Tbx1, a DiGeorge syndrome candidate gene, is regulated by sonic hedgehog during pharyngeal arch development

Vidu Garg, Chihiro Yamagishi, Tonghuan Hu, Irfan S. Kathiriya, Hiroyuki Yamagishi, Deepak Srivastava

研究成果: Article査読

235 被引用数 (Scopus)

抄録

Appropriate interactions between the epithelium and adjacent neural crest-derived mesenchyme are necessary for normal pharyngeal arch development. Disruption of pharyngeal arch development in humans underlies many of the craniofacial defects observed in the 22q11.2 deletion syndrome (del22q11), but the genes responsible remain unknown. Tbx1 is a T-box transcription factor that lies in the 22q11.2 locus. Tbx1 transcripts were found to be localized to the pharyngeal endoderm and the mesodermal core of the pharyngeal arches, but were not present in the neural crest-derived mesenchyme of the pharyngeal arches. Sonic hedgehog (Shh) is also expressed in the pharyngeal arches and is necessary for normal craniofacial development. We found that Tbx1 expression was dependent upon Shh signaling in mouse embryos, consistent with their overlapping expression in the pharyngeal arches. Furthermore, Shh was sufficient to induce Tbx1 expression when misexpressed in selected regions of chick embryos. These studies reveal a Shh-mediated pathway that regulates Tbx1 during pharyngeal arch development.

本文言語English
論文番号90283
ページ(範囲)62-73
ページ数12
ジャーナルDevelopmental Biology
235
1
DOI
出版ステータスPublished - 2001
外部発表はい

ASJC Scopus subject areas

  • 分子生物学
  • 発生生物学
  • 細胞生物学

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