Literature DB >> 22842711

DYT16: the original cases.

Sarah Camargos1, Andrew J Lees, Andrew Singleton, Francisco Cardoso.   

Abstract

OBJECTIVE: DYT16 is an autosomal recessive dystonia-parkinsonism due to putative mutations at PRKRA gene. The aim of this study was to describe clinical features providing video documentation of patients with DYT16 dystonia.
METHODS: We examined and videotaped all homozygous carriers of the DYT16 gene.
RESULTS: We identified two phenotypes, generalised dystonia and dystonia-parkinsonism non-responsive to levo-dopa, with three patients belonging to each of the groups. There was inter-individual and intra-family phenotypic heterogeneity.
CONCLUSIONS: DYT16 is a rare autosomal recessive dystonia characterised by generalised dystonia or dystonia-parkinsonism. Patients are refractory to pharmacological therapy.

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Year:  2012        PMID: 22842711      PMCID: PMC6376866          DOI: 10.1136/jnnp-2012-302841

Source DB:  PubMed          Journal:  J Neurol Neurosurg Psychiatry        ISSN: 0022-3050            Impact factor:   10.154


  7 in total

1.  PRKRAP1 Pseudogene Complicating the Diagnosis of Young-Onset Dystonia Due to PRKRA Gene Disease-Causing Variants (DYT-PRKRA).

Authors:  Joana Afonso Ribeiro; Mário Sousa; Isabel Alonso; Fradique Moreira; Ricardo Pereira; Filipe Palavra
Journal:  Mov Disord Clin Pract       Date:  2022-03-04

2.  DYT-PRKRA Mutation P222L Enhances PACT's Stimulatory Activity on Type I Interferon Induction.

Authors:  Lauren S Vaughn; Kenneth Frederick; Samuel B Burnett; Nutan Sharma; D Cristopher Bragg; Sarah Camargos; Francisco Cardoso; Rekha C Patel
Journal:  Biomolecules       Date:  2022-05-17

Review 3.  The Patho-Neurophysiological Basis and Treatment of Focal Laryngeal Dystonia: A Narrative Review and Two Case Reports Applying TMS over the Laryngeal Motor Cortex.

Authors:  Maja Rogić Vidaković; Ivana Gunjača; Josipa Bukić; Vana Košta; Joško Šoda; Ivan Konstantinović; Braco Bošković; Irena Bilić; Nikolina Režić Mužinić
Journal:  J Clin Med       Date:  2022-06-15       Impact factor: 4.964

4.  Altered activation of protein kinase PKR and enhanced apoptosis in dystonia cells carrying a mutation in PKR activator protein PACT.

Authors:  Lauren S Vaughn; D Cristopher Bragg; Nutan Sharma; Sarah Camargos; Francisco Cardoso; Rekha C Patel
Journal:  J Biol Chem       Date:  2015-07-31       Impact factor: 5.157

5.  Recent advances in the molecular pathogenesis of dystonia-plus syndromes and heredodegenerative dystonias.

Authors:  Catharina Casper; Eirini Kalliolia; Thomas T Warner
Journal:  Curr Neuropharmacol       Date:  2013-01       Impact factor: 7.363

Review 6.  Combined dystonias: clinical and genetic updates.

Authors:  Anne Weissbach; Gerard Saranza; Aloysius Domingo
Journal:  J Neural Transm (Vienna)       Date:  2020-10-24       Impact factor: 3.575

7.  Decreased N-TAF1 expression in X-linked dystonia-parkinsonism patient-specific neural stem cells.

Authors:  Naoto Ito; William T Hendriks; Jyotsna Dhakal; Christine A Vaine; Christina Liu; David Shin; Kyle Shin; Noriko Wakabayashi-Ito; Marisela Dy; Trisha Multhaupt-Buell; Nutan Sharma; Xandra O Breakefield; D Cristopher Bragg
Journal:  Dis Model Mech       Date:  2016-01-14       Impact factor: 5.758

  7 in total

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