Literature DB >> 8317483

Xeroderma pigmentosum complementation group G associated with Cockayne syndrome.

W Vermeulen1, J Jaeken, N G Jaspers, D Bootsma, J H Hoeijmakers.   

Abstract

Xeroderma pigmentosum (XP) and Cockayne syndrome (CS) are two rare inherited disorders with a clinical and cellular hypersensitivity to the UV component of the sunlight spectrum. Although the two traits are generally considered as clinically and genetically distinct entities, on the biochemical level a defect in the nucleotide excision-repair (NER) pathway is involved in both. Classical CS patients are primarily deficient in the preferential repair of DNA damage in actively transcribed genes, whereas in most XP patients the genetic defect affects both "preferential" and "overall" NER modalities. Here we report a genetic study of two unrelated, severely affected patients with the clinical characteristics of CS but with a biochemical defect typical of XP. By complementation analysis, using somatic cell fusion and nuclear microinjection of cloned repair genes, we assign these two patients to XP complementation group G, which previously was not associated with CS. This observation extends the earlier identification of two patients with a rare combined XP/CS phenotype within XP complementation groups B and D, respectively. It indicates that some mutations in at least three of the seven genes known to be involved in XP also can result in a picture of partial or even full-blown CS. We conclude that the syndromes XP and CS are biochemically closely related and may be part of a broader clinical disease spectrum. We suggest, as a possible molecular mechanism underlying this relation, that the XPGC repair gene has an additional vital function, as shown for some other NER genes.

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Year:  1993        PMID: 8317483      PMCID: PMC1682247     

Source DB:  PubMed          Journal:  Am J Hum Genet        ISSN: 0002-9297            Impact factor:   11.025


  29 in total

1.  Studies on a new case of xeroderma pigmentosum (XP3BR) from complementation group G with cellular sensitivity to ionizing radiation.

Authors:  C F Arlett; S A Harcourt; A R Lehmann; S Stevens; M A Ferguson-Smith; W N Morley
Journal:  Carcinogenesis       Date:  1980-09       Impact factor: 4.944

2.  Lack of complementation between xeroderma pigmentosum complementation groups D and H.

Authors:  R T Johnson; G C Elliott; S Squires; V C Joysey
Journal:  Hum Genet       Date:  1989-02       Impact factor: 4.132

3.  Xeroderma pigmentosum. Defective DNA repair causes skin cancer and neurodegeneration.

Authors:  J H Robbins
Journal:  JAMA       Date:  1988-07-15       Impact factor: 56.272

4.  Xeroderma pigmentosum. An inherited diseases with sun sensitivity, multiple cutaneous neoplasms, and abnormal DNA repair.

Authors:  J H Robbins; K H Kraemer; M A Lutzner; B W Festoff; H G Coon
Journal:  Ann Intern Med       Date:  1974-02       Impact factor: 25.391

5.  Defective repair replication of DNA in xeroderma pigmentosum.

Authors:  J E Cleaver
Journal:  Nature       Date:  1968-05-18       Impact factor: 49.962

6.  The cloned human DNA excision repair gene ERCC-1 fails to correct xeroderma pigmentosum complementation groups A through I.

Authors:  M van Duin; G Vredeveldt; L V Mayne; H Odijk; W Vermeulen; B Klein; G Weeda; J H Hoeijmakers; D Bootsma; A Westerveld
Journal:  Mutat Res       Date:  1989-03       Impact factor: 2.433

7.  Clinical and biochemical studies in three patients with severe early infantile Cockayne syndrome.

Authors:  J Jaeken; H Klocker; H Schwaiger; R Bellmann; M Hirsch-Kauffmann; M Schweiger
Journal:  Hum Genet       Date:  1989-11       Impact factor: 4.132

8.  [Photosensitization and DNA repair. Possible nosologic relationship between Xeroderma pigmentosum and Cockayne's syndrome].

Authors:  D Lafforet; J M Dupuy
Journal:  Arch Fr Pediatr       Date:  1978-12

9.  Transient correction of excision repair defects in fibroblasts of 9 xeroderma pigmentosum complementation groups by microinjection of crude human cell extracts.

Authors:  W Vermeulen; P Osseweijer; A J de Jonge; J H Hoeijmakers
Journal:  Mutat Res       Date:  1986-05       Impact factor: 2.433

Review 10.  Cockayne syndrome: review of 140 cases.

Authors:  M A Nance; S A Berry
Journal:  Am J Med Genet       Date:  1992-01-01
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  33 in total

Review 1.  Transcription-coupled repair of DNA damage: unanticipated players, unexpected complexities.

Authors:  S A Leadon
Journal:  Am J Hum Genet       Date:  1999-05       Impact factor: 11.025

2.  Reduced RNA polymerase II transcription in extracts of cockayne syndrome and xeroderma pigmentosum/Cockayne syndrome cells.

Authors:  G L Dianov; J F Houle; N Iyer; V A Bohr; E C Friedberg
Journal:  Nucleic Acids Res       Date:  1997-09-15       Impact factor: 16.971

3.  Cerebro-oculo-facio-skeletal syndrome with a nucleotide excision-repair defect and a mutated XPD gene, with prenatal diagnosis in a triplet pregnancy.

Authors:  J M Graham ; K Anyane-Yeboa; A Raams; E Appeldoorn; W J Kleijer; V H Garritsen; D Busch; T G Edersheim; N G Jaspers
Journal:  Am J Hum Genet       Date:  2001-07-03       Impact factor: 11.025

Review 4.  Cockayne syndrome: defective repair of transcription?

Authors:  A J van Gool; G T van der Horst; E Citterio; J H Hoeijmakers
Journal:  EMBO J       Date:  1997-07-16       Impact factor: 11.598

5.  Definition of a short region of XPG necessary for TFIIH interaction and stable recruitment to sites of UV damage.

Authors:  Fabrizio Thorel; Angelos Constantinou; Isabelle Dunand-Sauthier; Thierry Nouspikel; Philippe Lalle; Anja Raams; Nicolaas G J Jaspers; Wim Vermeulen; Mahmud K K Shivji; Richard D Wood; Stuart G Clarkson
Journal:  Mol Cell Biol       Date:  2004-12       Impact factor: 4.272

6.  Identification of the XPG region that causes the onset of Cockayne syndrome by using Xpg mutant mice generated by the cDNA-mediated knock-in method.

Authors:  Naoko Shiomi; Seiji Kito; Masaki Oyama; Tsukasa Matsunaga; Yoshi-Nobu Harada; Masahito Ikawa; Masaru Okabe; Tadahiro Shiomi
Journal:  Mol Cell Biol       Date:  2004-05       Impact factor: 4.272

7.  Xeroderma pigmentosum--Cockayne syndrome complex: a further case.

Authors:  B C Hamel; A Raams; A R Schuitema-Dijkstra; P Simons; I van der Burgt; N G Jaspers; W J Kleijer
Journal:  J Med Genet       Date:  1996-07       Impact factor: 6.318

8.  Clinical heterogeneity within xeroderma pigmentosum associated with mutations in the DNA repair and transcription gene ERCC3.

Authors:  W Vermeulen; R J Scott; S Rodgers; H J Müller; J Cole; C F Arlett; W J Kleijer; D Bootsma; J H Hoeijmakers; G Weeda
Journal:  Am J Hum Genet       Date:  1994-02       Impact factor: 11.025

9.  Yeast nucleotide excision repair proteins Rad2 and Rad4 interact with RNA polymerase II basal transcription factor b (TFIIH).

Authors:  A J Bardwell; L Bardwell; N Iyer; J Q Svejstrup; W J Feaver; R D Kornberg; E C Friedberg
Journal:  Mol Cell Biol       Date:  1994-06       Impact factor: 4.272

Review 10.  XPG: its products and biological roles.

Authors:  Orlando D Schärer
Journal:  Adv Exp Med Biol       Date:  2008       Impact factor: 2.622

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