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      Keratin 9 gene mutations in epidermolytic palmoplantar keratoderma (EPPK).

      Nature genetics
      Amino Acid Sequence, Base Sequence, Chromosomes, Human, Pair 17, Cloning, Molecular, DNA Mutational Analysis, DNA, Satellite, analysis, Female, Haplotypes, Humans, In Situ Hybridization, Fluorescence, Introns, genetics, Keratins, chemistry, Keratoderma, Palmoplantar, Male, Molecular Sequence Data, Pedigree, Point Mutation, Polymorphism, Genetic, Protein Structure, Secondary, Restriction Mapping

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          Abstract

          We have isolated the gene for human type I keratin 9 (KRT9) and localised it to chromosome 17q21. Patients with epidermolytic palmoplantar keratoderma (EPPK), an autosomal dominant skin disease, were investigated. Three KRT9 mutations, N160K, R162Q, and R162W, were identified. All the mutations are in the highly conserved coil 1A of the rod domain, thought to be important for heterodimerisation. R162W was detected in five unrelated families and affects the corresponding residue in the keratin 14 and keratin 10 genes that is also altered in cases of epidermolysis bullosa simplex and generalised epidermolytic hyperkeratosis, respectively. These findings provide further evidence that mutations in keratin genes may cause epidermolysis and hyperkeratosis and that hyperkeratosis of palms and soles may be caused by different mutations in the KRT9 gene.

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            Rapid and sensitive detection of point mutations and DNA polymorphisms using the polymerase chain reaction

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              Point mutations in human keratin 14 genes of epidermolysis bullosa simplex patients: genetic and functional analyses.

              Previously we demonstrated that transgenic mice expressing mutant basal epidermal keratin genes exhibited a phenotype resembling a group of autosomal dominant human skin disorders known as epidermolysis bullosa simplex (EBS). EBS diseases affect approximately 1: 50,000 and are of unknown etiology, although all subtypes exhibit blistering arising from basal cell cytolysis. We now demonstrate that two patients with spontaneous cases of Dowling-Meara EBS have point mutations in a critical region in one (K14) of two basal keratin genes. To demonstrate function, we engineered one of these point mutations in a cloned human K14 cDNA, and showed that a K14 with an Arg-125----Cys mutation disrupted keratin network formation in transfected keratinocytes and perturbed filament assembly in vitro. Since we had previously shown that keratin network perturbation is an essential component of EBS diseases, these data suggest that the basis for the phenotype in this patient resides in this point mutation.
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