Literature DB >> 35766110

Friedreich ataxia: clinical features and new developments.

Medina Keita1, Kellie McIntyre1, Layne N Rodden1, Kim Schadt1, David R Lynch1.   

Abstract

Friedreich's ataxia (FRDA), a neurodegenerative disease characterized by ataxia and other neurological features, affects 1 in 50,000-100,000 individuals in the USA. However, FRDA also includes cardiac, orthopedic and endocrine dysfunction, giving rise to many secondary disease characteristics. The multifaceted approach for clinical care has necessitated the development of disease-specific clinical care guidelines. New developments in FRDA include the advancement of clinical drug trials targeting the NRF2 pathway and frataxin restoration. Additionally, a novel understanding of gene silencing in FRDA, reflecting a variegated silencing pattern, will have applications to current and future therapeutic interventions. Finally, new perspectives on the neuroanatomy of FRDA and its developmental features will refine the time course and anatomical targeting of novel approaches.

Entities:  

Keywords:  Frataxin; NRF2; antioxidant; clinical care guideline; clinical trial; multisystem

Mesh:

Substances:

Year:  2022        PMID: 35766110      PMCID: PMC9517959          DOI: 10.2217/nmt-2022-0011

Source DB:  PubMed          Journal:  Neurodegener Dis Manag        ISSN: 1758-2024


  169 in total

Review 1.  Friedreich's ataxia: pathology, pathogenesis, and molecular genetics.

Authors:  Arnulf H Koeppen
Journal:  J Neurol Sci       Date:  2011-04-15       Impact factor: 3.181

2.  Friedreich's ataxia: a clinical and genetic analysis.

Authors:  P Leema Reddy; Raji P Grewal
Journal:  Clin Neurol Neurosurg       Date:  2006-10-17       Impact factor: 1.876

3.  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

4.  Characterizing cardiac phenotype in Friedreich's ataxia: The CARFA study.

Authors:  Lise Legrand; Jonathan W Weinsaft; Francoise Pousset; Claire Ewenczyk; Perrine Charles; Stéphane Hatem; Anna Heinzmann; Marie Biet; Alexandra Durr; Alban Redheuil
Journal:  Arch Cardiovasc Dis       Date:  2021-12-16       Impact factor: 2.340

5.  Heterochromatinization induced by GAA-repeat hyperexpansion in Friedreich's ataxia can be reduced upon HDAC inhibition by vitamin B3.

Authors:  Ping K Chan; Raul Torres; Cihangir Yandim; Pui P Law; Sanjay Khadayate; Marta Mauri; Crina Grosan; Nadine Chapman-Rothe; Paola Giunti; Mark Pook; Richard Festenstein
Journal:  Hum Mol Genet       Date:  2013-03-07       Impact factor: 6.150

6.  Epigenetic Heterogeneity in Friedreich Ataxia Underlies Variable FXN Reactivation.

Authors:  Layne N Rodden; Kaitlyn M Gilliam; Christina Lam; David R Lynch; Sanjay I Bidichandani
Journal:  Front Neurosci       Date:  2021-11-25       Impact factor: 4.677

7.  Repeat-induced epigenetic changes in intron 1 of the frataxin gene and its consequences in Friedreich ataxia.

Authors:  Eriko Greene; Lata Mahishi; Ali Entezam; Daman Kumari; Karen Usdin
Journal:  Nucleic Acids Res       Date:  2007-05-03       Impact factor: 16.971

8.  Bioenergetics of the calf muscle in Friedreich ataxia patients measured by 31P-MRS before and after treatment with recombinant human erythropoietin.

Authors:  Wolfgang Nachbauer; Sylvia Boesch; Rainer Schneider; Andreas Eigentler; Julia Wanschitz; Werner Poewe; Michael Schocke
Journal:  PLoS One       Date:  2013-07-29       Impact factor: 3.240

9.  Significance of NT-proBNP and High-sensitivity Troponin in Friedreich Ataxia.

Authors:  Lise Legrand; Carole Maupain; Marie-Lorraine Monin; Claire Ewenczyk; Richard Isnard; Rana Alkouri; Alexandra Durr; Francoise Pousset
Journal:  J Clin Med       Date:  2020-05-28       Impact factor: 4.241

10.  Depressive symptoms in Friedreich ataxia.

Authors:  Antonieta Nieto; Atteneri Hernández-Torres; Javier Pérez-Flores; Fernando Montón
Journal:  Int J Clin Health Psychol       Date:  2017-12-08
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