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In vertebrates, most cells have an immotile primary cilium that mediates cell signaling, and some specialized cells assemble one or multiple cilia that are motile and beat synchronously to move fluids in one direction. Gene mutations that alter cilia structure or function cause a broad spectrum of disorders termed ciliopathies that impact virtually every system in the body. A wide range of birth defects associated with ciliopathies underscores critical functions for cilia during embryonic development. In many cases, the mechanisms underlying cilia functions during development and disease remain poorly understood. This review describes different types of cilia in vertebrate embryos and discusses recent research results from diverse model systems that provide novel insights into how cilia form and function during embryo development. The work discussed here not only expands our understanding of in vivo cilia biology, but also opens new questions about cilia and their roles in establishing healthy embryos.<\/jats:p>","DOI":"10.1002\/mrd.23650","type":"journal-article","created":{"date-parts":[[2022,11,11]],"date-time":"2022-11-11T18:29:15Z","timestamp":1668191355000},"page":"579-596","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":35,"title":["Structures and functions of cilia during vertebrate embryo development"],"prefix":"10.1002","volume":"89","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-5465-9754","authenticated-orcid":false,"given":"Jeffrey D.","family":"Amack","sequence":"first","affiliation":[{"name":"Department of Cell and Developmental Biology State University of New York Upstate Medical University Syracuse New York USA"},{"name":"BioInspired Syracuse: Institute for Material and Living Systems Syracuse New York 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