4.6 Review

On being the right shape: Roles for motile cilia and cerebrospinal fluid flow in body and spine morphology

Journal

SEMINARS IN CELL & DEVELOPMENTAL BIOLOGY
Volume 110, Issue -, Pages 104-112

Publisher

ACADEMIC PRESS LTD- ELSEVIER SCIENCE LTD
DOI: 10.1016/j.semcdb.2020.07.005

Keywords

Motile cilia; Cerebrospinal fluid flow; Zebrafish; Scoliosis; Morphology; Reissner fiber

Funding

  1. NIH [R00AR70905]
  2. Donald E. and Delia B. Baxter Foundation
  3. Oregon Health and Science University Medical Research Foundation
  4. University of Oregon

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This review explores the central problem of how developing organisms attain their proper shape, focusing on the formation of the body axis and spine in vertebrates. Recent research in zebrafish has highlighted the role of motile cilia and cerebrospinal fluid flow in axial morphogenesis and spinal straightness. By investigating zebrafish mutants, researchers are gaining insight into human conditions like Idiopathic Scoliosis, shedding light on how body and spine shape are acquired and maintained through development and growth.
How developing and growing organisms attain their proper shape is a central problem of developmental biology. In this review, we investigate this question with respect to how the body axis and spine form in their characteristic linear head-to-tail fashion in vertebrates. Recent work in the zebrafish has implicated motile cilia and cerebrospinal fluid flow in axial morphogenesis and spinal straightness. We begin by introducing motile cilia, the fluid flows they generate and their roles in zebrafish development and growth. We then describe how cilia control body and spine shape through sensory cells in the spinal canal, a thread-like extracellular structure called the Reissner fiber, and expression of neuropeptide signals. Last, we discuss zebrafish mutants in which spinal straightness breaks down and three-dimensional curves form. These curves resemble the common but little understood human disease Idiopathic Scoliosis. Zebrafish research is therefore poised to make progress in our understanding of this condition and, more generally, how body and spine shape is acquired and maintained through development and growth.

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