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Molecular and Cellular Biology, May 2005, p. 4129-4137, Vol. 25, No. 10
0270-7306/05/$08.00+0     doi:10.1128/MCB.25.10.4129-4137.2005
Copyright © 2005, American Society for Microbiology. All Rights Reserved.

The Ciliary Rootlet Maintains Long-Term Stability of Sensory Cilia

Jun Yang,1 Jiangang Gao,2 Michael Adamian,1 Xiao-Hong Wen,3 Basil Pawlyk,1 Luo Zhang,4,{dagger} Michael J. Sanderson,4 Jian Zuo,2 Clint L. Makino,3 and Tiansen Li1*

The Berman-Gund Laboratory for the Study of Retinal Degenerations,1 Howe Laboratory, Department of Ophthalmology, Harvard Medical School, Massachusetts Eye and Ear Infirmary, Boston, Massachusetts 02114,3 Department of Developmental Neurobiology, St. Jude Children's Research Hospital, Memphis, Tennessee,2 Department of Physiology, University of Massachusetts Medical School, Worcester, Massachusetts4

Received 19 November 2004/ Returned for modification 2 December 2004/ Accepted 25 February 2005

The striated ciliary rootlet is a prominent cytoskeleton originating from basal bodies of ciliated cells. Although a familiar structure in cell biology, its function has remained unresolved. In this study, we carried out targeted disruption in mice of the gene for rootletin, a component of the rootlet. In the mutant, ciliated cells are devoid of rootlets. Phototransduction and ciliary beating in sensory and motile cilia initially exhibit no apparent functional deficits. However, photoreceptors degenerate over time, and mutant lungs appear prone to pathological changes consistent with insufficient mucociliary clearance. Further analyses revealed a striking fragility at the ciliary base in photoreceptors lacking rootlets. In vitro assays suggest that the rootlet is among the least dynamic of all cytoskeletons and interacts with actin filaments. Thus, a primary function of the rootlet is to provide structural support for the cilium. Inasmuch as photoreceptors elaborate an exceptionally enlarged sensory cilium, they are especially dependent on the rootlet for structural integrity and long-term survival.

* Corresponding author. Mailing address: Massachusetts Eye and Ear Infirmary, 243 Charles St., Boston, MA 02114. Phone: (617) 573-3904. Fax: (617) 753-3216. E-mail: tli{at}meei.harvard.edu.

{dagger} Present address: Department of Otolaryngology, Head and Neck Surgery, Capital University of Medical Sciences, Beijing Tong Ren Hospital, Beijing, People’s Republic of China.

Molecular and Cellular Biology, May 2005, p. 4129-4137, Vol. 25, No. 10
0022-538X/05/$08.00+0     doi:10.1128/MCB.25.10.4129-4137.2005
Copyright © 2005, American Society for Microbiology. All Rights Reserved.



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