Literature DB >> 22832776

Unanswered questions in Friedreich ataxia.

David R Lynch1, Eric C Deutsch, Robert B Wilson, Gihan Tennekoon.   

Abstract

During the past 15 years, the pace of research advancement in Friedreich ataxia has been rapid. The abnormal gene has been discovered and its gene product characterized, leading to the development of new evidence-based therapies. Still, various unsettled issues remain that affect clinical trials. These include the level of frataxin deficiency needed to cause disease, the mechanism by which frataxin-deficient mitochondrial dysfunction leads to symptomatology, and the reason selected cells are most affected in Friedreich ataxia. In this review, we summarize these questions and propose testable hypotheses for their resolution.

Entities:  

Mesh:

Substances:

Year:  2012        PMID: 22832776      PMCID: PMC3674553          DOI: 10.1177/0883073812453498

Source DB:  PubMed          Journal:  J Child Neurol        ISSN: 0883-0738            Impact factor:   1.987


  69 in total

Review 1.  Peripheral nerve regeneration and neurotrophic factors.

Authors:  G Terenghi
Journal:  J Anat       Date:  1999-01       Impact factor: 2.610

2.  Friedreich ataxia in carriers of unstable borderline GAA triplet-repeat alleles.

Authors:  Rajesh Sharma; Irene De Biase; Mariluz Gómez; Martin B Delatycki; Tetsuo Ashizawa; Sanjay I Bidichandani
Journal:  Ann Neurol       Date:  2004-12       Impact factor: 10.422

3.  Late-onset Friedreich ataxia: phenotypic analysis, magnetic resonance imaging findings, and review of the literature.

Authors:  Roongroj Bhidayasiri; Susan L Perlman; Stefan-M Pulst; Daniel H Geschwind
Journal:  Arch Neurol       Date:  2005-12

Review 4.  Iron and Friedreich ataxia.

Authors:  M Pandolfo
Journal:  J Neural Transm Suppl       Date:  2006

5.  Microcytic anemia, erythropoietic protoporphyria, and neurodegeneration in mice with targeted deletion of iron-regulatory protein 2.

Authors:  Sharon S Cooperman; Esther G Meyron-Holtz; Hayden Olivierre-Wilson; Manik C Ghosh; Joseph P McConnell; Tracey A Rouault
Journal:  Blood       Date:  2005-04-14       Impact factor: 22.113

Review 6.  Development and specification of muscle sensory neurons.

Authors:  H H Chen; E Frank
Journal:  Curr Opin Neurobiol       Date:  1999-08       Impact factor: 6.627

7.  Histone deacetylase inhibitors reverse gene silencing in Friedreich's ataxia.

Authors:  David Herman; Kai Jenssen; Ryan Burnett; Elisabetta Soragni; Susan L Perlman; Joel M Gottesfeld
Journal:  Nat Chem Biol       Date:  2006-08-20       Impact factor: 15.040

8.  Intermediate expansions of a GAA repeat in the frataxin gene are not associated with type 2 diabetes or altered glucose-induced beta-cell function in Danish Caucasians.

Authors:  L T Dalgaard; T Hansen; S A Urhammer; J O Clausen; H Eiberg; O Pedersen
Journal:  Diabetes       Date:  1999-04       Impact factor: 9.461

9.  Altered beta-cell characteristics in impaired glucose tolerant carriers of a GAA trinucleotide repeat polymorphism in the frataxin gene.

Authors:  L M t Hart; J B Ruige; J M Dekker; C D Stehouwer; J A Maassen; R J Heine
Journal:  Diabetes       Date:  1999-04       Impact factor: 9.461

10.  Friedreich's ataxia: point mutations and clinical presentation of compound heterozygotes.

Authors:  M Cossée; A Dürr; M Schmitt; N Dahl; P Trouillas; P Allinson; M Kostrzewa; A Nivelon-Chevallier; K H Gustavson; A Kohlschütter; U Müller; J L Mandel; A Brice; M Koenig; F Cavalcanti; A Tammaro; G De Michele; A Filla; S Cocozza; M Labuda; L Montermini; J Poirier; M Pandolfo
Journal:  Ann Neurol       Date:  1999-02       Impact factor: 10.422

View more
  13 in total

1.  Inducible and reversible phenotypes in a novel mouse model of Friedreich's Ataxia.

Authors:  Vijayendran Chandran; Kun Gao; Vivek Swarup; Revital Versano; Hongmei Dong; Maria C Jordan; Daniel H Geschwind
Journal:  Elife       Date:  2017-12-19       Impact factor: 8.140

2.  Paradigm for disease deconvolution in rare neurodegenerative disorders in Indian population: insights from studies in cerebellar ataxias.

Authors:  Renu Kumari; Deepak Kumar; Samir K Brahmachari; Achal K Srivastava; Mohammed Faruq; Mitali Mukerji
Journal:  J Genet       Date:  2018-07       Impact factor: 1.166

Review 3.  Transition of Thalassaemia and Friedreich ataxia from fatal to chronic diseases.

Authors:  Annita Kolnagou; Christina N Kontoghiorghe; George J Kontoghiorghes
Journal:  World J Methodol       Date:  2014-12-26

Review 4.  Friedreich ataxia: clinical features and new developments.

Authors:  Medina Keita; Kellie McIntyre; Layne N Rodden; Kim Schadt; David R Lynch
Journal:  Neurodegener Dis Manag       Date:  2022-06-29

5.  Human platelets as a platform to monitor metabolic biomarkers using stable isotopes and LC-MS.

Authors:  Sankha S Basu; Eric C Deutsch; Alec A Schmaier; David R Lynch; Ian A Blair
Journal:  Bioanalysis       Date:  2013-12       Impact factor: 2.681

6.  Gene Transfer of Brain-derived Neurotrophic Factor (BDNF) Prevents Neurodegeneration Triggered by FXN Deficiency.

Authors:  Yurika Katsu-Jiménez; Frida Loría; Juan Carlos Corona; Javier Díaz-Nido
Journal:  Mol Ther       Date:  2016-02-05       Impact factor: 11.454

7.  Progress in understanding Friedreich's ataxia using human induced pluripotent stem cells.

Authors:  Anna M Schreiber; Julia O Misiorek; Jill S Napierala; Marek Napierala
Journal:  Expert Opin Orphan Drugs       Date:  2019-01-09       Impact factor: 0.694

Review 8.  Induced pluripotent stem cell technology for modelling and therapy of cerebellar ataxia.

Authors:  Lauren M Watson; Maggie M K Wong; Esther B E Becker
Journal:  Open Biol       Date:  2015-07       Impact factor: 6.411

9.  Early VGLUT1-specific parallel fiber synaptic deficits and dysregulated cerebellar circuit in the KIKO mouse model of Friedreich ataxia.

Authors:  Hong Lin; Jordi Magrane; Elisia M Clark; Sarah M Halawani; Nathan Warren; Amy Rattelle; David R Lynch
Journal:  Dis Model Mech       Date:  2017-12-19       Impact factor: 5.758

10.  Early cerebellar deficits in mitochondrial biogenesis and respiratory chain complexes in the KIKO mouse model of Friedreich ataxia.

Authors:  Hong Lin; Jordi Magrane; Amy Rattelle; Anna Stepanova; Alexander Galkin; Elisia M Clark; Yi Na Dong; Sarah M Halawani; David R Lynch
Journal:  Dis Model Mech       Date:  2017-11-01       Impact factor: 5.758

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.