A specific requirement for PDGF-C in palate formation and PDGFR-α signaling

Hao Ding, Xiaoli Wu, Hans Boström, Injune Kim, Nicole Wong, Bonny Tsoi, Meredith O'Rourke, Gou Young Koh, Philippe Soriano, Christer Betsholtz, Thomas C. Hart, Mary L. Marazita, L. L. Field, Patrick P.L. Tam, Andras Nagy

Research output: Contribution to journalArticleResearchpeer-review

Abstract

PDGF-C is a member of the platelet-derived growth factor (PDGF) family, which signals through PDGF receptor (PDGFR) αα and αβ 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-α function, suggesting that both PDGF-A and PDGF-C signal through PDGFR-α 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.

Original languageEnglish
Pages (from-to)1111-1116
Number of pages6
JournalNature Genetics
Volume36
Issue number10
DOIs
Publication statusPublished - Oct 2004
Externally publishedYes

Cite this

Ding, H., Wu, X., Boström, H., Kim, I., Wong, N., Tsoi, B., ... Nagy, A. (2004). A specific requirement for PDGF-C in palate formation and PDGFR-α signaling. Nature Genetics, 36(10), 1111-1116. https://doi.org/10.1038/ng1415
Ding, Hao ; Wu, Xiaoli ; Boström, Hans ; Kim, Injune ; Wong, Nicole ; Tsoi, Bonny ; O'Rourke, Meredith ; Koh, Gou Young ; Soriano, Philippe ; Betsholtz, Christer ; Hart, Thomas C. ; Marazita, Mary L. ; Field, L. L. ; Tam, Patrick P.L. ; Nagy, Andras. / A specific requirement for PDGF-C in palate formation and PDGFR-α signaling. In: Nature Genetics. 2004 ; Vol. 36, No. 10. pp. 1111-1116.
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abstract = "PDGF-C is a member of the platelet-derived growth factor (PDGF) family, which signals through PDGF receptor (PDGFR) αα and αβ 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-α function, suggesting that both PDGF-A and PDGF-C signal through PDGFR-α 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.",
author = "Hao Ding and Xiaoli Wu and Hans Bostr{\"o}m and Injune Kim and Nicole Wong and Bonny Tsoi and Meredith O'Rourke and Koh, {Gou Young} and Philippe Soriano and Christer Betsholtz and Hart, {Thomas C.} and Marazita, {Mary L.} and Field, {L. L.} and Tam, {Patrick P.L.} and Andras Nagy",
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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, PPL & Nagy, A 2004, 'A specific requirement for PDGF-C in palate formation and PDGFR-α signaling' Nature Genetics, vol. 36, no. 10, pp. 1111-1116. https://doi.org/10.1038/ng1415

A specific requirement for PDGF-C in palate formation and PDGFR-α signaling. / Ding, Hao; Wu, Xiaoli; Boström, Hans; Kim, Injune; Wong, Nicole; Tsoi, Bonny; O'Rourke, Meredith; Koh, Gou Young; Soriano, Philippe; Betsholtz, Christer; Hart, Thomas C.; Marazita, Mary L.; Field, L. L.; Tam, Patrick P.L.; Nagy, Andras.

In: Nature Genetics, Vol. 36, No. 10, 10.2004, p. 1111-1116.

Research output: Contribution to journalArticleResearchpeer-review

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AU - O'Rourke, Meredith

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AU - Soriano, Philippe

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Ding H, Wu X, Boström H, Kim I, Wong N, Tsoi B et al. A specific requirement for PDGF-C in palate formation and PDGFR-α signaling. Nature Genetics. 2004 Oct;36(10):1111-1116. https://doi.org/10.1038/ng1415