PUBLICATION

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

Authors
Bearce, E.A., Grimes, D.T.
ID
ZDB-PUB-200724-2
Date
2020
Source
Seminars in cell & developmental biology   110: 104-112 (Review)
Registered Authors
Grimes, Daniel T.
Keywords
Cerebrospinal fluid flow, Morphology, Motile cilia, Reissner fiber, Scoliosis, Zebrafish
MeSH Terms
  • Animals
  • Axoneme/metabolism
  • Axoneme/ultrastructure
  • Cell Adhesion Molecules, Neuronal/deficiency
  • Cell Adhesion Molecules, Neuronal/genetics*
  • Cerebrospinal Fluid/chemistry
  • Cilia/metabolism*
  • Cilia/pathology
  • Cilia/ultrastructure
  • Cytoskeletal Proteins/deficiency
  • Cytoskeletal Proteins/genetics*
  • Disease Models, Animal
  • Embryo, Nonmammalian
  • Gene Expression Regulation, Developmental
  • Humans
  • Morphogenesis/genetics*
  • Mutation
  • Scoliosis/genetics*
  • Scoliosis/metabolism
  • Scoliosis/pathology
  • Signal Transduction
  • Spine/abnormalities
  • Spine/growth & development
  • Spine/metabolism*
  • Urotensins/genetics
  • Zebrafish
  • Zebrafish Proteins/deficiency
  • Zebrafish Proteins/genetics*
PubMed
32693941 Full text @ Sem. Cell Dev. Biol.
Abstract
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.
Genes / Markers
Figures
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Antibodies
Orthology
Engineered Foreign Genes
Mapping