Literature DB >> 10735635

Genotype-phenotype analysis in Apert syndrome suggests opposite effects of the two recurrent mutations on syndactyly and outcome of craniofacial surgery.

S von Gernet1, A Golla, Y Ehrenfels, S Schuffenhauer, J D Fairley.   

Abstract

Apert syndrome is an autosomal dominant condition characterized by craniosynostosis and severe syndactyly, caused by two recurrent mutations in the fibroblast growth factor receptor 2 gene (FGFR2). The genotype-phenotype correlations of 21 patients with Apert syndrome were analysed as to the craniofacial appearance following surgery and the degree of syndactlyly. The craniofacial appearance following craniofacial surgery was better in patients with the P253R mutation, whereas these patients showed a more pronounced severity of the syndactyly.

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Year:  2000        PMID: 10735635     DOI: 10.1034/j.1399-0004.2000.570208.x

Source DB:  PubMed          Journal:  Clin Genet        ISSN: 0009-9163            Impact factor:   4.438


  21 in total

1.  Comparison of ultrasound and magnetic resonance imaging in the prenatal diagnosis of Apert syndrome: report of a case.

Authors:  A Giancotti; V D'Ambrosio; A De Filippis; C Aliberti; G Pasquali; S Bernardo; L Manganaro
Journal:  Childs Nerv Syst       Date:  2014-02-25       Impact factor: 1.475

2.  Atypical presentation of a newborn with Apert syndrome.

Authors:  B Spruijt; B F M Rijken; K F M Joosten; H H Bredero-Boelhouwer; B Pullens; M H Lequin; E B Wolvius; M L C van Veelen-Vincent; I M J Mathijssen
Journal:  Childs Nerv Syst       Date:  2014-11-30       Impact factor: 1.475

Review 3.  Impact of genetics on the diagnosis and clinical management of syndromic craniosynostoses.

Authors:  Nneamaka B Agochukwu; Benjamin D Solomon; Maximilian Muenke
Journal:  Childs Nerv Syst       Date:  2012-08-08       Impact factor: 1.475

4.  Mouse models of Apert syndrome.

Authors:  Greg Holmes
Journal:  Childs Nerv Syst       Date:  2012-08-08       Impact factor: 1.475

Review 5.  Research advances in Apert syndrome.

Authors:  Satrupa Das; Anjana Munshi
Journal:  J Oral Biol Craniofac Res       Date:  2017-05-25

Review 6.  Craniosynostosis as a clinical and diagnostic problem: molecular pathology and genetic counseling.

Authors:  Anna Kutkowska-Kaźmierczak; Monika Gos; Ewa Obersztyn
Journal:  J Appl Genet       Date:  2018-02-01       Impact factor: 3.240

7.  Postnatal brain and skull growth in an Apert syndrome mouse model.

Authors:  Cheryl A Hill; Neus Martínez-Abadías; Susan M Motch; Jordan R Austin; Yingli Wang; Ethylin Wang Jabs; Joan T Richtsmeier; Kristina Aldridge
Journal:  Am J Med Genet A       Date:  2013-03-12       Impact factor: 2.802

8.  Activation of p38 MAPK pathway in the skull abnormalities of Apert syndrome Fgfr2(+P253R) mice.

Authors:  Yingli Wang; Miao Sun; Victoria L Uhlhorn; Xueyan Zhou; Inga Peter; Neus Martinez-Abadias; Cheryl A Hill; Christopher J Percival; Joan T Richtsmeier; David L Huso; Ethylin Wang Jabs
Journal:  BMC Dev Biol       Date:  2010-02-22       Impact factor: 1.978

9.  Quantification of facial skeletal shape variation in fibroblast growth factor receptor-related craniosynostosis syndromes.

Authors:  Yann Heuzé; Neus Martínez-Abadías; Jennifer M Stella; Eric Arnaud; Corinne Collet; Gemma García Fructuoso; Mariana Alamar; Lun-Jou Lo; Simeon A Boyadjiev; Federico Di Rocco; Joan T Richtsmeier
Journal:  Birth Defects Res A Clin Mol Teratol       Date:  2014-02-27

10.  Biochemical analysis of pathogenic ligand-dependent FGFR2 mutations suggests distinct pathophysiological mechanisms for craniofacial and limb abnormalities.

Authors:  Omar A Ibrahimi; Fuming Zhang; Anna V Eliseenkova; Nobuyuki Itoh; Robert J Linhardt; Moosa Mohammadi
Journal:  Hum Mol Genet       Date:  2004-07-28       Impact factor: 6.150

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