PUBLICATION

Zebrafish crocc2 mutants exhibit divergent craniofacial shape, misregulated variability, and aberrant cartilage morphogenesis

Authors
Packard, M., Gilbert, M.C., Tetrault, E., Albertson, R.C.
ID
ZDB-PUB-230410-48
Date
2023
Source
Developmental Dynamics : an official publication of the American Association of Anatomists   252(7): 1026-1045 (Journal)
Registered Authors
Keywords
none
MeSH Terms
  • Morphogenesis/genetics
  • Humans
  • Zebrafish*/genetics
  • Animals
  • Chondrocytes
  • Cartilage
  • Zebrafish Proteins*/genetics
(all 7)
PubMed
37032317 Full text @ Dev. Dyn.
Abstract
Background Phenotypic variation is of paramount importance in development, evolution, and human health; however, the molecular mechanisms that influence organ shape and shape variability are not well understood. During craniofacial development, the behavior of skeletal precursors is regulated by both biochemical and environmental inputs, and the primary cilia play critical roles in transducing both types of signals. Here we examine a gene that encodes a key constituent of the ciliary rootlets, crocc2, and its role in cartilage morphogenesis in larval zebrafish.
Results Geometric morphometric analysis of crocc2 mutants revealed altered craniofacial shapes and expanded variation. At the cellular level, we observed altered chondrocyte shapes and planar cell polarity across multiple stages in crocc2 mutants. Notably, cellular defects were specific to areas that experience direct mechanical input. Cartilage cell number, apoptosis, and bone patterning were not affected in crocc2 mutants.
Conclusions Whereas "regulatory" genes are widely implicated in patterning the craniofacial skeleton, genes that encode "structural" aspects of the cell are increasingly implicated in shaping the face. Our results add crocc2 to this list, and demonstrate that it affects craniofacial geometry and canalizes phenotypic variation. We propose that it does so via mechanosensing, possibly through the ciliary rootlet. If true, this would implicate a new organelle in skeletal development and evolution. This article is protected by copyright. All rights reserved.
Genes / Markers
Figures
Figure Gallery (8 images)
Show all Figures
Expression
Phenotype
No data available
Mutations / Transgenics
Allele Construct Type Affected Genomic Region
sa20707
    		Point Mutation
    sa20708
      		Point Mutation
      1 - 2 of 2
      Show
      Human Disease / Model
      Sequence Targeting Reagents
      Fish
      Antibodies
      Orthology
      Engineered Foreign Genes
      No data available
      Mapping
      Your Input Welcome
      We welcome your input and comments. Please use this form to recommend updates to the information in ZFIN. We appreciate as much detail as possible and references as appropriate. We will review your comments promptly.
      Citation
      Packard, M., Gilbert, M.C., Tetrault, E., Albertson, R.C. (2023) Zebrafish crocc2 mutants exhibit divergent craniofacial shape, misregulated variability, and aberrant cartilage morphogenesis. Developmental Dynamics : an official publication of the American Association of Anatomists. 252(7):1026-1045.