Analysis of Sphingosine-1-phosphate signaling mutants reveals endodermal requirements for the growth but not dorsoventral patterning of jaw skeletal precursors

Balczerski, B., Matsutani, M., Castillo, P., Osborne, N., Stainier, D.Y., and Crump, J.G.
Developmental Biology   361(2): 230-241 (Journal)
Registered Authors
Balczerski, Bartosz, Crump, Gage DeKoeyer, Matsutani, Megan, Osborne, Nick, Stainier, Didier
Sphingosine-1-phosphate, pharyngeal endoderm, facial ectoderm, craniofacial skeleton, Shh, zebrafish
MeSH Terms
  • Analysis of Variance
  • Animals
  • Body Patterning/genetics
  • Body Patterning/physiology
  • Carrier Proteins/genetics
  • Carrier Proteins/metabolism
  • Craniofacial Abnormalities/genetics*
  • DNA Primers/genetics
  • DNA, Complementary/genetics
  • Endoderm/metabolism*
  • Fibroblast Growth Factors/metabolism
  • Genotype
  • Hedgehog Proteins/metabolism
  • Image Interpretation, Computer-Assisted
  • In Situ Hybridization
  • Jaw/embryology*
  • Lysophospholipids/metabolism*
  • Membrane Proteins/genetics
  • Membrane Proteins/metabolism
  • Microscopy, Fluorescence
  • Mutation/genetics
  • Receptors, Lysosphingolipid/genetics*
  • Receptors, Lysosphingolipid/metabolism
  • Signal Transduction/genetics*
  • Sphingosine/analogs & derivatives*
  • Sphingosine/metabolism
  • Zebrafish/embryology*
  • Zebrafish Proteins/genetics
  • Zebrafish Proteins/metabolism
22185793 Full text @ Dev. Biol.

Development of the head skeleton involves reciprocal interactions between cranial neural crest cells (CNCCs) and the surrounding pharyngeal endoderm and ectoderm. Whereas elegant experiments in avians have shown a prominent role for the endoderm in facial skeleton development, the relative functions of the endoderm in growth versus regional identity of skeletal precursors have remained unclear. Here we describe novel craniofacial defects in zebrafish harboring mutations in the Sphingosine-1-phospate (S1P) type 2 receptor (s1pr2) or the S1P transporter Spinster 2 (spns2), and we show that S1P signaling functions in the endoderm for the proper growth and positioning of the jaw skeleton. Surprisingly, analysis of s1pr2 and spns2 mutants, as well as sox32 mutants that completely lack endoderm, reveals that the dorsal–ventral (DV) patterning of jaw skeletal precursors is largely unaffected even in the absence of endoderm. Instead, we observe reductions in the ectodermal expression of Fibroblast growth factor 8a (Fgf8a), and transgenic misexpression of Shha restores fgf8a expression and partially rescues the growth and differentiation of jaw skeletal precursors. Hence, we propose that the S1P-dependent anterior foregut endoderm functions primarily through Shh to regulate the growth but not DV patterning of zebrafish jaw precursors.

Genes / Markers
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Mutation and Transgenics
Human Disease / Model Data
Sequence Targeting Reagents
Engineered Foreign Genes
Errata and Notes