%0 Journal Article
%T Conditional expression of Spry1 in neural crest causes craniofacial and cardiac defects
%A Xuehui Yang
%A Sean Kilgallen
%A Viktoria Andreeva
%A Douglas B Spicer
%A Ilka Pinz
%A Robert Friesel
%J BMC Developmental Biology
%D 2010
%I BioMed Central
%R 10.1186/1471-213x-10-48
%X 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.Neural crest cells (NCC) are pleuripotent cells that migrate out of the dorsal neural tube during early vertebrate embryogenesis to populate many anatomical structures along the dorsoventral axis [1,2]. Cranial NCC migrate ventrolaterally from the forebrain and hindbrain region to populate craniofacial structures and branchial arches. The proliferation of cranial NCC results in a demarcation of each branchial arch. Once migration is complete, cranial NCC contribute to the maxilla, mandible, cranial ganglia, and other mesenchymally derived structures of the head and neck. Cardiac NCC emanating from rhombomeres 6-8 populate branchial arches 3, 4, and 6. Some cardiac NCC contributes to the development of the branchial arch arteries, cardiac outflow tract, and the spiral septum between the ascending aorta and the main pulmonary artery. Other cardiac NCC contribute to the formation of the outflow tract cushions/endocardial cushions and subsequently the semilunar valves and interventricular septum. Perturbations in normal neural crest development cause several congenital craniofacial and cardiac defects.Cell-cell and tissue interactions are
%U http://www.biomedcentral.com/1471-213X/10/48