Literature DB >> 25262888

Xeroderma pigmentosum: diagnostic procedures, interdisciplinary patient care, and novel therapeutic approaches.

Janin Lehmann1, Steffen Schubert, Steffen Emmert.   

Abstract

Xeroderma pigmentosum (XP) is an autosomal recessive disease, caused by a gene defect in the nucleotide-excision-repair (NER) pathway or in translesional DNA synthesis. At the age of eight, patients already develop their first skin cancers due to this DNA repair defect. In contrast, in the Caucasian population the first tumor formation in UV exposed skin regions occurs at a mean age of 60. The clinical picture among patients suffering from XP is highly diverse and includes signs of accelerated skin aging, and UV-induced skin cancers, as well as ophthalmologic and neurological symptoms. Patients should therefore receive interdisciplinary care. This includes dermatologists, ophthalmologists, ENT specialists, neurologists, and human geneticists. Patients with XP are clinically diagnosed, but this may be supported by molecular-genetic and functional analyses. These analyses allow pinpointing the exact disease-causing gene defect (complementation group assignment, detection of the type and location of the mutation within the gene). The resulting information is already relevant to predict the course of disease and symptoms and probably will be utilized for individualized therapeutic approaches in the future. Recently, enhanced repair of UV photolesions in xeroderma pigmentosum group C cells induced by translational readthrough of premature termination codons by certain antibiotics could be demonstrated.
© 2014 Deutsche Dermatologische Gesellschaft (DDG). Published by John Wiley & Sons Ltd.

Entities:  

Mesh:

Year:  2014        PMID: 25262888     DOI: 10.1111/ddg.12419

Source DB:  PubMed          Journal:  J Dtsch Dermatol Ges        ISSN: 1610-0379            Impact factor:   5.584


  10 in total

Review 1.  [Practical aspects of molecular diagnostics in genodermatoses].

Authors:  C Has; Y He
Journal:  Hautarzt       Date:  2016-01       Impact factor: 0.751

Review 2.  [Molecular genetic investigations as the basis for targeted treatment of basal cell carcinoma of the eye].

Authors:  L Boeckmann; M C Martens; V Kakkassery; L M Heindl; S Emmert
Journal:  Ophthalmologe       Date:  2020-02       Impact factor: 1.059

3.  XPF knockout via CRISPR/Cas9 reveals that ERCC1 is retained in the cytoplasm without its heterodimer partner XPF.

Authors:  Janin Lehmann; Christina Seebode; Sabine Smolorz; Steffen Schubert; Steffen Emmert
Journal:  Cell Mol Life Sci       Date:  2017-01-27       Impact factor: 9.261

Review 4.  Xeroderma Pigmentosum: Gene Variants and Splice Variants.

Authors:  Marie Christine Martens; Steffen Emmert; Lars Boeckmann
Journal:  Genes (Basel)       Date:  2021-07-29       Impact factor: 4.096

Review 5.  Xeroderma pigmentosum: an updated review.

Authors:  Alexander Kc Leung; Benjamin Barankin; Joseph M Lam; Kin Fon Leong; Kam Lun Hon
Journal:  Drugs Context       Date:  2022-04-25

6.  Identification and Functional Testing of ERCC2 Mutations in a Multi-national Cohort of Patients with Familial Breast- and Ovarian Cancer.

Authors:  Andreas Rump; Anna Benet-Pages; Steffen Schubert; Jan Dominik Kuhlmann; Ramūnas Janavičius; Eva Macháčková; Lenka Foretová; Zdenek Kleibl; Filip Lhota; Petra Zemankova; Elitza Betcheva-Krajcir; Luisa Mackenroth; Karl Hackmann; Janin Lehmann; Anke Nissen; Nataliya DiDonato; Romy Opitz; Holger Thiele; Karin Kast; Pauline Wimberger; Elke Holinski-Feder; Steffen Emmert; Evelin Schröck; Barbara Klink
Journal:  PLoS Genet       Date:  2016-08-09       Impact factor: 5.917

7.  Case report: Identification of a novel heterozygous germline ERCC2 mutation in a patient with dermatofibrosarcoma protuberans.

Authors:  Qing Zhang; Yongzhi Ju; Xia You; Tingting Sun; Yi Ding
Journal:  Front Oncol       Date:  2022-08-10       Impact factor: 5.738

Review 8.  RNA-based therapies for genodermatoses.

Authors:  Olivier Bornert; Patricia Peking; Jeroen Bremer; Ulrich Koller; Peter C van den Akker; Annemieke Aartsma-Rus; Anna M G Pasmooij; Eva M Murauer; Alexander Nyström
Journal:  Exp Dermatol       Date:  2017-01       Impact factor: 3.960

9.  Splice variants of the endonucleases XPF and XPG contain residual DNA repair capabilities and could be a valuable tool for personalized medicine.

Authors:  Janin Lehmann; Steffen Schubert; Christina Seebode; Antje Apel; Andreas Ohlenbusch; Steffen Emmert
Journal:  Oncotarget       Date:  2017-12-08

Review 10.  Context-Dependent Strategies for Enhanced Genome Editing of Genodermatoses.

Authors:  Oliver Patrick March; Thomas Kocher; Ulrich Koller
Journal:  Cells       Date:  2020-01-02       Impact factor: 6.600

  10 in total

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