PUBLICATION

Diversity and function of motile ciliated cell types within ependymal lineages of the zebrafish brain

Authors
D'Gama, P.P., Qiu, T., Cosacak, M.I., Rayamajhi, D., Konac, A., Hansen, J.N., Ringers, C., Acuña-Hinrichsen, F., Hui, S.P., Olstad, E.W., Chong, Y.L., Lim, C.K.A., Gupta, A., Ng, C.P., Nilges, B.S., Kashikar, N.D., Wachten, D., Liebl, D., Kikuchi, K., Kizil, C., Yaksi, E., Roy, S., Jurisch-Yaksi, N.
ID
ZDB-PUB-211006-9
Date
2021
Source
Cell Reports   37: 109775 (Journal)
Registered Authors
Cosacak, Mehmet Ilyas, Jurisch-Yaksi, Nathalie, Kikuchi, Kazu, Kizil, Caghan, Ringers, Christa, Roy, Sudipto
Keywords
brain, cerebrospinal fluid, choroid plexus, cilia, ependymal cell, foxj1, gmnc, multiciliated cells, scoliosis, zebrafish
Datasets
GEO:GSE161834
MeSH Terms
  • Animals
  • Animals, Genetically Modified/metabolism
  • Brain/cytology
  • Brain/metabolism*
  • Brain/pathology
  • Cell Lineage
  • Cerebrospinal Fluid/physiology
  • Cilia/metabolism*
  • Cilia/pathology
  • Embryo, Nonmammalian/metabolism
  • Ependyma/cytology
  • Ependyma/metabolism*
  • Ependyma/pathology
  • Forkhead Transcription Factors/genetics
  • Forkhead Transcription Factors/metabolism
  • Gene Editing
  • Morphogenesis
  • Nuclear Proteins/genetics
  • Nuclear Proteins/metabolism
  • Spine/growth & development
  • Spine/metabolism
  • Telencephalon/cytology
  • Telencephalon/metabolism
  • Telencephalon/pathology
  • Tubulin/metabolism
  • Zebrafish
  • Zebrafish Proteins/genetics
  • Zebrafish Proteins/metabolism
PubMed
34610312 Full text @ Cell Rep.
Abstract
Motile cilia defects impair cerebrospinal fluid (CSF) flow and can cause brain and spine disorders. The development of ciliated cells, their impact on CSF flow, and their function in brain and axial morphogenesis are not fully understood. We have characterized motile ciliated cells within the zebrafish brain ventricles. We show that the ventricles undergo restructuring through development, involving a transition from mono- to multiciliated cells (MCCs) driven by gmnc. MCCs co-exist with monociliated cells and generate directional flow patterns. These ciliated cells have different developmental origins and are genetically heterogenous with respect to expression of the Foxj1 family of ciliary master regulators. Finally, we show that cilia loss from the tela choroida and choroid plexus or global perturbation of multiciliation does not affect overall brain or spine morphogenesis but results in enlarged ventricles. Our findings establish that motile ciliated cells are generated by complementary and sequential transcriptional programs to support ventricular development.
Genes / Markers
Figures
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Expression
Phenotype
Mutation and Transgenics
Human Disease / Model Data
Sequence Targeting Reagents
Fish
Antibodies
Orthology
Engineered Foreign Genes
Mapping
Errata and Notes