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      Various facets of vertebrate cilia: motility, signaling, and role in adult neurogenesis

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          Abstract

          Cilia are microtubule-based cellular organelles that are widely distributed in vertebrate tissues. They were first observed hundreds of years ago. Recent studies indicate that this small organelle plays important roles in numerous physiological phenomena, including tissue morphogenesis, signal transduction, determination of left-right asymmetry during development, and adult neurogenesis. Ciliopathies, syndromes resulting from a genetic disorder of cilial components, frequently have complex effects involving many organ systems, owing to the broad distribution of cilia in the body.

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          Randomization of left-right asymmetry due to loss of nodal cilia generating leftward flow of extraembryonic fluid in mice lacking KIF3B motor protein.

          Shigenori Nonaka, Yosuke Tanaka, Yasushi Okada … (1998)
          Microtubule-dependent motor, murine KIF3B, was disrupted by gene targeting. The null mutants did not survive beyond midgestation, exhibiting growth retardation, pericardial sac ballooning, and neural tube disorganization. Prominently, the left-right asymmetry was randomized in the heart loop and the direction of embryonic turning. lefty-2 expression was either bilateral or absent. Furthermore, the node lacked monocilia while the basal bodies were present. Immunocytochemistry revealed KIF3B localization in wild-type nodal cilia. Video microscopy showed that these cilia were motile and generated a leftward flow. These data suggest that KIF3B is essential for the left-right determination through intraciliary transportation of materials for ciliogenesis of motile primary cilia that could produce a gradient of putative morphogen along the left-right axis in the node.
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            The ciliopathies: an emerging class of human genetic disorders.

            Jose L Badano, Norimasa Mitsuma, Phil Beales … (2006)
            Cilia and flagella are ancient, evolutionarily conserved organelles that project from cell surfaces to perform diverse biological roles, including whole-cell locomotion; movement of fluid; chemo-, mechano-, and photosensation; and sexual reproduction. Consistent with their stringent evolutionary conservation, defects in cilia are associated with a range of human diseases, such as primary ciliary dyskinesia, hydrocephalus, polycystic liver and kidney disease, and some forms of retinal degeneration. Recent evidence indicates that ciliary defects can lead to a broader set of developmental and adult phenotypes, with mutations in ciliary proteins now associated with nephronophthisis, Bardet-Biedl syndrome, Alstrom syndrome, and Meckel-Gruber syndrome. The molecular data linking seemingly unrelated clinical entities are beginning to highlight a common theme, where defects in ciliary structure and function can lead to a predictable phenotypic pattern that has potentially predictive and therapeutic value.
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              The vertebrate primary cilium in development, homeostasis, and disease.

              Jantje M Gerdes, Erica E. Davis, Nicholas Katsanis (2009)
              Cilia are complex structures that have garnered interest because of their roles in vertebrate development and their involvement in human genetic disorders. In contrast to multicellular invertebrates in which cilia are restricted to specific cell types, these organelles are found almost ubiquitously in vertebrate cells, where they serve a diverse set of signaling functions. Here, we highlight properties of vertebrate cilia, with particular emphasis on their relationship with other subcellular structures, and explore the physiological consequences of ciliary dysfunction.
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                Author and article information

                Journal
                Journal ID (nlm-ta): Proc Jpn Acad Ser B Phys Biol Sci
                Journal ID (iso-abbrev): Proc. Jpn. Acad., Ser. B, Phys. Biol. Sci
                Journal ID (publisher-id): PJAB
                Title: Proceedings of the Japan Academy. Series B, Physical and Biological Sciences
                Publisher: The Japan Academy
                ISSN (Print): 0386-2208
                ISSN (Electronic): 1349-2896
                Publication date (Print): October 2009
                Volume: 85
                Issue: 8
                Pages: 324-336
                Affiliations
                [*1 ] Department of Developmental and Regenerative Biology, Institute of Molecular Medicine, Nagoya City University Graduate School of Medical Sciences, Aichi, Japan.
                Author notes

                (Communicated by Nobutaka H irokawa, m.j.a.)

                [† ] Correspondence should be addressed: K. Sawamoto, Department of Developmental and Regenerative Biology, Institute of Molecular Medicine, Nagoya City University Graduate School of Medical Sciences, 1 Kawasumi, Mizuho-cho, Mizuho-ku, Nagoya, Aichi 467-8601, Japan (e-mail: sawamoto@ 123456med.nagoya-cu.ac.jp ).
                Article
                Publisher ID: pjab-85-324
                DOI: 10.2183/pjab.85.324
                PMC ID: 3621569
                PubMed ID: 19838012
                SO-VID: 84c3b2cc-b782-49a5-84d1-7da3b6329594
                Copyright © © 2009 The Japan Academy
                License:

                This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                Date received : 18 May 2009
                Date accepted : 10 July 2009
                Categories
                Subject: Review

                ScienceOpen disciplines: Life sciences
                Keywords: cilia,morphogenesis,ciliopathy
                Data availability:
                ScienceOpen disciplines: Life sciences
                Keywords: cilia, morphogenesis, ciliopathy

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