Literature DB >> 3339259

Assignment of three patients with xeroderma pigmentosum to complementation group E and their characteristics.

S Kondo1, S Fukuro, A Mamada, A Kawada, Y Satoh, Y Fujiwara.   

Abstract

Three cases belonging to xeroderma pigmentosum (XP) complementation group E were analyzed clinically and photobiologically. The three Japanese patients were a 50-yr-old female (XP80TO), a 42-yr-old female (XP81TO), and a 41-yr-old female (XP82TO). They were assigned to complementation group E by the cell hybridization study. All showed lowered minimal erythema doses between those of normal Japanese and XP group A subjects at wavelengths of 280, 290, and 300 nm of monochromatic ultraviolet (UV) light. Patients XP80TO and XP81TO, but not patient XP82TO, showed a delayed peak reaction at 48 h to UV erythema. All fibroblast strains from these patients had a reduced level of 40%-44% unscheduled DNA synthesis (UDS) after irradiation with 10 J/m2 of 254 nm UV. Primary cultured epidermal cells from these patients exhibited a relatively low level of UDS (ie, 38%-51% of normal epidermal cells). All of the group E fibroblast strains were twice as sensitive to 254 nm UV killing [n (extrapolation number) = 1.3-1.8, Do (mean lethal dose) = 2.2-2.8 J/m2)] as normal fibroblasts (n = 1.5, Do = 5.0 J/m2). All of the above group E patients had mild XP symptoms, but not neurological abnormalities, at the fifth decade of age. Patients XP80TO and XP81TO had developed skin malignancies (patients XP80TO developed three basaliomas; patient XP81TO developed two basaliomas) at the ages of 46 and 41 yr, respectively.

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Year:  1988        PMID: 3339259     DOI: 10.1111/1523-1747.ep12462130

Source DB:  PubMed          Journal:  J Invest Dermatol        ISSN: 0022-202X            Impact factor:   8.551


  8 in total

1.  No lack of complementation for unscheduled DNA synthesis between xeroderma pigmentosum complementation groups D and H.

Authors:  J H Robbins
Journal:  Hum Genet       Date:  1989-12       Impact factor: 4.132

2.  The xeroderma pigmentosum group E gene product DDB2 is a specific target of cullin 4A in mammalian cells.

Authors:  A Nag; T Bondar; S Shiv; P Raychaudhuri
Journal:  Mol Cell Biol       Date:  2001-10       Impact factor: 4.272

3.  The naturally occurring mutants of DDB are impaired in stimulating nuclear import of the p125 subunit and E2F1-activated transcription.

Authors:  P Shiyanov; S A Hayes; M Donepudi; A F Nichols; S Linn; B L Slagle; P Raychaudhuri
Journal:  Mol Cell Biol       Date:  1999-07       Impact factor: 4.272

Review 4.  Expanding molecular roles of UV-DDB: Shining light on genome stability and cancer.

Authors:  Maria Beecher; Namrata Kumar; Sunbok Jang; Vesna Rapić-Otrin; Bennett Van Houten
Journal:  DNA Repair (Amst)       Date:  2020-04-27

5.  A 127 kDa component of a UV-damaged DNA-binding complex, which is defective in some xeroderma pigmentosum group E patients, is homologous to a slime mold protein.

Authors:  M Takao; M Abramic; M Moos; V R Otrin; J C Wootton; M McLenigan; A S Levine; M Protic
Journal:  Nucleic Acids Res       Date:  1993-08-25       Impact factor: 16.971

6.  Evidence that xeroderma pigmentosum cells from complementation group E are deficient in a homolog of yeast photolyase.

Authors:  M Patterson; G Chu
Journal:  Mol Cell Biol       Date:  1989-11       Impact factor: 4.272

7.  Damage sensor role of UV-DDB during base excision repair.

Authors:  Sunbok Jang; Namrata Kumar; Emily C Beckwitt; Muwen Kong; Elise Fouquerel; Vesna Rapić-Otrin; Rajendra Prasad; Simon C Watkins; Cindy Khuu; Chandrima Majumdar; Sheila S David; Samuel H Wilson; Marcel P Bruchez; Patricia L Opresko; Bennett Van Houten
Journal:  Nat Struct Mol Biol       Date:  2019-07-22       Impact factor: 15.369

8.  Deep phenotyping of 89 xeroderma pigmentosum patients reveals unexpected heterogeneity dependent on the precise molecular defect.

Authors:  Hiva Fassihi; Mieran Sethi; Heather Fawcett; Jonathan Wing; Natalie Chandler; Shehla Mohammed; Emma Craythorne; Ana M S Morley; Rongxuan Lim; Sally Turner; Tanya Henshaw; Isabel Garrood; Paola Giunti; Tammy Hedderly; Adesoji Abiona; Harsha Naik; Gemma Harrop; David McGibbon; Nicolaas G J Jaspers; Elena Botta; Tiziana Nardo; Miria Stefanini; Antony R Young; Robert P E Sarkany; Alan R Lehmann
Journal:  Proc Natl Acad Sci U S A       Date:  2016-02-16       Impact factor: 11.205

  8 in total

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