Bbof1 is required to maintain cilia orientation
Citations Over TimeTop 10% of 2013 papers
Abstract
Multiciliate cells (MCCs) are highly specialized epithelial cells that employ hundreds of motile cilia to produce a vigorous directed flow in a variety of organ systems. The production of this flow requires the establishment of planar cell polarity (PCP) whereby MCCs align hundreds of beating cilia along a common planar axis. The planar axis of cilia in MCCs is known to be established via the PCP pathway and hydrodynamic cues, but the downstream steps required for cilia orientation remain poorly defined. Here, we describe a new component of cilia orientation, based on the phenotypic analysis of an uncharacterized coiled-coil protein, called bbof1. We show that the expression of bbof1 is induced during the early phases of MCC differentiation by the master regulator foxj1. MCC differentiation and ciliogenesis occurs normally in embryos where bbof1 activity is reduced, but cilia orientation is severely disrupted. We show that cilia in bbof1 mutants can still respond to patterning and hydrodynamic cues, but lack the ability to maintain their precise orientation. Misexpression of bbof1 promotes cilia alignment, even in the absence of flow or in embryos where microtubules and actin filaments are disrupted. Bbof1 appears to mediate cilia alignment by localizing to a polar structure adjacent to the basal body. Together, these results suggest that bbof1 is a basal body component required in MCCs to align and maintain cilia orientation in response to flow.
Related Papers
- → Stages of ciliogenesis and regulation of ciliary length(2011)231 cited
- → Transcriptional control of genes involved in ciliogenesis: a first step in making cilia(2010)166 cited
- → C2cd3 is critical for centriolar distal appendage assembly and ciliary vesicle docking in mammals(2014)151 cited
- → Centrioles initiate cilia assembly but are dispensable for maturation and maintenance in C. elegans(2017)66 cited
- → Chlamydomonas Basal Bodies as Flagella Organizing Centers(2018)34 cited