Literature DB >> 20865765

Direct interaction between causative genes of DYT1 and DYT6 primary dystonia.

Sophie Gavarini1, Corinne Cayrol, Tania Fuchs, Natalia Lyons, Michelle E Ehrlich, Jean-Philippe Girard, Laurie J Ozelius.   

Abstract

Primary dystonia is a movement disorder characterized by sustained muscle contractions and in which dystonia is the only or predominant clinical feature. TOR1A(DYT1) and the transcription factor THAP1(DYT6) are the only genes identified thus far for primary dystonia. Using electromobility shift assays and chromatin immunoprecipitation (ChIP) quantitative polymerase chain reaction (qPCR), we demonstrate a physical interaction between THAP1 and the TOR1A promoter that is abolished by pathophysiologic mutations. Our findings provide the first evidence that causative genes for primary dystonia intersect in a common pathway and raise the possibility of developing novel therapies targeting this pathway.

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Year:  2010        PMID: 20865765      PMCID: PMC3038652          DOI: 10.1002/ana.22138

Source DB:  PubMed          Journal:  Ann Neurol        ISSN: 0364-5134            Impact factor:   10.422


  24 in total

1.  Identification of a novel THAP1 mutation at R29 amino-acid residue in sporadic patients with early-onset dystonia.

Authors:  Coro Paisán-Ruiz; Javier Ruiz-Martinez; Marta Ruibal; Kin Y Mok; Begona Indakoetxea; Ana Gorostidi; José F Martí Massó
Journal:  Mov Disord       Date:  2009-12-15       Impact factor: 10.338

2.  Relative tissue expression of homologous torsinB correlates with the neuronal specific importance of DYT1 dystonia-associated torsinA.

Authors:  Michael Jungwirth; Mary Lynn Dear; Patricia Brown; Kristen Holbrook; Rose Goodchild
Journal:  Hum Mol Genet       Date:  2009-12-16       Impact factor: 6.150

3.  Mutation screening of the DYT6/THAP1 gene in Italy.

Authors:  Monica Bonetti; Chiara Barzaghi; Francesco Brancati; Alessandro Ferraris; Emanuele Bellacchio; Alessandro Giovanetti; Tamara Ialongo; Giovanna Zorzi; Carla Piano; Martina Petracca; Alberto Albanese; Nardo Nardocci; Bruno Dallapiccola; Anna Rita Bentivoglio; Barbara Garavaglia; Enza Maria Valente
Journal:  Mov Disord       Date:  2009-12-15       Impact factor: 10.338

4.  Novel THAP1 sequence variants in primary dystonia.

Authors:  J Xiao; Y Zhao; R W Bastian; J S Perlmutter; B A Racette; S D Tabbal; M Karimi; R C Paniello; Z K Wszolek; R J Uitti; J A Van Gerpen; D K Simon; D Tarsy; P Hedera; D D Truong; K P Frei; S Dev Batish; A Blitzer; R F Pfeiffer; S Gong; M S LeDoux
Journal:  Neurology       Date:  2010-01-19       Impact factor: 9.910

5.  Mutations in THAP1 (DYT6) and generalised dystonia with prominent spasmodic dysphonia: a genetic screening study.

Authors:  Ana Djarmati; Susanne A Schneider; Katja Lohmann; Susen Winkler; Heike Pawlack; Johann Hagenah; Norbert Brüggemann; Simone Zittel; Tania Fuchs; Aleksandar Raković; Alexander Schmidt; Hans-Christian Jabusch; Robert Wilcox; Vladimir S Kostić; Hartwig Siebner; Eckart Altenmüller; Alexander Münchau; Laurie J Ozelius; Christine Klein
Journal:  Lancet Neurol       Date:  2009-04-01       Impact factor: 44.182

6.  Factors predicting improvement in primary generalized dystonia treated by pallidal deep brain stimulation.

Authors:  Xavier Vasques; Laura Cif; Victoria Gonzalez; Claire Nicholson; Philippe Coubes
Journal:  Mov Disord       Date:  2009-04-30       Impact factor: 10.338

7.  THAP1 mutations (DYT6) are an additional cause of early-onset dystonia.

Authors:  H Houlden; S A Schneider; R Paudel; A Melchers; P Schwingenschuh; M Edwards; J Hardy; K P Bhatia
Journal:  Neurology       Date:  2010-03-09       Impact factor: 9.910

8.  E2f1-3 switch from activators in progenitor cells to repressors in differentiating cells.

Authors:  Jean-Leon Chong; Pamela L Wenzel; M Teresa Sáenz-Robles; Vivek Nair; Antoney Ferrey; John P Hagan; Yorman M Gomez; Nidhi Sharma; Hui-Zi Chen; Madhu Ouseph; Shu-Huei Wang; Prashant Trikha; Brian Culp; Louise Mezache; Douglas J Winton; Owen J Sansom; Danian Chen; Rod Bremner; Paul G Cantalupo; Michael L Robinson; James M Pipas; Gustavo Leone
Journal:  Nature       Date:  2009-12-17       Impact factor: 49.962

Review 9.  Convergent evidence for abnormal striatal synaptic plasticity in dystonia.

Authors:  David A Peterson; Terrence J Sejnowski; Howard Poizner
Journal:  Neurobiol Dis       Date:  2009-12-18       Impact factor: 5.996

10.  THAP proteins target specific DNA sites through bipartite recognition of adjacent major and minor grooves.

Authors:  Alex Sabogal; Artem Y Lyubimov; Jacob E Corn; James M Berger; Donald C Rio
Journal:  Nat Struct Mol Biol       Date:  2009-12-13       Impact factor: 15.369

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  35 in total

Review 1.  Genotype-phenotype correlations in THAP1 dystonia: molecular foundations and description of new cases.

Authors:  Mark S LeDoux; Jianfeng Xiao; Monika Rudzińska; Robert W Bastian; Zbigniew K Wszolek; Jay A Van Gerpen; Andreas Puschmann; Dragana Momčilović; Satya R Vemula; Yu Zhao
Journal:  Parkinsonism Relat Disord       Date:  2012-02-28       Impact factor: 4.891

Review 2.  Genetic and clinical features of primary torsion dystonia.

Authors:  Laurie J Ozelius; Susan B Bressman
Journal:  Neurobiol Dis       Date:  2010-12-17       Impact factor: 5.996

Review 3.  Inherited isolated dystonia: clinical genetics and gene function.

Authors:  William Dauer
Journal:  Neurotherapeutics       Date:  2014-10       Impact factor: 7.620

4.  Dimerization of the DYT6 dystonia protein, THAP1, requires residues within the coiled-coil domain.

Authors:  Cem Sengel; Sophie Gavarini; Nutan Sharma; Laurie J Ozelius; D Cristopher Bragg
Journal:  J Neurochem       Date:  2011-08-08       Impact factor: 5.372

5.  Identification and functional analysis of novel THAP1 mutations.

Authors:  Katja Lohmann; Nils Uflacker; Alev Erogullari; Thora Lohnau; Susen Winkler; Andreas Dendorfer; Susanne A Schneider; Alma Osmanovic; Marina Svetel; Andreas Ferbert; Simone Zittel; Andrea A Kühn; Alexander Schmidt; Eckart Altenmüller; Alexander Münchau; Christoph Kamm; Matthias Wittstock; Andreas Kupsch; Elena Moro; Jens Volkmann; Vladimir Kostic; Frank J Kaiser; Christine Klein; Norbert Brüggemann
Journal:  Eur J Hum Genet       Date:  2011-08-17       Impact factor: 4.246

Review 6.  NMR studies of a new family of DNA binding proteins: the THAP proteins.

Authors:  Virginie Gervais; Sébastien Campagne; Jade Durand; Isabelle Muller; Alain Milon
Journal:  J Biomol NMR       Date:  2013-01-11       Impact factor: 2.835

Review 7.  Understanding the anatomy of dystonia: determinants of penetrance and phenotype.

Authors:  Renata P Lerner; Martin Niethammer; David Eidelberg
Journal:  Curr Neurol Neurosci Rep       Date:  2013-11       Impact factor: 5.081

8.  Improved motor performance in Dyt1 ΔGAG heterozygous knock-in mice by cerebellar Purkinje-cell specific Dyt1 conditional knocking-out.

Authors:  Fumiaki Yokoi; Mai Tu Dang; Yuqing Li
Journal:  Behav Brain Res       Date:  2012-02-25       Impact factor: 3.332

9.  Whispering dysphonia (DYT4 dystonia) is caused by a mutation in the TUBB4 gene.

Authors:  Katja Lohmann; Robert A Wilcox; Susen Winkler; Alfredo Ramirez; Aleksandar Rakovic; Jin-Sung Park; Björn Arns; Thora Lohnau; Justus Groen; Meike Kasten; Norbert Brüggemann; Johann Hagenah; Alexander Schmidt; Frank J Kaiser; Kishore R Kumar; Katja Zschiedrich; Daniel Alvarez-Fischer; Eckart Altenmüller; Andreas Ferbert; Anthony E Lang; Alexander Münchau; Vladimir Kostic; Kristina Simonyan; Marc Agzarian; Laurie J Ozelius; Antonius P M Langeveld; Carolyn M Sue; Marina A J Tijssen; Christine Klein
Journal:  Ann Neurol       Date:  2013-04-17       Impact factor: 10.422

10.  Neural expression of the transcription factor THAP1 during development in rat.

Authors:  Y Zhao; J Xiao; S Gong; J A Clara; M S Ledoux
Journal:  Neuroscience       Date:  2012-12-05       Impact factor: 3.590

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