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Functional Diversity of Ciliary Proteins in Bone Development and Disease

  • Skeletal Development (P Trainor and K Svoboda, Section Editors)
  • Published:
Current Osteoporosis Reports Aims and scope Submit manuscript

Abstract

Purpose of Review

The primary cilium is a non-motile microtubule-based organelle that senses a diverse range of extracellular signals. While recent studies highlight the importance of ciliary-dependent developmental signals, including Hedgehog, Wnt, and platelet-derived growth factor, it is not well understood whether and how bone morphogenetic protein (BMP) signaling, a key regulator of skeletogenesis, is involved in cilia-related bone developmental aspects and in the etiology of skeletal disorders.

Recent Findings

Increasing evidence suggests that osteoblast- or osteocyte-specific deletion of ciliary proteins leads to diverse skeletal malformations, reinforcing the idea that primary cilia are indispensable for regulating bone development and maintenance. Furthermore, it became evident that ciliary proteins not only contribute to ciliogenesis but also orchestrate cellular trafficking.

Summary

This review summarizes the current understanding of ciliary proteins in bone development and discusses the potential role of BMP signaling in primary cilia, enabling us to unravel the potential pathogenesis of skeletal ciliopathies.

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Acknowledgements

We gratefully acknowledge Patricia Fonseca for editorial assistance. This study was supported by a Grant-in-Aid for Scientific Research from the Japan Society for the Promotion of Science (26293407, 15KK0337, and 15K15704 to MK), and by a grant from the NIDCR/NIH (R00DE021054 to YK).

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Authors and Affiliations

  1. Division of Bioprosthodontics, Niigata University Graduate School of Medical and Dental Sciences, Niigata, 951-8514, Japan

    Masaru Kaku

  2. Department of Pediatrics, The University of Texas Medical School at Houston, Houston, TX, 77030, USA

    Yoshihiro Komatsu

  3. Graduate Program in Genes and Development, The University of Texas Graduate School of Biomedical Sciences at Houston, Houston, TX, 77030, USA

    Yoshihiro Komatsu

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  1. Masaru Kaku
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Correspondence to Masaru Kaku or Yoshihiro Komatsu.

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Masaru Kaku and Yoshihiro Komatsu declare no conflict of interest.

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This article is part of the Topical Collection on Skeletal Development

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Kaku, M., Komatsu, Y. Functional Diversity of Ciliary Proteins in Bone Development and Disease. Curr Osteoporos Rep 15, 96–102 (2017). https://doi.org/10.1007/s11914-017-0351-6

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