PMID:15361870

Citation	Ding, H, Wu, X, Boström, H, Kim, I, Wong, N, Tsoi, B, O'Rourke, M, Koh, GY, Soriano, P, Betsholtz, C, Hart, TC, Marazita, ML, Field, LL, Tam, PP and Nagy, A (2004) A specific requirement for PDGF-C in palate formation and PDGFR-alpha signaling. Nat. Genet. 36:1111-6
Abstract	PDGF-C is a member of the platelet-derived growth factor (PDGF) family, which signals through PDGF receptor (PDGFR) alphaalpha and alphabeta dimers. Here we show that Pdgfc(-/-) mice die in the perinatal period owing to feeding and respiratory difficulties associated with a complete cleft of the secondary palate. This phenotype was less severe than that of Pdgfra(-/-) embryos. Pdgfc(-/-) Pdgfa(-/-) embryos developed a cleft face, subepidermal blistering, deficiency of renal cortex mesenchyme, spina bifida and skeletal and vascular defects. Complete loss of function of both ligands, therefore, phenocopied the loss of PDGFR-alpha function, suggesting that both PDGF-A and PDGF-C signal through PDGFR-alpha to regulate the development of craniofacial structures, the neural tube and mesodermal organs. Our results also show that PDGF-C signaling is a new pathway in palatogenesis, different from, and independent of, those previously implicated.
Links	PubMed Online version:10.1038/ng1415
Keywords	Abnormalities, Multiple/embryology; Abnormalities, Multiple/genetics; Animals; Animals, Newborn; Cleft Palate/embryology; Cleft Palate/genetics; Gene Expression Regulation, Developmental; Lymphokines; Mice; Mice, Knockout; Palate/embryology; Phenotype; Platelet-Derived Growth Factor/deficiency; Platelet-Derived Growth Factor/genetics; Platelet-Derived Growth Factor/physiology; Receptor, Platelet-Derived Growth Factor alpha/deficiency; Receptor, Platelet-Derived Growth Factor alpha/genetics; Receptor, Platelet-Derived Growth Factor alpha/physiology; Signal Transduction; Spina Bifida Occulta/embryology; Spina Bifida Occulta/genetics
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