PUBLICATION

The Reissner Fiber Is Highly Dynamic In Vivo and Controls Morphogenesis of the Spine

Authors

Troutwine, B.R., Gontarz, P., Konjikusic, M.J., Minowa, R., Monstad-Rios, A., Sepich, D.S., Kwon, R.Y., Solnica-Krezel, L., Gray, R.S.

ID

ZDB-PUB-200511-2

Date

2020

Source

Current biology : CB 30(12): 2353-2362.e3 (Journal)

Registered Authors

Gray, Ryan, Konjikusic, Mia, Kwon, Ronald, Minowa, Ryoko, Monstad-Rios, Adrian, Sepich, Diane, Solnica-Krezel, Lilianna, Troutwine, Benjamin

Keywords

Reissner fiber, SCO-spondin, scoliosis, zebrafish

MeSH Terms

Zebrafish/abnormalities*
Zebrafish/growth & development
Cell Adhesion Molecules, Neuronal/genetics*
Cell Adhesion Molecules, Neuronal/metabolism
Animals
Spine/abnormalities
Spine/growth & development*
Morphogenesis*

(all 8)

PubMed

32386529 Full text @ Curr. Biol.

Abstract

Cerebrospinal fluid (CSF) physiology is important for the development and homeostasis of the central nervous system, and its disruption has been linked to scoliosis in zebrafish [1, 2]. Suspended in the CSF is an extracellular structure called the Reissner fiber, which extends from the brain through the central canal of the spinal cord. Zebrafish scospondin-null mutants are unable to assemble a Reissner fiber and fail to form a straight body axis during embryonic development [3]. Here, we describe hypomorphic missense mutations of scospondin, which allow Reissner fiber assembly and extension of a straight axis. However, during larval development, these mutants display progressive Reissner fiber disassembly, which is concomitant with the emergence of axial curvatures and scoliosis in adult animals. Using a scospondin-GFP knockin zebrafish line, we demonstrate several dynamic properties of the Reissner fiber in vivo, including embryonic fiber assembly, the continuous rostral to caudal movement of the fiber within the brain and central canal, and subcommissural organ (SCO)-spondin-GFP protein secretion from the floor plate to merge with the fiber. Finally, we show that disassembly of the Reissner fiber is also associated with the progression of axial curvatures in distinct scoliosis mutant zebrafish models. Together, these data demonstrate a critical role for the Reissner fiber for the maintenance of a straight body axis and spine morphogenesis in adult zebrafish. Our study establishes a framework for future investigations to address the cellular effectors responsible for Reissner-fiber-dependent regulation of axial morphology. VIDEO ABSTRACT.

Genes / Markers

Figures

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Expression

Gene	Fish	Conditions	Stage	Anatomy	Assay	Figure
EGFP	sspo^ut24Tg	standard conditions	Protruding-mouth	flexural organ	IFL	Fig. 3
EGFP	sspo^ut24Tg	standard conditions	Protruding-mouth	floor plate	IFL	Fig. 3
EGFP	sspo^ut24Tg	standard conditions	Protruding-mouth	Reissner's fiber	IFL	Fig. 3
EGFP	sspo^ut24Tg	standard conditions	Protruding-mouth	subcommissural organ	IFL	Fig. 3

1 - 4 of 4

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Phenotype

Fish	Conditions	Stage	Phenotype	Figure
druk^dut26Et/+	standard conditions	Days 14-20	whole organism axis curved, abnormal	Fig. 4
druk^dut26Et/+; sspo^ut24Tg/+	standard conditions	Day 5	floor plate morphology, normal	Fig. 4
druk^dut26Et/+; sspo^ut24Tg/+	standard conditions	Day 5	Reissner's fiber morphology, normal	Fig. 4
druk^dut26Et/+; sspo^ut24Tg/+	standard conditions	Days 14-20	whole organism axis curved, abnormal	Fig. 4
kif6^gw326/+	standard conditions	Prim-15	Reissner's fiber morphology, normal	Fig. 4
kif6^gw326/+	standard conditions	Protruding-mouth	Reissner's fiber morphology, normal	Fig. 4
kif6^gw326/+	standard conditions	Day 5	Reissner's fiber morphology, normal	Fig. 4
kif6^gw326/gw326	standard conditions	Prim-15	Reissner's fiber morphology, normal	Fig. 4
kif6^gw326/gw326	standard conditions	Protruding-mouth	Reissner's fiber disassembled, abnormal	Fig. 4
kif6^gw326/gw326	standard conditions	Day 5	Reissner's fiber disassembled, abnormal	Fig. 4

1 - 10 of 40

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Mutations / Transgenics

Allele	Construct	Type	Affected Genomic Region
dut26Et	Et(Mmu.Fos:EGFP)	Transgenic Insertion	druk
gw326		Point Mutation	kif6
stl297		Point Mutation	sspo
stl300		Point Mutation	sspo
ut24Tg	Tg3(EGFP)	Transgenic Insertion	sspo

1 - 5 of 5

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Human Disease / Model

Human Disease	Fish	Conditions	Evidence
idiopathic scoliosis	sspo^{stl297/stl297}	standard conditions	TAS
idiopathic scoliosis	sspo^ut24Tg/+	standard conditions	TAS
idiopathic scoliosis	sspo^{stl300/stl300}	standard conditions	TAS

1 - 3 of 3

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Sequence Targeting Reagents

Target	Reagent	Reagent Type
sspo	CRISPR2-sspo	CRISPR

Fish

Fish
druk^dut26Et/+
druk^dut26Et/+; sspo^ut24Tg/+
kif6^gw326/gw326
kif6^gw326/+
sspo^{stl297/stl297}
sspo^{stl297/stl297}
sspo^{stl297/stl297}
sspo^stl297/+
sspo^stl297/+; sspo^stl300/+
sspo^{stl300/stl300}

1 - 10 of 13

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Antibodies

Orthology

Engineered Foreign Genes

Marker	Marker Type	Name
EGFP	EFG	EGFP

Mapping

Troutwine et al., 2020 ZDB-PUB-200511-2 PMID:32386529

The Reissner Fiber Is Highly Dynamic In Vivo and Controls Morphogenesis of the Spine

Troutwine et al., 2020

ZDB-PUB-200511-2
PMID:32386529