Literature DB >> 26668653

Novel keratin 5 mutation in a family with epidermolysis bullosa simplex.

Jiajia Gao1, Xuebin Wang1, Fang Zheng1, Sufang Dong1, Xueping Qiu1.   

Abstract

The aim of the present study was to identify the causative gene defects associated with epidermolysis bullosa simplex (EBS) in a pedigree. The diagnosis of EBS was confirmed in two patients from that pedigree based on the clinical manifestations, histopathological examination of the skin and family history. Blood samples were collected from 6 family members and 100 heathy controls, and genomic DNA and RNA were extracted. Mutation analysis of the keratin 5 gene (KRT5) was conducted using polymerase chain reaction (PCR) direct sequencing and PCR-restriction fragment length polymorphism. In the pedigree, the results of PCR direct sequencing revealed a heterozygous missense mutation in codon 202 of exon 2 of KRT5 (c.605T>A), which led to an amino acid change (p.L202Q) in the patients with EBS but was absent from the unaffected family members and 100 population controls. In conclusion, a novel missense mutation in the KRT5 gene was identified that had a pathogenic role in EBS in the population studied, which enriches the germline mutation spectrum of the KRT5 gene.

Entities:  

Keywords:  epidermolysis bullosa simplex; keratin 5; screening mutation

Year:  2015        PMID: 26668653      PMCID: PMC4665702          DOI: 10.3892/etm.2015.2811

Source DB:  PubMed          Journal:  Exp Ther Med        ISSN: 1792-0981            Impact factor:   2.447


  13 in total

1.  A novel keratin 5 mutation (K5V186L) in a family with EBS-K: a conservative substitution can lead to development of different disease phenotypes.

Authors:  M Liovic; J Stojan; P E Bowden; D Gibbs; A Vahlquist; E B Lane; R Komel
Journal:  J Invest Dermatol       Date:  2001-06       Impact factor: 8.551

2.  Modeling effects of mutations in coiled-coil structures: case study using epidermolysis bullosa simplex mutations in segment 1a of K5/K14 intermediate filaments.

Authors:  Thomasin A Smith; Peter M Steinert; David A D Parry
Journal:  Proteins       Date:  2004-06-01

Review 3.  Epidermolysis bullosa in Japan.

Authors:  Satoru Shinkuma; Ken Natsuga; Wataru Nishie; Hiroshi Shimizu
Journal:  Dermatol Clin       Date:  2010-04       Impact factor: 3.478

4.  The classification of inherited epidermolysis bullosa (EB): Report of the Third International Consensus Meeting on Diagnosis and Classification of EB.

Authors:  Jo-David Fine; Robin A J Eady; Eugene A Bauer; Johann W Bauer; Leena Bruckner-Tuderman; Adrian Heagerty; Helmut Hintner; Alain Hovnanian; Marcel F Jonkman; Irene Leigh; John A McGrath; Jemima E Mellerio; Dedee F Murrell; Hiroshi Shimizu; Jouni Uitto; Anders Vahlquist; David Woodley; Giovanna Zambruno
Journal:  J Am Acad Dermatol       Date:  2008-04-18       Impact factor: 11.527

5.  Epidermolysis bullosa simplex in Japanese and Korean patients: genetic studies in 19 cases.

Authors:  K Yasukawa; D Sawamura; M Goto; H Nakamura; S-Y Jung; S-C Kim; H Shimizu
Journal:  Br J Dermatol       Date:  2006-08       Impact factor: 9.302

6.  Epidermolysis bullosa simplex in Israel: clinical and genetic features.

Authors:  Dan Ciubotaru; Reuven Bergman; David Baty; Margarita Indelman; Ellen Pfendner; Danny Petronius; Hannah Moualem; Moien Kanaan; Danny Ben Amitai; W H Irwin McLean; Jouni Uitto; Eli Sprecher
Journal:  Arch Dermatol       Date:  2003-04

7.  Mutations in KRT5 and KRT14 cause epidermolysis bullosa simplex in 75% of the patients.

Authors:  M C Bolling; H H Lemmink; G H L Jansen; M F Jonkman
Journal:  Br J Dermatol       Date:  2011-02-17       Impact factor: 9.302

8.  The two-chain coiled-coil molecule of native epidermal keratin intermediate filaments is a type I-type II heterodimer.

Authors:  P M Steinert
Journal:  J Biol Chem       Date:  1990-05-25       Impact factor: 5.157

9.  The yin and the yang of keratin amino acid substitutions and epidermolysis bullosa simplex.

Authors:  Dedee F Murrell; Niken Trisnowati; Spiros Miyakis; Amy S Paller
Journal:  J Invest Dermatol       Date:  2011-09       Impact factor: 8.551

10.  Evaluation of different cytomegalovirus (CMV) DNA PCR protocols for analysis of dried blood spots from consecutive cases of neonates with congenital CMV infections.

Authors:  Oriane Soetens; Christelle Vauloup-Fellous; Ina Foulon; Pascal Dubreuil; Ben De Saeger; Liliane Grangeot-Keros; Anne Naessens
Journal:  J Clin Microbiol       Date:  2008-01-16       Impact factor: 5.948

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  1 in total

1.  A Novel Mutation p.L461P in KRT5 Causing Localized Epidermolysis Bullosa Simplex.

Authors:  Xin Jiang; Yingyu Zhu; Huihui Sun; Feng Gu
Journal:  Ann Dermatol       Date:  2020-12-30       Impact factor: 1.444

  1 in total

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