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Understanding craniofacial variability caused by loss of Gata3 function
Author(s) -
Eberhart Johann K.,
Swartz Mary E.
Publication year - 2013
Publication title -
the faseb journal
Language(s) - English
Resource type - Journals
SCImago Journal Rank - 1.709
H-Index - 277
eISSN - 1530-6860
pISSN - 0892-6638
DOI - 10.1096/fasebj.27.1_supplement.967.4
Subject(s) - neural crest , phenotype , biology , zebrafish , mutant , microbiology and biotechnology , craniofacial , loss function , gata3 , genetics , anatomy , embryo , gene , transcription factor
Craniofacial malformations are extremely variable and we seek to understand the causes of this variability. We identified a zebrafish gata3 mutant with defects to the palatal skeleton; these defects are highly variable dependant upon the genetic background. Mutants in one inbred genetic background almost completely lose the trabeculae, a portion of the zebrafish palate. Lineage tracing of the trabeculae precursors shows that these cells become mislocalized anterior to their normal location. In a second genetic background, the trabeculae are present but the chondrocytes are not stacked out as a single row of flattened cells, as is found in wild‐type embryos. This failure of stacking results in shortened trabeculae and is consistent with a defect in cell intercalations. In order to characterize these background‐dependent phenotypic differences, we are seeking to understand the signaling pathways regulating gata3 ‐mediated cellular behaviors. We have found neural crest cells express gata3 and require the function of Gata3 for proper palatogenesis. Loss of Smad5 function causes loss of gata3 expression in the neural crest, consistent with the more severe mutant phenotype resembling Bmp loss of function phenotypes. We are currently examining neural crest cell movements and the expression of members of the planar cell polarity pathway in gata3 mutants in both genetic backgrounds. This work is supported by NIH R00DE018088.