Journal
DEVELOPMENTAL BIOLOGY
Volume 344, Issue 2, Pages 720-730Publisher
ACADEMIC PRESS INC ELSEVIER SCIENCE
DOI: 10.1016/j.ydbio.2010.05.509
Keywords
Six2; Cranial base; Endochondral ossification; Development; Mouse
Categories
Funding
- BBSRC [BB/E017355/1]
- Manchester Academic Health Science Centre
- Manchester NIHR Biomedical Research Centre
- BBSRC [BB/E017355/1] Funding Source: UKRI
- Biotechnology and Biological Sciences Research Council [BB/E017355/1] Funding Source: researchfish
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The cranial base is essential for integrated craniofacial development and growth. It develops as a cartilaginous template that is replaced by bone through the process of endochondral ossification. Here, we describe a novel and specific role for the homeoprotein Six2 in the growth and elongation of the cranial base. Six2-null newborn mice display premature fusion of the bones in the cranial base. Chondrocyte differentiation is abnormal in the Six2-null cranial base, with reduced proliferation and increased terminal differentiation. Gain-of-function experiments indicate that Six2 promotes cartilage development and growth in other body areas and appears therefore to control general regulators of chondrocyte differentiation. Our data indicate that the main factors restricting Six2 function to the cranial base are tissue-specific transcription of the gene and compensatory effects of other Six family members. The comparable expression during human embryogenesis and the high protein conservation from mouse to human implicate SIX2 loss-of-function as a potential congenital cause of anterior cranial base defects in humans. (C) 2010 Elsevier Inc. All rights reserved.
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