Literature DB >> 20624921

Physical basis behind achondroplasia, the most common form of human dwarfism.

Lijuan He1, William Horton, Kalina Hristova.   

Abstract

Fibroblast growth factor receptor 3 (FGFR3) is a receptor tyrosine kinase that plays an important role in long bone development. The G380R mutation in FGFR3 transmembrane domain is known as the genetic cause for achondroplasia, the most common form of human dwarfism. Despite many studies, there is no consensus about the exact mechanism underlying the pathology. To gain further understanding into the physical basis behind the disorder, here we measure the activation of wild-type and mutant FGFR3 in mammalian cells using Western blots, and we analyze the activation within the frame of a physical-chemical model describing dimerization, ligand binding, and phosphorylation probabilities within the dimers. The data analysis presented here suggests that the mutation does not increase FGFR3 dimerization, as proposed previously. Instead, FGFR3 activity in achondroplasia is increased due to increased probability for phosphorylation of the unliganded mutant dimers. This finding has implications for the design of targeted molecular treatments for achondroplasia.

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Year:  2010        PMID: 20624921      PMCID: PMC2943285          DOI: 10.1074/jbc.M109.094086

Source DB:  PubMed          Journal:  J Biol Chem        ISSN: 0021-9258            Impact factor:   5.157


  32 in total

1.  Insights into the molecular basis for fibroblast growth factor receptor autoinhibition and ligand-binding promiscuity.

Authors:  Shaun K Olsen; Omar A Ibrahimi; Angela Raucci; Fuming Zhang; Anna V Eliseenkova; Avner Yayon; Claudio Basilico; Robert J Linhardt; Joseph Schlessinger; Moosa Mohammadi
Journal:  Proc Natl Acad Sci U S A       Date:  2004-01-19       Impact factor: 11.205

2.  Common and distinct elements in cellular signaling via EGF and FGF receptors.

Authors:  Joseph Schlessinger
Journal:  Science       Date:  2004-11-26       Impact factor: 47.728

3.  Improved prediction for the structure of the dimeric transmembrane domain of glycophorin A obtained through global searching.

Authors:  P D Adams; D M Engelman; A T Brünger
Journal:  Proteins       Date:  1996-11

Review 4.  FGFR activation in skeletal disorders: too much of a good thing.

Authors:  M K Webster; D J Donoghue
Journal:  Trends Genet       Date:  1997-05       Impact factor: 11.639

5.  Activation of FGF receptors by mutations in the transmembrane domain.

Authors:  Y Li; K Mangasarian; A Mansukhani; C Basilico
Journal:  Oncogene       Date:  1997-03-27       Impact factor: 9.867

6.  Skeletal growth in achondroplasia.

Authors:  I V Ponseti
Journal:  J Bone Joint Surg Am       Date:  1970-06       Impact factor: 5.284

7.  Computational searching and mutagenesis suggest a structure for the pentameric transmembrane domain of phospholamban.

Authors:  P D Adams; I T Arkin; D M Engelman; A T Brünger
Journal:  Nat Struct Biol       Date:  1995-02

8.  Effect of transmembrane and kinase domain mutations on fibroblast growth factor receptor 3 chimera signaling in PC12 cells. A model for the control of receptor tyrosine kinase activation.

Authors:  S Raffioni; Y Z Zhu; R A Bradshaw; L M Thompson
Journal:  J Biol Chem       Date:  1998-12-25       Impact factor: 5.157

9.  Constitutive activation of fibroblast growth factor receptor 3 by the transmembrane domain point mutation found in achondroplasia.

Authors:  M K Webster; D J Donoghue
Journal:  EMBO J       Date:  1996-02-01       Impact factor: 11.598

10.  Skeletal overgrowth and deafness in mice lacking fibroblast growth factor receptor 3.

Authors:  J S Colvin; B A Bohne; G W Harding; D G McEwen; D M Ornitz
Journal:  Nat Genet       Date:  1996-04       Impact factor: 38.330

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  25 in total

1.  The RTK Interactome: Overview and Perspective on RTK Heterointeractions.

Authors:  Michael D Paul; Kalina Hristova
Journal:  Chem Rev       Date:  2018-12-27       Impact factor: 60.622

2.  FGFR3 unliganded dimer stabilization by the juxtamembrane domain.

Authors:  Sarvenaz Sarabipour; Kalina Hristova
Journal:  J Mol Biol       Date:  2015-02-15       Impact factor: 5.469

3.  Analytical characterization of plasma membrane-derived vesicles produced via osmotic and chemical vesiculation.

Authors:  Sarvenaz Sarabipour; Robin B Chan; Bowen Zhou; Gilbert Di Paolo; Kalina Hristova
Journal:  Biochim Biophys Acta       Date:  2015-04-17

4.  High-throughput selection of transmembrane sequences that enhance receptor tyrosine kinase activation.

Authors:  Lijuan He; Andrew R Hoffmann; Christopher Serrano; Kalina Hristova; William C Wimley
Journal:  J Mol Biol       Date:  2011-07-12       Impact factor: 5.469

Review 5.  Transmembrane helix dimerization: beyond the search for sequence motifs.

Authors:  Edwin Li; William C Wimley; Kalina Hristova
Journal:  Biochim Biophys Acta       Date:  2011-09-01

6.  Pathogenic Cysteine Removal Mutations in FGFR Extracellular Domains Stabilize Receptor Dimers and Perturb the TM Dimer Structure.

Authors:  Sarvenaz Sarabipour; Kalina Hristova
Journal:  J Mol Biol       Date:  2016-09-03       Impact factor: 5.469

7.  FGFR3 transmembrane domain interactions persist in the presence of its extracellular domain.

Authors:  Sarvenaz Sarabipour; Kalina Hristova
Journal:  Biophys J       Date:  2013-07-02       Impact factor: 4.033

8.  The A391E mutation enhances FGFR3 activation in the absence of ligand.

Authors:  Fenghao Chen; Catherine Degnin; Melanie Laederich; William A Horton; Kalina Hristova
Journal:  Biochim Biophys Acta       Date:  2011-04-22

9.  Quantifying the strength of heterointeractions among receptor tyrosine kinases from different subfamilies: Implications for cell signaling.

Authors:  Michael D Paul; Hana N Grubb; Kalina Hristova
Journal:  J Biol Chem       Date:  2020-05-27       Impact factor: 5.157

10.  FGFR3 induces degradation of BMP type I receptor to regulate skeletal development.

Authors:  Huabing Qi; Min Jin; Yaqi Duan; Xiaolan Du; Yuanquan Zhang; Fangli Ren; Yinyin Wang; Qingyun Tian; Xiaofeng Wang; Quan Wang; Ying Zhu; Yangli Xie; Chuanju Liu; Xu Cao; Yuji Mishina; Di Chen; Chu-xia Deng; Zhijie Chang; Lin Chen
Journal:  Biochim Biophys Acta       Date:  2014-03-20
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