects Cilia Structure and Function microtubule doublets. All proteins necessary for the form and function of the cilium must be transported into and within the cilium as there is no translation within the structure. A combination of molecular motors and cargo-associated proteins actively transport cargo within the cilium in a process known as intraflagellar transport (IFT) [4, 5]

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