An essential role for zebrafish Fgfrl1 during gill cartilage development

Hall, C., Flores, M.V., Murison, G., Crosier, K., and Crosier, P.
Mechanisms of Development   123(12): 925-940 (Journal)
Registered Authors
Crosier, Kathy, Crosier, Phil, Flores, Maria, Hall, Chris
Fgfrl1a, Fgfrl1b, FGF signaling, Pharyngeal endoderm, Cartilage, Fgf3, Fgf8, Gcm2
MeSH Terms
  • Amino Acid Sequence
  • Animals
  • Branchial Region/chemistry
  • Branchial Region/embryology
  • Cartilage/chemistry
  • Cartilage/embryology*
  • Cell Movement/genetics
  • DNA-Binding Proteins/analysis
  • DNA-Binding Proteins/genetics
  • DNA-Binding Proteins/metabolism
  • Ectoderm/chemistry
  • Ectoderm/metabolism
  • Embryo, Nonmammalian/metabolism
  • Embryonic Development/genetics
  • Gills/chemistry
  • Gills/embryology*
  • HMGB Proteins/analysis
  • HMGB Proteins/genetics
  • HMGB Proteins/metabolism
  • Molecular Sequence Data
  • Neural Crest/cytology
  • Phylogeny
  • Receptors, Fibroblast Growth Factor/analysis
  • Receptors, Fibroblast Growth Factor/genetics
  • Receptors, Fibroblast Growth Factor/physiology*
  • SOX9 Transcription Factor
  • Transcription Factors/analysis
  • Transcription Factors/genetics
  • Transcription Factors/metabolism
  • Zebrafish/embryology*
  • Zebrafish/genetics
  • Zebrafish Proteins/analysis
  • Zebrafish Proteins/genetics
  • Zebrafish Proteins/metabolism
  • Zebrafish Proteins/physiology*
17011755 Full text @ Mech. Dev.
The vertebrate craniofacial skeleton develops via a complex process involving signaling cascades in all three germ layers. Fibroblast growth factor (FGF) signaling is essential for several steps in pharyngeal arch development. In zebrafish, Fgf3 and Fgf8 in the mesoderm and hindbrain have an early role to pattern the pouch endoderm, influencing craniofacial integrity. Endodermal FGF signaling is required for the differentiation and survival of postmigratory neural crest cells that form the pharyngeal skeleton. We identify a novel role for zebrafish Fgf receptor-like 1a (Fgfrl1a) that is indispensable during gill cartilage development. We show that depletion of Fgfrl1a is sufficient to abolish cartilage derivatives of the ceratobranchials. Using an Fgfrl1a-deficient model, we analyzed expression of genes critical for chondrogenesis in the different compartments of the developing pharyngeal arch. Fgfrl1a-depleted animals demonstrate typical neural crest specification and migration to populate the arch primordia as well as normal pouch segmentation. However, in the absence of Fgfrl1a, larvae fail to express the transcription factor glial cells missing 2 (gcm2), a gene necessary for cartilage and gill filament formation, in the ectodermal lining of the branchial arches. In addition, two transcription factors essential for chondrogenesis, sox9a and runx2b, fail to express within the mesenchymal condensations of the branchial arches. A duplicate zebrafish gene, fgfrl1b, has now been identified. We show that Fgfrl1b is also required for proper formation of all ventral cartilage elements and acts cooperatively with Fgfrl1a during gill cartilage formation.
Genes / Markers
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Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Engineered Foreign Genes