Literature DB >> 15827748

Novel keratin 5 and 14 gene mutations in patients with epidermolysis bullosa simplex from Poland.

Takahiro Hamada1, Yuko Kawano, Weronika Szczecinska, Katarzyna Wozniak, Shinichiro Yasumoto, Cezary Kowalewski, Takashi Hashimoto.   

Abstract

Mutation analysis in keratins 5/14 (K5/14) had been performed in five Polish families with epidermolysis bullosa simplex (EBS) to extend genotype-phenotype correlation and to add to the mutation database. All the patients had been clinically classified into two subtypes of EBS; Weber-Cockayne (EBS-WC) and Dowling-Meara (EBS-DM) as well as one case of EBS with mottled pigmentation (EBS-MP). DNA from patients and their family members was assessed for mutations in K5 or 14 using polymerase chain reaction amplification and subsequent direct sequencing. We identified four different missense mutations in K5 and one missense mutation in K14. Three of these are novel. Mutations associated EBS-DM resided in the highly conserved 20 amino acids end of the 1A domain in K5. Direct nucleotide sequencing of a case of EBS-MP revealed a heterozygous P25L mutation in K5. However, no genotype-phenotype correlation was identified in families with EBS-WC. The present study demonstrates the first series of molecular genetic data in EBS from Poland.

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Year:  2005        PMID: 15827748     DOI: 10.1007/s00403-005-0560-1

Source DB:  PubMed          Journal:  Arch Dermatol Res        ISSN: 0340-3696            Impact factor:   3.017


  2 in total

1.  Novel sporadic and recurrent mutations in KRT5 and KRT14 genes in Polish epidermolysis bullosa simplex patients: further insights into epidemiology and genotype-phenotype correlation.

Authors:  K Wertheim-Tysarowska; M Ołdak; A Giza; A Kutkowska-Kaźmierczak; J Sota; D Przybylska; K Woźniak; D Śniegórska; K Niepokój; A Sobczyńska-Tomaszewska; A M Rygiel; R Płoski; J Bal; C Kowalewski
Journal:  J Appl Genet       Date:  2015-10-02       Impact factor: 3.240

2.  Bullous diseases caused by KRT1 gene mutations: from epidermolytic hyperkeratosis to a novel variant of epidermolysis bullosa simplex.

Authors:  Katarzyna Osipowicz; Katarzyna Wertheim-Tysarowska; Bartłomiej Kwiek; Ewa Jankowska; Monika Gos; Agnieszka Charzewska; Katarzyna Woźniak; Cezary Kowalewski
Journal:  Postepy Dermatol Alergol       Date:  2020-09-02       Impact factor: 1.837

  2 in total

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