Literature DB >> 3341805

No apparent neurologic defect in a patient with xeroderma pigmentosum complementation group D.

M Ichihashi1, K Yamamura, T Hiramoto, Y Fujiwara.   

Abstract

A 31-year-old female patient with xeroderma pigmentosum (XP) XP43KO was assigned to complementation group D by the cell-fusion complementation methods. Cultured XP43KO cells from our patient had the defective DNA repair phenotype showing a residual level of ultraviolet (UV)-induced unscheduled DNA synthesis (45% of normal) and an eightfold higher sensitivity to 254-nm UV killing, compared with normal cells. The phototest on the patient revealed the delayed maximum reaction to UV-B-induced erythema and lower minimal erythema doses at 290- and 300-nm monochromatic wavelengths. However, the XP43KO patient showed no apparent neurologic abnormalities and rather mild or moderate skin lesions at the age of 31 years, although DNA repair deficiency in XP43KO cells from our patient fell into the range of group D cells.

Entities:  

Mesh:

Year:  1988        PMID: 3341805

Source DB:  PubMed          Journal:  Arch Dermatol        ISSN: 0003-987X


  5 in total

1.  Xeroderma pigmentosum complementation group H is withdrawn and reassigned to group D.

Authors:  J H Robbins
Journal:  Hum Genet       Date:  1991-12       Impact factor: 4.132

2.  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

3.  Xeroderma pigmentosum and trichothiodystrophy are associated with different mutations in the XPD (ERCC2) repair/transcription gene.

Authors:  E M Taylor; B C Broughton; E Botta; M Stefanini; A Sarasin; N G Jaspers; H Fawcett; S A Harcourt; C F Arlett; A R Lehmann
Journal:  Proc Natl Acad Sci U S A       Date:  1997-08-05       Impact factor: 11.205

4.  Phenotype-specific adverse effects of XPD mutations on human prenatal development implicate impairment of TFIIH-mediated functions in placenta.

Authors:  Roxana Moslehi; Anil Kumar; James L Mills; Xavier Ambroggio; Caroline Signore; Amiran Dzutsev
Journal:  Eur J Hum Genet       Date:  2012-01-11       Impact factor: 4.246

5.  A Drosophila XPD model links cell cycle coordination with neuro-development and suggests links to cancer.

Authors:  Karin Stettler; Xiaoming Li; Björn Sandrock; Sophie Braga-Lagache; Manfred Heller; Lutz Dümbgen; Beat Suter
Journal:  Dis Model Mech       Date:  2014-11-27       Impact factor: 5.758

  5 in total

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