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Nibrin, a novel DNA double-strand break repair protein, is mutated in Nijmegen breakage syndrome.
- 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.
- Subjects :
- Amino Acid Sequence
Base Sequence
Chromosome Aberrations genetics
Chromosome Disorders
Chromosome Mapping
Chromosomes, Human, Pair 8 genetics
Cloning, Molecular methods
DNA Damage
DNA Repair
Female
Founder Effect
Humans
Linkage Disequilibrium
Male
Molecular Sequence Data
RNA, Messenger analysis
Sequence Analysis, DNA
Sequence Homology, Amino Acid
Syndrome
Cell Cycle Proteins genetics
Chromosome Breakage genetics
Genes, Recessive genetics
Microcephaly genetics
Nuclear Proteins
Sequence Deletion genetics
Subjects
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