Aquatic models of human ciliary diseases

Genesis. 2021 Feb;59(1-2):e23410. doi: 10.1002/dvg.23410. Epub 2021 Jan 26.

Abstract

Cilia are microtubule-based structures that either transmit information into the cell or move fluid outside of the cell. There are many human diseases that arise from malfunctioning cilia. Although mammalian models provide vital insights into the underlying pathology of these diseases, aquatic organisms such as Xenopus and zebrafish provide valuable tools to help screen and dissect out the underlying causes of these diseases. In this review we focus on recent studies that identify or describe different types of human ciliopathies and outline how aquatic organisms have aided our understanding of these diseases.

Keywords: Xenopus; cilia; ciliopathy; cystic kidney; kidney; nasal; node; zebrafish.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't
  • Review

MeSH terms

  • Animals
  • Ciliopathies / genetics*
  • Ciliopathies / metabolism
  • Ciliopathies / pathology
  • Disease Models, Animal*
  • Phenotype
  • Xenopus laevis / genetics*
  • Xenopus laevis / metabolism
  • Zebrafish / genetics*
  • Zebrafish / metabolism