Literature DB >> 24048522

Postnatal soluble FGFR3 therapy rescues achondroplasia symptoms and restores bone growth in mice.

Stéphanie Garcia1, Béatrice Dirat, Thomas Tognacci, Nathalie Rochet, Xavier Mouska, Stéphanie Bonnafous, Stéphanie Patouraux, Albert Tran, Philippe Gual, Yannick Le Marchand-Brustel, Isabelle Gennero, Elvire Gouze.   

Abstract

Achondroplasia is a rare genetic disease characterized by abnormal bone development, resulting in short stature. It is caused by a single point mutation in the gene coding for fibroblast growth factor receptor 3 (FGFR3), which leads to prolonged activation upon ligand binding. To prevent excessive intracellular signaling and rescue the symptoms of achondroplasia, we have developed a recombinant protein therapeutic approach using a soluble form of human FGFR3 (sFGFR3), which acts as a decoy receptor and prevents FGF from binding to mutant FGFR3. sFGFR3 was injected subcutaneously to newborn Fgfr3(ach/+) mice-the mouse model of achondroplasia-twice per week throughout the growth period during 3 weeks. Effective maturation of growth plate chondrocytes was restored in bones of treated mice, with a dose-dependent enhancement of skeletal growth in Fgfr3(ach/+) mice. This resulted in normal stature and a significant decrease in mortality and associated complications, without any evidence of toxicity. These results describe a new approach for restoring bone growth and suggest that sFGFR3 could be a potential therapy for children with achondroplasia and related disorders.

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Year:  2013        PMID: 24048522     DOI: 10.1126/scitranslmed.3006247

Source DB:  PubMed          Journal:  Sci Transl Med        ISSN: 1946-6234            Impact factor:   17.956


  34 in total

Review 1.  Advances in Skeletal Dysplasia Genetics.

Authors:  Krista A Geister; Sally A Camper
Journal:  Annu Rev Genomics Hum Genet       Date:  2015-04-22       Impact factor: 8.929

2.  Cell reprogramming for skeletal dysplasia drug repositioning.

Authors:  Peter Karagiannis; Noriyuki Tsumaki
Journal:  Cell Cycle       Date:  2014       Impact factor: 4.534

Review 3.  Achondroplasia: Development, pathogenesis, and therapy.

Authors:  David M Ornitz; Laurence Legeai-Mallet
Journal:  Dev Dyn       Date:  2017-03-02       Impact factor: 3.780

4.  Statin treatment rescues FGFR3 skeletal dysplasia phenotypes.

Authors:  Akihiro Yamashita; Miho Morioka; Hiromi Kishi; Takeshi Kimura; Yasuhito Yahara; Minoru Okada; Kaori Fujita; Hideaki Sawai; Shiro Ikegawa; Noriyuki Tsumaki
Journal:  Nature       Date:  2014-09-17       Impact factor: 49.962

5.  Mutant activated FGFR3 impairs endochondral bone growth by preventing SOX9 downregulation in differentiating chondrocytes.

Authors:  Zi-Qiang Zhou; Sara Ota; Chuxia Deng; Haruhiko Akiyama; Peter J Hurlin
Journal:  Hum Mol Genet       Date:  2014-11-28       Impact factor: 6.150

Review 6.  Molecular therapeutic strategies for FGFR3 gene-related skeletal dysplasia.

Authors:  Jia Chen; Jiaqi Liu; Yangzhong Zhou; Sen Liu; Gang Liu; Yuzhi Zuo; Zhihong Wu; Nan Wu; Guixing Qiu
Journal:  J Mol Med (Berl)       Date:  2017-10-23       Impact factor: 4.599

7.  FGFR3/fibroblast growth factor receptor 3 inhibits autophagy through decreasing the ATG12-ATG5 conjugate, leading to the delay of cartilage development in achondroplasia.

Authors:  Xiaofeng Wang; Huabing Qi; Quan Wang; Ying Zhu; Xianxing Wang; Min Jin; Qiaoyan Tan; Qizhao Huang; Wei Xu; Xiaogang Li; Liang Kuang; Yubing Tang; Xiaolan Du; Di Chen; Lin Chen
Journal:  Autophagy       Date:  2015-11-02       Impact factor: 16.016

Review 8.  Achondroplasia: a comprehensive clinical review.

Authors:  Richard M Pauli
Journal:  Orphanet J Rare Dis       Date:  2019-01-03       Impact factor: 4.123

9.  Tyrosine kinase inhibitor NVP-BGJ398 functionally improves FGFR3-related dwarfism in mouse model.

Authors:  Davide Komla-Ebri; Emilie Dambroise; Ina Kramer; Catherine Benoist-Lasselin; Nabil Kaci; Cindy Le Gall; Ludovic Martin; Patricia Busca; Florent Barbault; Diana Graus-Porta; Arnold Munnich; Michaela Kneissel; Federico Di Rocco; Martin Biosse-Duplan; Laurence Legeai-Mallet
Journal:  J Clin Invest       Date:  2016-04-11       Impact factor: 14.808

Review 10.  Recent research on the growth plate: Recent insights into the regulation of the growth plate.

Authors:  Julian C Lui; Ola Nilsson; Jeffrey Baron
Journal:  J Mol Endocrinol       Date:  2014-04-16       Impact factor: 5.098

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