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Alexander, C., Zuniga, E., Blitz, I.L., Wada, N., Le, P, Javidan, P., Zhang, Y., Cho, T., Crump, J.G. and Schilling T.F. (2011) Combinatorial roles for BMPs and Endothelin 1 in patterning the dorsal-ventral axis of the craniofacial skeleton. Development, 138, 5135-5146. doi:10.1242/dev.067801

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• TITLE: Early expression of the tbx22 gene in zebrafish influences positioning of pharyngeal arch cartilages

  AUTHORS: Tammy Lynne Silva, Angela Allard Cox, V. P. Boominathan, Peter Alan Jezewski, Tracie Lee Ferreira

  KEYWORDS: Zebrafish; Tbx Signaling; Pharyngeal Pouch

  JOURNAL NAME: American Journal of Molecular Biology, Vol.2 No.4, October 31, 2012

  ABSTRACT: Mutations in human TBX22 cause X-linked cleft palate with anklyoglossia syndrome. The two zebrafish tbx22 splice isoforms, tbx22-1 and tbx22-2, encode proteins of 444 and 400 amino acids, respectively. Zebrafish tbx22 mRNA expression mirrors mammalian TBX22 expression and is consistent with early patterning of the vertebrate face. In zebrafish, tbx22 mRNA is strongly expressed during early pharyngeal arch development in the ventral mesenchyme, and a later expression domain is found in ectomesenchymal cells underlying the stomodeum, a bilaminar epithelial structure demarcating the early forming mouth. Therefore, tbx22 is hypothesized to be involved in craniofacial development. The objective of this work is to characterize the role of tbx22 during craniofacial development in zebrafish. Tbx22 knockdown revealed that defects in tbx22 signaling cause mild clefting, joint defects and dorsoventral patterning defects in cartilages. Quantitative PCR and in situ analysis revealed that knockdown of tbx22 also causes a dramatic decrease in expression of osr1 and gdf5. Craniofacial patterning is dependent on proper signals from endoderm, mesoderm and ectoderm. The early influence of tbx22 on signals within the ventral mesenchyme impacts the domains of several key pharynx-geal arch signals, thereby helping to regulate proper patterning of the developing jaw.