PUBLICATION

Urotensin II-related peptides, Urp1 and Urp2, control zebrafish spine morphology

Authors
Bearce, E.A., Irons, Z.H., O'Hara-Smith, J.R., Kuhns, C.J., Fisher, S.I., Crow, W.E., Grimes, D.T.
ID
ZDB-PUB-221202-6
Date
2022
Source
eLIFE   11: (Journal)
Registered Authors
Grimes, Daniel T.
Keywords
developmental biology, genetics, genomics, zebrafish
MeSH Terms
  • Urotensins*/genetics
  • Zebrafish/genetics
  • Scoliosis*
  • Animals
  • Spine
PubMed
36453722 Full text @ Elife
Abstract
The spine provides structure and support to the body, yet how it develops its characteristic morphology as the organism grows is little understood. This is underscored by the commonality of conditions in which the spine curves abnormally such as scoliosis, kyphosis and lordosis. Understanding the origin of such spinal curves has been challenging in part due to the lack of appropriate animal models. Recently, zebrafish have emerged as promising tools with which to understand the origin of spinal curves. Using zebrafish, we demonstrate that the Urotensin II-related peptides (URPs), Urp1 and Urp2, are essential for maintaining spine morphology. Urp1 and Urp2 are 10-amino acid cyclic peptides expressed by neurons lining the central canal of the spinal cord. Upon combined genetic loss of Urp1 and Urp2, adolescent-onset planar curves manifested in the caudal region of the spine. Highly similar curves were caused by mutation of Uts2r3, an URP receptor. Quantitative comparisons revealed that Urotensin-associated curves were distinct from other zebrafish spinal curve mutants in curve position and direction. Last, we found that the Reissner fiber, a proteinaceous thread that sits in the central canal and has been implicated in the control of spine morphology, breaks down prior to curve formation mutants with perturbed cilia motility but was unaffected by loss of Uts2r3. This suggests a Reissner fiber-independent mechanism of curvature in Urotensin-deficient mutants. Overall, our results show that Urp1 and Urp2 control zebrafish spine morphology and establish new animal models of spine deformity.
Genes / Markers
Figures
Figure Gallery (5 images)
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Expression
Phenotype
Mutations / Transgenics
Allele Construct Type Affected Genomic Region
b1420
    Indel
    b1421
      Small Deletion
      b1436
        Small Deletion
        b1446
          Indel
          icm02
            Small Deletion
            tm304
              Point Mutation
              ut24TgTransgenic Insertion
              1 - 7 of 7
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              Human Disease / Model
              1 - 1 of 1
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              Sequence Targeting Reagents
              1 - 7 of 7
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              Fish
              Antibodies
              No data available
              Orthology
              No data available
              Engineered Foreign Genes
              Marker Marker Type Name
              EGFPEFGEGFP
              1 - 1 of 1
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              Mapping
              No data available