Literature DB >> 9590180

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

R Varon1, C Vissinga, M Platzer, K M Cerosaletti, K H Chrzanowska, K Saar, G Beckmann, E Seemanová, P R Cooper, N J Nowak, M Stumm, C M Weemaes, R A Gatti, R K Wilson, M Digweed, A Rosenthal, K Sperling, P Concannon, A Reis.   

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.

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Year:  1998        PMID: 9590180     DOI: 10.1016/s0092-8674(00)81174-5

Source DB:  PubMed          Journal:  Cell        ISSN: 0092-8674            Impact factor:   41.582


  262 in total

Review 1.  The pathogenesis of ataxia-telangiectasia. Learning from a Rosetta Stone.

Authors:  R A Gatti; S Becker-Catania; H H Chun; X Sun; M Mitui; C H Lai; N Khanlou; M Babaei; R Cheng; C Clark; Y Huo; N C Udar; R K Iyer
Journal:  Clin Rev Allergy Immunol       Date:  2001-02       Impact factor: 8.667

2.  A mechanistic basis for Mre11-directed DNA joining at microhomologies.

Authors:  T T Paull; M Gellert
Journal:  Proc Natl Acad Sci U S A       Date:  2000-06-06       Impact factor: 11.205

Review 3.  Reverse genetic studies of homologous DNA recombination using the chicken B-lymphocyte line, DT40.

Authors:  E Sonoda; C Morrison; Y M Yamashita; M Takata; S Takeda
Journal:  Philos Trans R Soc Lond B Biol Sci       Date:  2001-01-29       Impact factor: 6.237

Review 4.  The mammalian Mre11-Rad50-nbs1 protein complex: integration of functions in the cellular DNA-damage response.

Authors:  J H Petrini
Journal:  Am J Hum Genet       Date:  1999-05       Impact factor: 11.025

Review 5.  Immunodeficiency associated with DNA repair defects.

Authors:  A R Gennery; A J Cant; P A Jeggo
Journal:  Clin Exp Immunol       Date:  2000-07       Impact factor: 4.330

6.  The MRE11-NBS1-RAD50 pathway is perturbed in SV40 large T antigen-immortalized AT-1, AT-2 and HL-1 cardiomyocytes.

Authors:  N A Lanson; D B Egeland; B A Royals; W C Claycomb
Journal:  Nucleic Acids Res       Date:  2000-08-01       Impact factor: 16.971

7.  Reconstitution of the mammalian DNA double-strand break end-joining reaction reveals a requirement for an Mre11/Rad50/NBS1-containing fraction.

Authors:  Juren Huang; William S Dynan
Journal:  Nucleic Acids Res       Date:  2002-02-01       Impact factor: 16.971

Review 8.  Manipulating the mammalian genome by homologous recombination.

Authors:  K M Vasquez; K Marburger; Z Intody; J H Wilson
Journal:  Proc Natl Acad Sci U S A       Date:  2001-07-17       Impact factor: 11.205

9.  Suppression of genome instability by redundant S-phase checkpoint pathways in Saccharomyces cerevisiae.

Authors:  Kyungjae Myung; Richard D Kolodner
Journal:  Proc Natl Acad Sci U S A       Date:  2002-03-26       Impact factor: 11.205

10.  Mre11 complex and DNA replication: linkage to E2F and sites of DNA synthesis.

Authors:  R S Maser; O K Mirzoeva; J Wells; H Olivares; B R Williams; R A Zinkel; P J Farnham; J H Petrini
Journal:  Mol Cell Biol       Date:  2001-09       Impact factor: 4.272

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