Literature DB >> 26600631

Molecular analysis of exons 8, 9 and 10 of the fibroblast growth factor receptor 2 (FGFR2) gene in two families with index cases of Apert Syndrome.

Lilian Torres1, Gualberto Hernández1, Alejandro Barrera1, Sandra Ospina2, Rolando Prada3.   

Abstract

INTRODUCTION: Apert syndrome (AS) is a craniosynostosis condition caused by mutations in the Fibroblast Growth Factor Receptor 2 (FGFR2) gene. Clinical features include cutaneous and osseous symmetric syndactily in hands and feet, with variable presentations in bones, brain, skin and other internal organs.
METHODS: Members of two families with an index case of Apert Syndrome were assessed to describe relevant clinical features and molecular analysis (sequencing and amplification) of exons 8, 9 and 10 of FGFR2 gen.
RESULTS: Family 1 consists of the mother, the index case and half -brother who has a cleft lip and palate. In this family we found a single FGFR2 mutation, S252W, in the sequence of exon 8. Although mutations were not found in the study of the patient affected with cleft lip and palate, it is known that these diseases share signaling pathways, allowing suspected alterations in shared genes. In the patient of family 2, we found a sequence variant T78.501A located near the splicing site, which could interfere in this process, and consequently with the protein function.

Entities:  

Keywords:  Apert syndrome; FGFR2 gene; cleft palate; intron; mutation

Mesh:

Substances:

Year:  2015        PMID: 26600631      PMCID: PMC4640438     

Source DB:  PubMed          Journal:  Colomb Med (Cali)        ISSN: 0120-8322


  7 in total

1.  A splicing switch and gain-of-function mutation in FgfR2-IIIc hemizygotes causes Apert/Pfeiffer-syndrome-like phenotypes.

Authors:  M K Hajihosseini; S Wilson; L De Moerlooze; C Dickson
Journal:  Proc Natl Acad Sci U S A       Date:  2001-03-27       Impact factor: 11.205

2.  Differential effects of FGFR2 mutations on syndactyly and cleft palate in Apert syndrome.

Authors:  S F Slaney; M Oldridge; J A Hurst; G M Moriss-Kay; C M Hall; M D Poole; A O Wilkie
Journal:  Am J Hum Genet       Date:  1996-05       Impact factor: 11.025

3.  Impaired FGF signaling contributes to cleft lip and palate.

Authors:  Bridget M Riley; M Adela Mansilla; Jinghong Ma; Sandra Daack-Hirsch; Brion S Maher; Lisa M Raffensperger; Erilynn T Russo; Alexandre R Vieira; Catherine Dodé; Moosa Mohammadi; Mary L Marazita; Jeffrey C Murray
Journal:  Proc Natl Acad Sci U S A       Date:  2007-03-06       Impact factor: 11.205

4.  Structural basis for fibroblast growth factor receptor 2 activation in Apert syndrome.

Authors:  O A Ibrahimi; A V Eliseenkova; A N Plotnikov; K Yu; D M Ornitz; M Mohammadi
Journal:  Proc Natl Acad Sci U S A       Date:  2001-06-05       Impact factor: 11.205

Review 5.  Development of the lip and palate: FGF signalling.

Authors:  Philip Stanier; Erwin Pauws
Journal:  Front Oral Biol       Date:  2012-06-25

6.  Apert syndrome results from localized mutations of FGFR2 and is allelic with Crouzon syndrome.

Authors:  A O Wilkie; S F Slaney; M Oldridge; M D Poole; G J Ashworth; A D Hockley; R D Hayward; D J David; L J Pulleyn; P Rutland
Journal:  Nat Genet       Date:  1995-02       Impact factor: 38.330

Review 7.  Signals and switches in Mammalian neural crest cell differentiation.

Authors:  Shachi Bhatt; Raul Diaz; Paul A Trainor
Journal:  Cold Spring Harb Perspect Biol       Date:  2013-02-01       Impact factor: 10.005

  7 in total
  5 in total

1.  Apert Syndrome: Dental management considerations and objectives.

Authors:  Line Droubi; Mohannad Laflouf; Yasser Alsayed Tolibah; John C Comisi
Journal:  J Oral Biol Craniofac Res       Date:  2022-04-28

Review 2.  Genetic determinants of syndactyly: perspectives on pathogenesis and diagnosis.

Authors:  Afraah Cassim; Dineshani Hettiarachchi; Vajira H W Dissanayake
Journal:  Orphanet J Rare Dis       Date:  2022-05-12       Impact factor: 4.303

Review 3.  Cleft Palate in Apert Syndrome.

Authors:  Delayna Willie; Greg Holmes; Ethylin Wang Jabs; Meng Wu
Journal:  J Dev Biol       Date:  2022-08-11

Review 4.  Dental approach for Apert syndrome in children: a systematic review.

Authors:  A-S López-Estudillo; M-A Rosales-Bérber; S Ruiz-Rodríguez; A Pozos-Guillén; M-Á Noyola-Frías; A Garrocho-Rangel
Journal:  Med Oral Patol Oral Cir Bucal       Date:  2017-11-01

5.  Apert Syndrome With FGFR2 758 C > G Mutation: A Chinese Case Report.

Authors:  Yahong Li; Dingyuan Ma; Yun Sun; Lulu Meng; Yanyun Wang; Tao Jiang
Journal:  Front Genet       Date:  2018-05-17       Impact factor: 4.599

  5 in total

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