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Open Access Highly Accessed Open Badges Research article

Conditional expression of Spry1 in neural crest causes craniofacial and cardiac defects

Xuehui Yang1, Sean Kilgallen1, Viktoria Andreeva12, Douglas B Spicer1, Ilka Pinz1 and Robert Friesel1*

Author Affiliations

1 Center for Molecular Medicine, Maine Medical Center Research Institute, Scarborough, ME 04074, USA

2 Department of Dental Research, Tufts University School of Dental Medicine, Boston, MA 02111, USA

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BMC Developmental Biology 2010, 10:48  doi:10.1186/1471-213X-10-48

Published: 11 May 2010



Growth factors and their receptors are mediators of organogenesis and must be tightly regulated in a temporal and spatial manner for proper tissue morphogenesis. Intracellular regulators of growth factor signaling pathways provide an additional level of control. Members of the Sprouty family negatively regulate receptor tyrosine kinase pathways in several developmental contexts. To gain insight into the role of Spry1 in neural crest development, we analyzed the developmental effects of conditional expression of Spry1 in neural crest-derived tissues.


Here we report that conditional expression of Spry1 in neural crest cells causes defects in craniofacial and cardiac development in mice. Spry1;Wnt1-Cre embryos die perinatally and exhibit facial clefting, cleft palate, cardiac and cranial nerve defects. These defects appear to be the result of decreased proliferation and increased apoptosis of neural crest and neural crest-derived cell populations. In addition, the domains of expression of several key transcription factors important to normal craniofacial and cardiac development including AP2, Msx2, Dlx5, and Dlx6 were reduced in Spry1;Wnt1-Cre transgenic embryos.


Collectively, these data suggest that Spry1 is an important regulator of craniofacial and cardiac morphogenesis and perturbations in Spry1 levels may contribute to congenital disorders involving tissues of neural crest origin.