Literature DB >> 25606360

Current research, diagnosis, and treatment of fragile X-associated tremor/ataxia syndrome.

Zukhrofi Muzar1, Reymundo Lozano1.   

Abstract

Fragile X-associated tremor/ataxia syndrome (FXTAS) is caused by a premutation CGG-repeat expansion in the 5'UTR of the fragile X mental retardation 1 (FMR1) gene. The classical clinical manifestations include tremor, cerebellar ataxia, cognitive decline and psychiatric disorders. Other less frequent features are peripheral neuropathy and autonomic dysfunction. Cognitive decline, a form of frontal subcortical dementia, memory loss and executive function deficits are also characteristics of this disorder. In this review, we present an expansion of recommendations for genetic testing for adults with suspected premutation disorders and provide an update of the clinical, radiological and molecular research of FXTAS, as well as the current research in the treatment for this intractable complex neurodegenerative genetic disorder.

Entities:  

Keywords:  FMPR; FMR1; FMR1 mRNA; FXTAS; late-onset neurological disorder and neurodegenerative disorder; premutation carrier; tremor/ataxia

Year:  2014        PMID: 25606360      PMCID: PMC4298640          DOI: 10.5582/irdr.2014.01029

Source DB:  PubMed          Journal:  Intractable Rare Dis Res        ISSN: 2186-3644


  103 in total

1.  Symptomatic treatment in the fragile X-associated tremor/ataxia syndrome.

Authors:  Deborah A Hall; Elizabeth Berry-Kravis; Randi J Hagerman; Paul J Hagerman; Cathlin D Rice; Maureen A Leehey
Journal:  Mov Disord       Date:  2006-10       Impact factor: 10.338

2.  The fragile X prevalence paradox.

Authors:  Paul J Hagerman
Journal:  J Med Genet       Date:  2008-04-15       Impact factor: 6.318

3.  Hyperintensity in the basis pontis: atypical neuroradiological findings in a woman with FXTAS.

Authors:  Germán Morís; Mónica Arias; Maria Valle López; Victoria Alvarez
Journal:  Mov Disord       Date:  2010-04-15       Impact factor: 10.338

4.  The neurocognitive phenotype of female carriers of fragile X: additional evidence for specificity.

Authors:  M M Mazzocco; B F Pennington; R J Hagerman
Journal:  J Dev Behav Pediatr       Date:  1993-10       Impact factor: 2.225

5.  Intrafamilial variability in fragile X-associated tremor/ataxia syndrome.

Authors:  Nils Peters; Christoph Kamm; Friedrich Asmus; Elke Holinski-Feder; Eduard Kraft; Martin Dichgans; Roland Brüning; Thomas Gasser; Kai Bötzel
Journal:  Mov Disord       Date:  2006-01       Impact factor: 10.338

6.  Expanded clinical phenotype of women with the FMR1 premutation.

Authors:  Sarah M Coffey; Kylee Cook; Nicole Tartaglia; Flora Tassone; Danh V Nguyen; Ruiqin Pan; Hannah E Bronsky; Jennifer Yuhas; Mariya Borodyanskaya; Jim Grigsby; Melanie Doerflinger; Paul J Hagerman; Randi J Hagerman
Journal:  Am J Med Genet A       Date:  2008-04-15       Impact factor: 2.802

7.  Lifespan changes in working memory in fragile X premutation males.

Authors:  Kim M Cornish; Cary S Kogan; Lexin Li; Jeremy Turk; Sebastien Jacquemont; Randi J Hagerman
Journal:  Brain Cogn       Date:  2008-12-27       Impact factor: 2.310

8.  Pur alpha binds to rCGG repeats and modulates repeat-mediated neurodegeneration in a Drosophila model of fragile X tremor/ataxia syndrome.

Authors:  Peng Jin; Ranhui Duan; Abrar Qurashi; Yunlong Qin; Donghua Tian; Tracie C Rosser; Huijie Liu; Yue Feng; Stephen T Warren
Journal:  Neuron       Date:  2007-08-16       Impact factor: 17.173

9.  Deep brain stimulation for tremor associated with underlying ataxia syndromes: a case series and discussion of issues.

Authors:  Genko Oyama; Amanda Thompson; Kelly D Foote; Natlada Limotai; Muhammad Abd-El-Barr; Nicholas Maling; Irene A Malaty; Ramon L Rodriguez; Sankarasubramoney H Subramony; Tetsuo Ashizawa; Michael S Okun
Journal:  Tremor Other Hyperkinet Mov (N Y)       Date:  2014-07-09

Review 10.  Treatment of fragile X-associated tremor ataxia syndrome (FXTAS) and related neurological problems.

Authors:  Randi J Hagerman; Deborah A Hall; Sarah Coffey; Maureen Leehey; James Bourgeois; John Gould; Lin Zhang; Andreea Seritan; Elizabeth Berry-Kravis; John Olichney; Joshua W Miller; Amy L Fong; Randall Carpenter; Cathy Bodine; Louise W Gane; Edgar Rainin; Hillary Hagerman; Paul J Hagerman
Journal:  Clin Interv Aging       Date:  2008       Impact factor: 4.458

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  6 in total

Review 1.  Current concepts in the treatment of hereditary ataxias.

Authors:  Pedro Braga Neto; José Luiz Pedroso; Sheng-Han Kuo; C França Marcondes Junior; Hélio Afonso Ghizoni Teive; Orlando Graziani Povoas Barsottini
Journal:  Arq Neuropsiquiatr       Date:  2016-03       Impact factor: 1.420

2.  Aging in Fragile X Premutation Carriers.

Authors:  Reymundo Lozano; Naomi Saito; Dallas Reed; Marwa Eldeeb; Andrea Schneider; David Hessl; Flora Tassone; Laurel Beckett; Randi Hagerman
Journal:  Cerebellum       Date:  2016-10       Impact factor: 3.847

3.  Women with Fragile X-associated Tremor/Ataxia Syndrome.

Authors:  Andrea Schneider; Scott Summers; Flora Tassone; Andreea Seritan; David Hessl; Paul Hagerman; Randi Hagerman
Journal:  Mov Disord Clin Pract       Date:  2020-09-23

Review 4.  Conventional MRI findings in hereditary degenerative ataxias: a pictorial review.

Authors:  Sirio Cocozza; Giuseppe Pontillo; Giovanna De Michele; Martina Di Stasi; Elvira Guerriero; Teresa Perillo; Chiara Pane; Anna De Rosa; Lorenzo Ugga; Arturo Brunetti
Journal:  Neuroradiology       Date:  2021-03-17       Impact factor: 2.804

Review 5.  Fragile X-Associated Tremor/Ataxia Syndrome: From Molecular Pathogenesis to Development of Therapeutics.

Authors:  Ha Eun Kong; Juan Zhao; Shunliang Xu; Peng Jin; Yan Jin
Journal:  Front Cell Neurosci       Date:  2017-05-05       Impact factor: 5.505

6.  Study of patterns of inheritance of premature ovarian failure syndrome carrying maternal and paternal premutations.

Authors:  Artur Beke; Henriett Piko; Iren Haltrich; Veronika Karcagi; Janos Rigo; Maria Judit Molnar; György Fekete
Journal:  BMC Med Genet       Date:  2018-07-09       Impact factor: 2.103

  6 in total

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