PUBLICATION

The Reissner Fiber in the Cerebrospinal Fluid Controls Morphogenesis of the Body Axis

Authors

Cantaut-Belarif, Y., Sternberg, J.R., Thouvenin, O., Wyart, C., Bardet, P.L.

ID

ZDB-PUB-180731-1

Date

2018

Source

Current biology : CB 28(15): 2479-2486.e4 (Journal)

Registered Authors

Bardet, Pierre-Luc, Wyart, Claire

Keywords

Reissner fiber, body axis morphogenesis, central canal, cerebrospinal fluid, cilia, development, extracellular protein, fluid dynamics, zebrafish

MeSH Terms

Cell Adhesion Molecules, Neuronal/genetics
Cell Adhesion Molecules, Neuronal/metabolism
Body Patterning/physiology*
Zebrafish/embryology*
Zebrafish/growth & development
Cilia/physiology*
Mutation
Animals
Cerebrospinal Fluid/metabolism*

(all 9)

PubMed

30057305 Full text @ Curr. Biol.

Abstract

Organ development depends on the integration of coordinated long-range communication between cells. The cerebrospinal fluid composition and flow properties regulate several aspects of central nervous system development, including progenitor proliferation, neurogenesis, and migration [1-3]. One understudied component of the cerebrospinal fluid, described over a century ago in vertebrates, is the Reissner fiber. This extracellular thread forming early in development results from the assembly of the SCO-spondin protein in the third and fourth brain ventricles and central canal of the spinal cord [4]. Up to now, the function of the Reissner fiber has remained elusive, partly due to the lack of genetic invalidation models [4]. Here, by mutating the scospondin gene, we demonstrate that the Reissner fiber is critical for the morphogenesis of a straight posterior body axis. In zebrafish mutants where the Reissner fiber is lost, ciliogenesis and cerebrospinal fluid flow are intact but body axis morphogenesis is impaired. Our results also explain the frequently observed phenotype that mutant embryos with defective cilia exhibit defects in body axis curvature. Here, we reveal that these mutants systematically fail to assemble the Reissner fiber. We show that cilia promote the formation of the Reissner fiber and that the fiber is necessary for proper body axis morphogenesis. Our study sets the stage for future investigations of the mechanisms linking the Reissner fiber to the control of body axis curvature during vertebrate development.

Genes / Markers

Figures

Show all Figures

Expression

Fish	Conditions	Stage	Anatomy	Assay	Figure
hsc5Tg	standard conditions	Prim-15	ependymal cell motile cilium	IFL	Fig. 2
hsc5Tg	standard conditions	Prim-15	floor plate spinal cord region motile cilium	IFL	Fig. 2
sspo^icm13/icm13; hsc5Tg	standard conditions	Prim-15	ependymal cell motile cilium	IFL	Fig. 2
sspo^icm13/icm13; hsc5Tg	standard conditions	Prim-15	floor plate spinal cord region motile cilium	IFL	Fig. 2

1 - 4 of 4

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Phenotype

Fish	Conditions	Stage	Phenotype	Figure
cfap298^tm304/tm304	standard conditions	Prim-5	Reissner's fiber Ab1-RF labeling spatial pattern, abnormal	Fig. 4
cfap298^tm304/tm304	standard conditions	Prim-5	Reissner's fiber malformed, abnormal	Fig. 4
cfap298^tm304/tm304	standard conditions	Long-pec	Reissner's fiber Ab1-RF labeling decreased amount, abnormal	Fig. 4
cfap298^tm304/tm304	standard conditions	Long-pec	Reissner's fiber Ab1-RF labeling spatial pattern, abnormal	Fig. 4
cfap298^tm304/tm304	standard conditions	Long-pec	Reissner's fiber malformed, abnormal	Fig. 4
dzip1^{ts294e/ts294e}	standard conditions	Long-pec	Reissner's fiber Ab1-RF labeling decreased amount, abnormal	Fig. 4
dzip1^{ts294e/ts294e}	standard conditions	Long-pec	Reissner's fiber Ab1-RF labeling spatial pattern, abnormal	Fig. 4
dzip1^{ts294e/ts294e}	standard conditions	Long-pec	Reissner's fiber malformed, abnormal	Fig. 4
ift88^tz288/tz288	standard conditions	Long-pec	Reissner's fiber Ab1-RF labeling spatial pattern, abnormal	Fig. 4
ift88^tz288/tz288	standard conditions	Long-pec	Reissner's fiber malformed, abnormal	Fig. 4

1 - 10 of 46

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Mutations / Transgenics

Allele	Construct	Type	Affected Genomic Region
hsc5Tg	Tg(actb2:Mmu.Arl13b-GFP)	Transgenic Insertion
icm13		Small Deletion	sspo
icm15		Insertion	sspo
tm304		Point Mutation	cfap298
tp49d		Point Mutation	traf3ip1
ts294e		Point Mutation	dzip1
tz288		Point Mutation	ift88

1 - 7 of 7

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Human Disease / Model

Sequence Targeting Reagents

Target	Reagent	Reagent Type
sspo	CRISPR1-sspo	CRISPR

Fish

Fish
cfap298^tm304/tm304
dzip1^{ts294e/ts294e}
hsc5Tg
ift88^tz288/tz288
sspo^icm13/icm13
sspo^icm13/icm13; hsc5Tg
sspo^icm15/icm15
traf3ip1^tp49d/tp49d
WT

Antibodies

Orthology

Engineered Foreign Genes

Marker	Marker Type	Name
GFP	EFG	GFP

Mapping

Cantaut-Belarif et al., 2018 ZDB-PUB-180731-1 PMID:30057305

The Reissner Fiber in the Cerebrospinal Fluid Controls Morphogenesis of the Body Axis

Cantaut-Belarif et al., 2018

ZDB-PUB-180731-1
PMID:30057305