Literature DB >> 10581485

Novel PRNP sequence variant associated with familial encephalopathy.

L Cervenáková1, C Buetefisch, H S Lee, I Taller, G Stone, C J Gibbs, P Brown, M Hallett, L G Goldfarb.   

Abstract

Human transmissible spongiform encephalopathies (TSEs) are a group of chronic progressive neurodegenerative disorders that may be hereditary, infectious, or sporadic. Hereditary TSEs are associated with mutations in the PRNP gene on chromosome 20p12-pter. We report on a family in which seven patients developed limb and truncal ataxia, dysarthria, myoclonic jerks, and cognitive decline. The age of onset in the 30s, 40s, or 50s, prolonged disease duration, cerebellar atrophy on imaging, and the presence of synchronic periodic discharges on electroencephalogram suggested a familial encephalopathy resembling Gerstmann-Sträussler-Scheinker disease. A novel H187R mutation has been identified in affected, but not in unaffected, family members or unrelated controls suggesting a pathogenic role for this mutation. Am. J. Med. Genet. (Neuropsychiatr. Genet.) 88:653-656, 1999. Published 1999 Wiley-Liss, Inc.

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Year:  1999        PMID: 10581485     DOI: 10.1002/(sici)1096-8628(19991215)88:6<653::aid-ajmg14>3.0.co;2-e

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  13 in total

Review 1.  Genetic PrP Prion Diseases.

Authors:  Mee-Ohk Kim; Leonel T Takada; Katherine Wong; Sven A Forner; Michael D Geschwind
Journal:  Cold Spring Harb Perspect Biol       Date:  2018-05-01       Impact factor: 10.005

2.  Influence of pH on the human prion protein: insights into the early steps of misfolding.

Authors:  Marc W van der Kamp; Valerie Daggett
Journal:  Biophys J       Date:  2010-10-06       Impact factor: 4.033

3.  Structural and dynamic properties of the human prion protein.

Authors:  Wei Chen; Marc W van der Kamp; Valerie Daggett
Journal:  Biophys J       Date:  2014-03-04       Impact factor: 4.033

4.  Stability and Cu(II) binding of prion protein variants related to inherited human prion diseases.

Authors:  Grazia M Cereghetti; Arthur Schweiger; Rudi Glockshuber; Sabine Van Doorslaer
Journal:  Biophys J       Date:  2003-03       Impact factor: 4.033

Review 5.  Genetic prion disease: Experience of a rapidly progressive dementia center in the United States and a review of the literature.

Authors:  Leonel T Takada; Mee-Ohk Kim; Ross W Cleveland; Katherine Wong; Sven A Forner; Ignacio Illán Gala; Jamie C Fong; Michael D Geschwind
Journal:  Am J Med Genet B Neuropsychiatr Genet       Date:  2017-01       Impact factor: 3.568

6.  Systematic investigation of predicted effect of nonsynonymous SNPs in human prion protein gene: a molecular modeling and molecular dynamics study.

Authors:  Samad Jahandideh; Degui Zhi
Journal:  J Biomol Struct Dyn       Date:  2013-03-25

Review 7.  Copper binding extrinsic to the octarepeat region in the prion protein.

Authors:  Eric D Walter; Dan J Stevens; Ann R Spevacek; Micah P Visconte; Andrew Dei Rossi; Glenn L Millhauser
Journal:  Curr Protein Pept Sci       Date:  2009-10       Impact factor: 3.272

8.  Checking the pH-induced conformational transition of prion protein by molecular dynamics simulations: effect of protonation of histidine residues.

Authors:  Emma Langella; Roberto Improta; Vincenzo Barone
Journal:  Biophys J       Date:  2004-09-17       Impact factor: 4.033

9.  Searching for factors that distinguish disease-prone and disease-resistant prions via sequence analysis.

Authors:  Kanaka Durga Kedarisetti; Scott Dick; Lukasz Kurgan
Journal:  Bioinform Biol Insights       Date:  2008-03-12

10.  Decrypting Prion Protein Conversion into a β-Rich Conformer by Molecular Dynamics.

Authors:  Nesrine Chakroun; Arianna Fornili; Stéphanie Prigent; Jens Kleinjung; Cécile A Dreiss; Human Rezaei; Franca Fraternali
Journal:  J Chem Theory Comput       Date:  2013-04-04       Impact factor: 6.006

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