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
  • Cell Adhesion Molecules, Neuronal/genetics*
  • Cell Adhesion Molecules, Neuronal/metabolism
  • Animals
  • Zebrafish/abnormalities*
  • Zebrafish/growth & development
  • Morphogenesis*
  • Spine/abnormalities
  • Spine/growth & development*
(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
Figure Gallery (4 images)
Show all Figures
Expression
Gene Antibody Fish Conditions Stage Qualifier Anatomy Assay Figure
EGFPsspout24Tgstandard conditionsProtruding-mouthIFL
EGFPsspout24Tgstandard conditionsProtruding-mouthIFL
EGFPsspout24Tgstandard conditionsProtruding-mouthIFL
EGFPsspout24Tgstandard conditionsProtruding-mouthIFL
1 - 4 of 4
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Phenotype
Fish Conditions Stage Phenotype Figure
drukdut26Et/+standard conditionsDays 14-20
drukdut26Et/+; sspout24Tg/+standard conditionsDay 5
drukdut26Et/+; sspout24Tg/+standard conditionsDay 5
drukdut26Et/+; sspout24Tg/+standard conditionsDays 14-20
kif6gw326/+standard conditionsPrim-15
kif6gw326/+standard conditionsProtruding-mouth
kif6gw326/+standard conditionsDay 5
kif6gw326/gw326standard conditionsPrim-15
kif6gw326/gw326standard conditionsProtruding-mouth
kif6gw326/gw326standard conditionsDay 5
1 - 10 of 40
Show
Mutations / Transgenics
Allele Construct Type Affected Genomic Region
dut26EtTransgenic Insertion
gw326
    		Point Mutation
    stl297
      		Point Mutation
      stl300
        		Point Mutation
        ut24TgTransgenic Insertion
        1 - 5 of 5
        Show
        Human Disease / Model
        Human Disease Fish Conditions Evidence
        idiopathic scoliosissspostl297/stl297standard conditionsTAS
        idiopathic scoliosissspout24Tg/+standard conditionsTAS
        idiopathic scoliosissspostl300/stl300standard conditionsTAS
        1 - 3 of 3
        Show
        Sequence Targeting Reagents
        Target Reagent Reagent Type
        sspoCRISPR2-sspoCRISPR
        1 - 1 of 1
        Show
        Fish
        Fish
        drukdut26Et/+
        drukdut26Et/+; sspout24Tg/+
        kif6gw326/gw326
        kif6gw326/+
        sspostl297/stl297
        sspostl297/stl297
        sspostl297/stl297
        sspostl297/+
        sspostl297/+; sspostl300/+
        sspostl300/stl300
        1 - 10 of 13
        Show
        Antibodies
        Name Type Antigen Genes Isotypes Host Organism
        Ab1-tjp1monoclonalIgG1Mouse
        1 - 1 of 1
        Show
        Orthology
        No data available
        Engineered Foreign Genes
        Marker Marker Type Name
        EGFPEFGEGFP
        1 - 1 of 1
        Show
        Mapping
        No data available
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        Citation
        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. (2020) The Reissner Fiber Is Highly Dynamic In Vivo and Controls Morphogenesis of the Spine. Current biology : CB. 30(12):2353-2362.e3.