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De novo nonsense mutations in ASXL1 cause Bohring-Opitz syndrome

Authors :
Hoischen, Alexander
van Bon, Bregje W M
Rodríguez-Santiago, Benjamín
Gilissen, Christian
Vissers, Lisenka E L M
de Vries, Petra
Janssen, Irene
van Lier, Bart
Hastings, Rob
Smithson, Sarah F
Newbury-Ecob, Ruth
Kjærgaard, Susanne
Goodship, Judith
McGowan, Ruth
Bartholdi, Deborah
Rauch, Anita
Peippo, Maarit
Cobben, Jan M
Wieczorek, Dagmar
Gillessen-Kaesbach, Gabriele
Veltman, Joris A
Brunner, Han G
de Vries, Bert B B A
Hoischen, Alexander
van Bon, Bregje W M
Rodríguez-Santiago, Benjamín
Gilissen, Christian
Vissers, Lisenka E L M
de Vries, Petra
Janssen, Irene
van Lier, Bart
Hastings, Rob
Smithson, Sarah F
Newbury-Ecob, Ruth
Kjærgaard, Susanne
Goodship, Judith
McGowan, Ruth
Bartholdi, Deborah
Rauch, Anita
Peippo, Maarit
Cobben, Jan M
Wieczorek, Dagmar
Gillessen-Kaesbach, Gabriele
Veltman, Joris A
Brunner, Han G
de Vries, Bert B B A
Source :
Hoischen , A , van Bon , B W M , Rodríguez-Santiago , B , Gilissen , C , Vissers , L E L M , de Vries , P , Janssen , I , van Lier , B , Hastings , R , Smithson , S F , Newbury-Ecob , R , Kjærgaard , S , Goodship , J , McGowan , R , Bartholdi , D , Rauch , A , Peippo , M , Cobben , J M , Wieczorek , D , Gillessen-Kaesbach , G , Veltman , J A , Brunner , H G & de Vries , B B B A 2011 , ' De novo nonsense mutations in ASXL1 cause Bohring-Opitz syndrome ' , Nature Genetics , vol. 43 , no. 8 , pp. 729-31 .
Publication Year :
2011

Abstract

Bohring-Opitz syndrome is characterized by severe intellectual disability, distinctive facial features and multiple congenital malformations. We sequenced the exomes of three individuals with Bohring-Opitz syndrome and in each identified heterozygous de novo nonsense mutations in ASXL1, which is required for maintenance of both activation and silencing of Hox genes. In total, 7 out of 13 subjects with a Bohring-Opitz phenotype had de novo ASXL1 mutations, suggesting that the syndrome is genetically heterogeneous.

Details

Database :
OAIster
Journal :
Hoischen , A , van Bon , B W M , Rodríguez-Santiago , B , Gilissen , C , Vissers , L E L M , de Vries , P , Janssen , I , van Lier , B , Hastings , R , Smithson , S F , Newbury-Ecob , R , Kjærgaard , S , Goodship , J , McGowan , R , Bartholdi , D , Rauch , A , Peippo , M , Cobben , J M , Wieczorek , D , Gillessen-Kaesbach , G , Veltman , J A , Brunner , H G & de Vries , B B B A 2011 , ' De novo nonsense mutations in ASXL1 cause Bohring-Opitz syndrome ' , Nature Genetics , vol. 43 , no. 8 , pp. 729-31 .
Notes :
English
Publication Type :
Electronic Resource
Accession number :
edsoai.ocn826493676
Document Type :
Electronic Resource