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First published online 1 September 2005
doi: 10.1242/dev.02013

Development 132, 4397-4406 (2005)
Published by The Company of Biologists 2005
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Shox2-deficient mice exhibit a rare type of incomplete clefting of the secondary palate

Ling Yu1,*, Shuping Gu1,*,{dagger}, Sylvia Alappat1, Yiqiang Song1, Mingquan Yan1, Xiaoyun Zhang1, Guozhong Zhang2, Yiping Jiang1, Zunyi Zhang1, Yanding Zhang2 and YiPing Chen1,2,{ddagger}

1 Division of Developmental Biology, Department of Cell and Molecular Biology, and Center for Bioenvironmental Research, Tulane University, New Orleans, LA 70118, USA
2 College of Bioengineering, Fujian Normal University, Fuzhou, Fujian 350007, PR China

{ddagger} Author for correspondence (e-mail: ychen{at}tulane.edu)

Accepted 28 July 2005

The short stature homeobox gene SHOX is associated with idiopathic short stature in humans, as seen in Turner syndrome and Leri-Weill dyschondrosteosis, while little is known about its close relative SHOX2. We report the restricted expression of Shox2 in the anterior domain of the secondary palate in mice and humans. Shox2-/- mice develop an incomplete cleft that is confined to the anterior region of the palate, an extremely rare type of clefting in humans. The Shox2-/- palatal shelves initiate, grow and elevate normally, but the anterior region fails to contact and fuse at the midline, owing to altered cell proliferation and apoptosis, leading to incomplete clefting within the presumptive hard palate. Accompanied with these cellular alterations is an ectopic expression of Fgf10 and Fgfr2c in the anterior palatal mesenchyme of the mutants. Tissue recombination and bead implantation experiments revealed that signals from the anterior palatal epithelium are responsible for the restricted mesenchymal Shox2 expression. BMP activity is necessary but not sufficient for the induction of palatal Shox2 expression. Our results demonstrate an intrinsic requirement for Shox2 in palatogenesis, and support the idea that palatogenesis is differentially regulated along the anteroposterior axis. Furthermore, our results demonstrate that fusion of the posterior palate can occur independently of fusion in the anterior palate.

Key words: Mouse, Cleft palate, Shox2, Epithelial-mesenchymal interaction




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