Craniofacial malformation in R-spondin2 knockout mice

Wakako Yamada, Kenji Nagao, Kaori Horikoshi, Ayako Fujikura, Eiji Ikeda, Yoshimasa Inagaki, Makoto Kakitani, Kazuma Tomizuka, Hiroshi Miyazaki, Toshio Suda, Keiyo Takubo

研究成果: Article査読

58 被引用数 (Scopus)

抄録

In vertebrates, craniofacial formation is accomplished by synergistic interaction of many small elements which are generated independently from distinct germ layers. Because of its complexity, the imbalance of one signaling cascade such as Wnt/β-catenin pathway easily leads to craniofacial malformation, which is the most frequent birth defect in humans. To investigate the developmental role of a newly identified activator of Wnt/β-catenin signaling, Rspo2, we generated and characterized Rspo2-/- mice. We found CLP with mild facial skeletal defects in Rspo2-/- mice. Additionally, Rspo2-/- mice also exhibited distal limb loss and lung hypoplasia, and died immediately after birth with respiratory failure. We showed the apparent reduction of Wnt/β-catenin signaling activity at the branchial arch and the apical ectodermal ridge in Rspo2-/- mice. These findings indicate that Rspo2 regulates midfacial, limb, and lung morphogenesis during development through the Wnt/β-catenin signaling.

本文言語English
ページ(範囲)453-458
ページ数6
ジャーナルBiochemical and Biophysical Research Communications
381
3
DOI
出版ステータスPublished - 2009 4 10

ASJC Scopus subject areas

  • 生物理学
  • 生化学
  • 分子生物学
  • 細胞生物学

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