Masaru Kaku1, Yoshihiro Komatsu2,3. 1. Division of Bioprosthodontics, Niigata University Graduate School of Medical and Dental Sciences, Niigata, 951-8514, Japan. kakum@dent.niigata-u.ac.jp. 2. Department of Pediatrics, The University of Texas Medical School at Houston, Houston, TX, 77030, USA. Yoshihiro.Komatsu@uth.tmc.edu. 3. Graduate Program in Genes and Development, The University of Texas Graduate School of Biomedical Sciences at Houston, Houston, TX, 77030, USA. Yoshihiro.Komatsu@uth.tmc.edu.
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. 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.
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. 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.
Entities:
Keywords:
Bone disease; Bone morphogenetic protein; Cilia; Growth factor signaling
Authors: Courtney J Haycraft; Qihong Zhang; Buer Song; Walker S Jackson; Peter J Detloff; Rosa Serra; Bradley K Yoder Journal: Development Date: 2006-12-13 Impact factor: 6.868
Authors: Amanda M D Malone; Charles T Anderson; Padmaja Tummala; Ronald Y Kwon; Tyler R Johnston; Tim Stearns; Christopher R Jacobs Journal: Proc Natl Acad Sci U S A Date: 2007-08-02 Impact factor: 11.205
Authors: David M Thomas; Sandra A Johnson; Natalie A Sims; Melanie K Trivett; John L Slavin; Brian P Rubin; Paul Waring; Grant A McArthur; Carl R Walkley; Andrew J Holloway; Dileepa Diyagama; Jonathon E Grim; Bruce E Clurman; David D L Bowtell; Jong-Seo Lee; Gabriel M Gutierrez; Denise M Piscopo; Shannon A Carty; Philip W Hinds Journal: J Cell Biol Date: 2004-12-06 Impact factor: 10.539
Authors: Krzysztof Jeziorny; Ewa Zmyslowska-Polakowska; Krystyna Wyka; Aleksandra Pyziak-Skupień; Maciej Borowiec; Agnieszka Szadkowska; Agnieszka Zmysłowska Journal: Bone Rep Date: 2022-07-01
Authors: Christian Louis Bonatto Paese; Evan C Brooks; Megan Aarnio-Peterson; Samantha A Brugmann Journal: Development Date: 2021-02-15 Impact factor: 6.868
Authors: Birgit Weiss; Birgit Eberle; Ralph Roeth; Christiaan de Bruin; Julian C Lui; Nagarajan Paramasivam; Katrin Hinderhofer; Hermine A van Duyvenvoorde; Jeffrey Baron; Jan M Wit; Gudrun A Rappold Journal: Front Endocrinol (Lausanne) Date: 2021-06-04 Impact factor: 5.555
Authors: Yangmengfan Chen; Romina H Aspera-Werz; Maximilian M Menger; Karsten Falldorf; Michael Ronniger; Christina Stacke; Tina Histing; Andreas K Nussler; Sabrina Ehnert Journal: Int J Mol Sci Date: 2021-06-29 Impact factor: 5.923
Authors: Mari S Lehti; Henna Henriksson; Petri Rummukainen; Fan Wang; Liina Uusitalo-Kylmälä; Riku Kiviranta; Terhi J Heino; Noora Kotaja; Anu Sironen Journal: Sci Rep Date: 2018-01-16 Impact factor: 4.379