Literature DB >> 7539673

Keratin 16 and keratin 17 mutations cause pachyonychia congenita.

W H McLean1, E L Rugg, D P Lunny, S M Morley, E B Lane, O Swensson, P J Dopping-Hepenstal, W A Griffiths, R A Eady, C Higgins.   

Abstract

Pachyonychia congenita (PC) is a group of autosomal dominant disorders characterized by dystrophic nails and other ectodermal aberrations. A gene for Jackson-Lawler PC was recently mapped to the type I keratin cluster on 17q. Here, we show that a heterozygous missense mutation in the helix initiation motif of K17 (Asn92Asp) co-segregates with the disease in this kindred. We also show that Jadassohn-Lewandowsky PC is caused by a heterozygous missense mutation in the helix initiation peptide of K16 (Leu130Pro). The known expression patterns of these keratins in epidermal structures correlates with the specific abnormalities observed in each form of PC.

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Year:  1995        PMID: 7539673     DOI: 10.1038/ng0395-273

Source DB:  PubMed          Journal:  Nat Genet        ISSN: 1061-4036            Impact factor:   38.330


  57 in total

1.  The gene for hypotrichosis of Marie Unna maps between D8S258 and D8S298: exclusion of the hr gene by cDNA and genomic sequencing.

Authors:  M van Steensel; F J Smith; P M Steijlen; I Kluijt; H P Stevens; A Messenger; H Kremer; M G Dunnill; C Kennedy; C S Munro; V R Doherty; J A McGrath; S P Covello; C M Coleman; J Uitto; W H McLean
Journal:  Am J Hum Genet       Date:  1999-08       Impact factor: 11.025

2.  Identification of a locus for type I punctate palmoplantar keratoderma on chromosome 15q22-q24.

Authors:  A Martinez-Mir; A Zlotogorski; D Londono; D Gordon; A Grunn; E Uribe; L Horev; I M Ruiz; N O Davalos; O Alayan; J Liu; T C Gilliam; J C Salas-Alanis; A M Christiano
Journal:  J Med Genet       Date:  2003-12       Impact factor: 6.318

3.  Novel mutations in the keratin-74 (KRT74) gene underlie autosomal dominant woolly hair/hypotrichosis in Pakistani families.

Authors:  Naveed Wasif; Syed Kamran ul-Hassan Naqvi; Sulman Basit; Nadir Ali; Muhammad Ansar; Wasim Ahmad
Journal:  Hum Genet       Date:  2010-12-28       Impact factor: 4.132

4.  Type I keratin 17 protein is phosphorylated on serine 44 by p90 ribosomal protein S6 kinase 1 (RSK1) in a growth- and stress-dependent fashion.

Authors:  Xiaoou Pan; Lesley A Kane; Jennifer E Van Eyk; Pierre A Coulombe
Journal:  J Biol Chem       Date:  2011-10-17       Impact factor: 5.157

5.  Localization of the gene causing keratolytic winter erythema to chromosome 8p22-p23, and evidence for a founder effect in South African Afrikaans-speakers.

Authors:  M Starfield; H C Hennies; M Jung; T Jenkins; T Wienker; P Hull; A Spurdle; W Küster; M Ramsay; A Reis
Journal:  Am J Hum Genet       Date:  1997-08       Impact factor: 11.025

6.  Altered keratinocyte differentiation is an early driver of keratin mutation-based palmoplantar keratoderma.

Authors:  Abigail G Zieman; Brian G Poll; Jingqun Ma; Pierre A Coulombe
Journal:  Hum Mol Genet       Date:  2019-07-01       Impact factor: 6.150

7.  The keratin 16 null phenotype is modestly impacted by genetic strain background in mice.

Authors:  Abigail Zieman; Pierre A Coulombe
Journal:  Exp Dermatol       Date:  2018-04-19       Impact factor: 3.960

Review 8.  "IF-pathies": a broad spectrum of intermediate filament-associated diseases.

Authors:  M Bishr Omary
Journal:  J Clin Invest       Date:  2009-07-01       Impact factor: 14.808

9.  Mutation of human keratin 18 in association with cryptogenic cirrhosis.

Authors:  N O Ku; T L Wright; N A Terrault; R Gish; M B Omary
Journal:  J Clin Invest       Date:  1997-01-01       Impact factor: 14.808

Review 10.  The molecular basis for inherited bullous diseases.

Authors:  B P Korge; T Krieg
Journal:  J Mol Med (Berl)       Date:  1996-02       Impact factor: 4.599

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