Bbof1 is required to maintain cilia orientation

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...

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Published in:	Development (Cambridge) Vol. 140; no. 16; pp. 3468 - 3477
Main Authors:	Chien, Yuan-Hung, Werner, Michael E, Stubbs, Jennifer, Joens, Matt S, Li, Julie, Chien, Shu, Fitzpatrick, James A J, Mitchell, Brian J, Kintner, Chris
Format:	Journal Article
Language:	English
Published:	England Company of Biologists 15-08-2013
Subjects:	Actins - metabolism Animals Axoneme - metabolism Body Patterning Cell Differentiation Cilia - metabolism Cilia - physiology Embryo, Nonmammalian - drug effects Embryo, Nonmammalian - metabolism Embryo, Nonmammalian - physiology Forkhead Transcription Factors - genetics Forkhead Transcription Factors - metabolism Gene Expression Regulation, Developmental Hydrodynamics Movement Nocodazole - pharmacology Recombinant Fusion Proteins - genetics Recombinant Fusion Proteins - metabolism Xenopus laevis - metabolism Xenopus laevis - physiology Xenopus Proteins - genetics Xenopus Proteins - metabolism Xenopus Planar cell polarity Cilia
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Summary:	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.
Bibliography:	ObjectType-Article-1 SourceType-Scholarly Journals-1 ObjectType-Feature-2 content type line 23 Present address: Pathway Genomics, 4045 Sorrento Valley Boulevard, San Diego, CA 92121, USA
ISSN:	0950-1991 1477-9129
DOI:	10.1242/dev.096727