Literature DB >> 7758520

A glycine 375-to-cysteine substitution in the transmembrane domain of the fibroblast growth factor receptor-3 in a newborn with achondroplasia.

A Superti-Furga1, G Eich, H U Bucher, J Wisser, A Giedion, R Gitzelmann, B Steinmann.   

Abstract

Achondroplasia, the most common form of chondrodysplasia, has been associated with mutations in the gene of the fibroblast growth factor receptor-3 (FGFR-3) on chromosome 4p. All 39 achondroplasia alleles studied so far carried point mutations which caused the same amino acid exchange, a substitution of glycine by arginine at position 380 (G380R) in the transmembrane domain of the receptor. We report on a newborn with achondroplasia who does not carry a G380R mutation but has a mutation causing substitution of a nearby glycine with a cysteine (G375C). This observation indicates allelic heterogeneity and confirms the role of mutations in the transmembrane domain of FGFR-3 in the pathogenesis of achondroplasia.

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Year:  1995        PMID: 7758520     DOI: 10.1007/BF01954274

Source DB:  PubMed          Journal:  Eur J Pediatr        ISSN: 0340-6199            Impact factor:   3.183


  11 in total

1.  Isolation of an additional member of the fibroblast growth factor receptor family, FGFR-3.

Authors:  K Keegan; D E Johnson; L T Williams; M J Hayman
Journal:  Proc Natl Acad Sci U S A       Date:  1991-02-15       Impact factor: 11.205

2.  A distinctive family of embryonic protein-tyrosine kinase receptors.

Authors:  E B Pasquale
Journal:  Proc Natl Acad Sci U S A       Date:  1990-08       Impact factor: 11.205

3.  A gene encoding a fibroblast growth factor receptor isolated from the Huntington disease gene region of human chromosome 4.

Authors:  L M Thompson; S Plummer; M Schalling; M R Altherr; J F Gusella; D E Housman; J J Wasmuth
Journal:  Genomics       Date:  1991-12       Impact factor: 5.736

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Authors:  M Robinow; W C Chumlea
Journal:  Radiology       Date:  1982-05       Impact factor: 11.105

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Authors:  L O Langer; P A Baumann; R J Gorlin
Journal:  Am J Roentgenol Radium Ther Nucl Med       Date:  1967-05

6.  Ligand specificity and heparin dependence of fibroblast growth factor receptors 1 and 3.

Authors:  D M Ornitz; P Leder
Journal:  J Biol Chem       Date:  1992-08-15       Impact factor: 5.157

7.  Maternal phenylketonuria syndrome in cousins caused by mild, unrecognized phenylketonuria in their mothers homozygous for the phenylalanine hydroxylase Arg-261-Gln mutation.

Authors:  A Superti-Furga; B Steinmann; G Duc; R Gitzelmann
Journal:  Eur J Pediatr       Date:  1991-05       Impact factor: 3.183

8.  A gene for achondroplasia-hypochondroplasia maps to chromosome 4p.

Authors:  M Le Merrer; F Rousseau; L Legeai-Mallet; J C Landais; A Pelet; J Bonaventure; M Sanak; J Weissenbach; C Stoll; A Munnich
Journal:  Nat Genet       Date:  1994-03       Impact factor: 38.330

9.  The gene for achondroplasia maps to the telomeric region of chromosome 4p.

Authors:  M Velinov; S A Slaugenhaupt; I Stoilov; C I Scott; J F Gusella; P Tsipouras
Journal:  Nat Genet       Date:  1994-03       Impact factor: 38.330

10.  Mutations in the gene encoding fibroblast growth factor receptor-3 in achondroplasia.

Authors:  F Rousseau; J Bonaventure; L Legeai-Mallet; A Pelet; J M Rozet; P Maroteaux; M Le Merrer; A Munnich
Journal:  Nature       Date:  1994-09-15       Impact factor: 49.962

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  11 in total

1.  Typical achondroplasia secondary to a unique insertional variant of FGFR3 with in vitro demonstration of its effect on FGFR3 function.

Authors:  April N Meyer; Peggy Modaff; Clark G Wang; Elizabeth Wohler; Nara L Sobreira; Daniel J Donoghue; Richard M Pauli
Journal:  Am J Med Genet A       Date:  2020-12-02       Impact factor: 2.802

Review 2.  Molecular genesis of non-muscle-invasive urothelial carcinoma (NMIUC).

Authors:  Courtney Pollard; Steven C Smith; Dan Theodorescu
Journal:  Expert Rev Mol Med       Date:  2010-03-25       Impact factor: 5.600

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

Review 4.  Advances in research on and diagnosis and treatment of achondroplasia in China.

Authors:  Yao Wang; Zeying Liu; Zhenxing Liu; Heng Zhao; Xiaoyan Zhou; Yazhou Cui; Jinxiang Han
Journal:  Intractable Rare Dis Res       Date:  2013-05

5.  Chimeras of the native form or achondroplasia mutant (G375C) of human fibroblast growth factor receptor 3 induce ligand-dependent differentiation of PC12 cells.

Authors:  L M Thompson; S Raffioni; J J Wasmuth; R A Bradshaw
Journal:  Mol Cell Biol       Date:  1997-07       Impact factor: 4.272

6.  Gly369Cys mutation in mouse FGFR3 causes achondroplasia by affecting both chondrogenesis and osteogenesis.

Authors:  L Chen; R Adar; X Yang; E O Monsonego; C Li; P V Hauschka; A Yayon; C X Deng
Journal:  J Clin Invest       Date:  1999-12       Impact factor: 14.808

Review 7.  FGF signaling in the developing endochondral skeleton.

Authors:  David M Ornitz
Journal:  Cytokine Growth Factor Rev       Date:  2005-04-01       Impact factor: 7.638

8.  A novel skeletal dysplasia with developmental delay and acanthosis nigricans is caused by a Lys650Met mutation in the fibroblast growth factor receptor 3 gene.

Authors:  P L Tavormina; G A Bellus; M K Webster; M J Bamshad; A E Fraley; I McIntosh; J Szabo; W Jiang; E W Jabs; W R Wilcox; J J Wasmuth; D J Donoghue; L M Thompson; C A Francomano
Journal:  Am J Hum Genet       Date:  1999-03       Impact factor: 11.025

9.  Analysis of the clinical and molecular characteristics of a child with achondroplasia: A case report.

Authors:  Jingfang Liu; Xulei Tang; Jianguo Cheng; Liting Wang; Xiaomei Yang; Yan Wang
Journal:  Exp Ther Med       Date:  2015-03-02       Impact factor: 2.447

10.  Constitutive receptor activation by Crouzon syndrome mutations in fibroblast growth factor receptor (FGFR)2 and FGFR2/Neu chimeras.

Authors:  B D Galvin; K C Hart; A N Meyer; M K Webster; D J Donoghue
Journal:  Proc Natl Acad Sci U S A       Date:  1996-07-23       Impact factor: 11.205

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