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Nibrin, a novel DNA double-strand break repair protein, is mutated in Nijmegen breakage syndrome.

Authors :
Varon R
Vissinga C
Platzer M
Cerosaletti KM
Chrzanowska KH
Saar K
Beckmann G
Seemanová E
Cooper PR
Nowak NJ
Stumm M
Weemaes CM
Gatti RA
Wilson RK
Digweed M
Rosenthal A
Sperling K
Concannon P
Reis A
Source :
Cell [Cell] 1998 May 01; Vol. 93 (3), pp. 467-76.
Publication Year :
1998

Abstract

Nijmegen breakage syndrome (NBS) is an autosomal recessive chromosomal instability syndrome characterized by microcephaly, growth retardation, immunodeficiency, and cancer predisposition. Cells from NBS patients are hypersensitive to ionizing radiation with cytogenetic features indistinguishable from ataxia telangiectasia. We describe the positional cloning of a gene encoding a novel protein, nibrin. It contains two modules found in cell cycle checkpoint proteins, a forkhead-associated domain adjacent to a breast cancer carboxy-terminal domain. A truncating 5 bp deletion was identified in the majority of NBS patients, carrying a conserved marker haplotype. Five further truncating mutations were identified in patients with other distinct haplotypes. The domains found in nibrin and the NBS phenotype suggest that this disorder is caused by defective responses to DNA double-strand breaks.

Details

Language :
English
ISSN :
0092-8674
Volume :
93
Issue :
3
Database :
MEDLINE
Journal :
Cell
Publication Type :
Academic Journal
Accession number :
9590180
Full Text :
https://doi.org/10.1016/s0092-8674(00)81174-5