A combined series of Fgf9 and Fgf18 mutant alleles identifies unique and redundant roles in skeletal development

Developmental Biology
Irene H HungDavid M Ornitz

Abstract

Fibroblast growth factor (FGF) signaling is a critical regulator of skeletal development. Fgf9 and Fgf18 are the only FGF ligands with identified functions in embryonic bone growth. Mice lacking Fgf9 or Fgf18 have distinct skeletal phenotypes; however, the extent of overlapping or redundant functions for these ligands and the stage-specific contributions of FGF signaling to chondrogenesis and osteogenesis are not known. To identify separate versus shared roles for FGF9 and FGF18, we generated a combined series of Fgf9 and Fgf18 null alleles. Analysis of embryos lacking alleles of Fgf9 and Fgf18 shows that both encoded ligands function redundantly to control all stages of skeletogenesis; however, they have variable potencies along the proximodistal limb axis, suggesting gradients of activity during formation of the appendicular skeleton. Congenital absence of both Fgf9 and Fgf18 results in a striking osteochondrodysplasia and revealed functions for FGF signaling in early proximal limb chondrogenesis. Additional defects were also noted in craniofacial bones, vertebrae, and ribs. Loss of alleles of Fgf9 and Fgf18 also affect the expression of genes encoding other key intrinsic skeletal regulators, including IHH, PTHLH (PTHrP), and...Continue Reading

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Citations

Jul 16, 2016·Seminars in Cell & Developmental Biology·William E SamsaGuang Zhou
Dec 18, 2016·Developmental Dynamics : an Official Publication of the American Association of Anatomists·David M Ornitz, Laurence Legeai-Mallet
May 13, 2018·Disease Models & Mechanisms·Linnea SchmidtTrevor Williams
Jun 18, 2019·Journal of Bone and Mineral Research : the Official Journal of the American Society for Bone and Mineral Research·Jennifer McKenzieDavid M Ornitz
Jul 11, 2019·Developmental Dynamics : an Official Publication of the American Association of Anatomists·Andrew S HaganDavid M Ornitz
May 27, 2020·Human Molecular Genetics·Anthony D BirdVincent R Harley
Aug 3, 2017·Molecular Medicine Reports·Feng ZhaiHaijun Wang
Sep 22, 2020·EFORT Open Reviews·Yücel Ağırdil
Jan 9, 2019·Frontiers in Genetics·Chunyan ZhangGraham S Plastow
Dec 15, 2020·Journal of Bone and Mineral Research : the Official Journal of the American Society for Bone and Mineral Research·Lingyun TangZhugang Wang
Feb 13, 2021·The Spine Journal : Official Journal of the North American Spine Society·Hasibullah HabibiHiroaki Nakamura
Jun 7, 2021·Developmental Dynamics : an Official Publication of the American Association of Anatomists·Connor C LeekMegan L Killian
Aug 8, 2021·Seminars in Cell & Developmental Biology·Megan L Killian

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