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FGF9 regulates early hypertrophic chondrocyte differentiation and skeletal vascularization in the developing stylopod.
Dev Biol. 2007 Jul 15; 307(2):300-13.DB

Abstract

Gain-of-function mutations in fibroblast growth factor (FGF) receptors result in chondrodysplasia and craniosynostosis syndromes, highlighting the critical role for FGF signaling in skeletal development. Although the FGFRs involved in skeletal development have been well characterized, only a single FGF ligand, FGF18, has been identified that regulates skeletal development during embryogenesis. Here we identify Fgf9 as a second FGF ligand that is critical for skeletal development. We show that Fgf9 is expressed in the proximity of developing skeletal elements and that Fgf9-deficient mice exhibit rhizomelia (a disproportionate shortening of proximal skeletal elements), which is a prominent feature of patients with FGFR3-induced chondrodysplasia syndromes. Although Fgf9 is expressed in the apical ectodermal ridge in the limb bud, we demonstrate that the Fgf9-/- limb phenotype results from loss of FGF9 functions after formation of the mesenchymal condensation. In developing stylopod elements, FGF9 promotes chondrocyte hypertrophy at early stages and regulates vascularization of the growth plate and osteogenesis at later stages of skeletal development.

Authors+Show Affiliations

Department of Molecular Biology and Pharmacology, Washington University School of Medicine, Campus Box 8103, 660 S. Euclid Avenue, St. Louis, MO 63110, USA.No affiliation info availableNo affiliation info availableNo affiliation info available

Pub Type(s)

Journal Article
Research Support, N.I.H., Extramural

Language

eng

PubMed ID

17544391

Citation

Hung, Irene H., et al. "FGF9 Regulates Early Hypertrophic Chondrocyte Differentiation and Skeletal Vascularization in the Developing Stylopod." Developmental Biology, vol. 307, no. 2, 2007, pp. 300-13.
Hung IH, Yu K, Lavine KJ, et al. FGF9 regulates early hypertrophic chondrocyte differentiation and skeletal vascularization in the developing stylopod. Dev Biol. 2007;307(2):300-13.
Hung, I. H., Yu, K., Lavine, K. J., & Ornitz, D. M. (2007). FGF9 regulates early hypertrophic chondrocyte differentiation and skeletal vascularization in the developing stylopod. Developmental Biology, 307(2), 300-13.
Hung IH, et al. FGF9 Regulates Early Hypertrophic Chondrocyte Differentiation and Skeletal Vascularization in the Developing Stylopod. Dev Biol. 2007 Jul 15;307(2):300-13. PubMed PMID: 17544391.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - FGF9 regulates early hypertrophic chondrocyte differentiation and skeletal vascularization in the developing stylopod. AU - Hung,Irene H, AU - Yu,Kai, AU - Lavine,Kory J, AU - Ornitz,David M, Y1 - 2007/05/06/ PY - 2007/03/01/received PY - 2007/04/05/revised PY - 2007/04/30/accepted PY - 2007/6/5/pubmed PY - 2007/9/6/medline PY - 2007/6/5/entrez SP - 300 EP - 13 JF - Developmental biology JO - Dev. Biol. VL - 307 IS - 2 N2 - Gain-of-function mutations in fibroblast growth factor (FGF) receptors result in chondrodysplasia and craniosynostosis syndromes, highlighting the critical role for FGF signaling in skeletal development. Although the FGFRs involved in skeletal development have been well characterized, only a single FGF ligand, FGF18, has been identified that regulates skeletal development during embryogenesis. Here we identify Fgf9 as a second FGF ligand that is critical for skeletal development. We show that Fgf9 is expressed in the proximity of developing skeletal elements and that Fgf9-deficient mice exhibit rhizomelia (a disproportionate shortening of proximal skeletal elements), which is a prominent feature of patients with FGFR3-induced chondrodysplasia syndromes. Although Fgf9 is expressed in the apical ectodermal ridge in the limb bud, we demonstrate that the Fgf9-/- limb phenotype results from loss of FGF9 functions after formation of the mesenchymal condensation. In developing stylopod elements, FGF9 promotes chondrocyte hypertrophy at early stages and regulates vascularization of the growth plate and osteogenesis at later stages of skeletal development. SN - 0012-1606 UR - https://www.unboundmedicine.com/medline/citation/17544391/FGF9_regulates_early_hypertrophic_chondrocyte_differentiation_and_skeletal_vascularization_in_the_developing_stylopod_ L2 - https://linkinghub.elsevier.com/retrieve/pii/S0012-1606(07)00871-8 DB - PRIME DP - Unbound Medicine ER -