Literature DB >> 26170003

Gene Expression Profile in Peripheral Blood Cells of Friedreich Ataxia Patients.

Agessandro Abrahao1,2, Jose Luiz Pedroso1,2, Patricia Maria de Carvalho Aguiar3,4, Orlando Graziani Povoas Barsottini1,2.   

Abstract

Friedreich ataxia (FRDA) is the most common autosomal recessive ataxia characterized by a combination of neurological involvement, cardiomyopathy, and skeletal and glucose metabolism disturbances. FRDA is caused by mutations in FXN gene that results in reduction of mRNA and protein levels of frataxin. Previous microarray and real-time quantitative PCR (qPCR) studies showed that the downregulation of FXN is associated with a complex gene expression profile. However, these studies showed a wide variability in the subset of genes with altered expression among tissues and models. Genes differentially expressed in peripheral blood cells (PBC) could potentially help in the understanding of FRDA pathophysiology and also function as reliable disease biomarkers obtained from an easily accessible tissue, which could have implications in clinical practice. This study aimed to validate by qPCR the expression of 26 genes, revealed as differentially expressed by other studies, using peripheral blood cells (PBC) of 11 FRDA patients compared to 11 healthy controls. We found a robust downregulation of FXN, but no statistically significant differences were found between FRDA and controls for the remaining genes. Except for FXN, our study did not find a differential gene expression profile in PBC of FRDA patients and a reliable gene expression profile biomarker in a clinical relevant and noninvasive tissue remains unclear.

Entities:  

Keywords:  Biomarker; Friedreich ataxia; Gene expression; Real-time quantitative PCR; qPCR

Mesh:

Substances:

Year:  2016        PMID: 26170003     DOI: 10.1007/s12311-015-0700-x

Source DB:  PubMed          Journal:  Cerebellum        ISSN: 1473-4222            Impact factor:   3.847


  13 in total

1.  A gene expression phenotype in lymphocytes from Friedreich ataxia patients.

Authors:  Giovanni Coppola; Ryan Burnett; Susan Perlman; Revital Versano; Fuying Gao; Heather Plasterer; Myriam Rai; Francesco Saccá; Alessandro Filla; David R Lynch; James R Rusche; Joel M Gottesfeld; Massimo Pandolfo; Daniel H Geschwind
Journal:  Ann Neurol       Date:  2011-11       Impact factor: 10.422

Review 2.  Friedreich's ataxia: pathology, pathogenesis, and molecular genetics.

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

Review 3.  Clinical features of Friedreich's ataxia: classical and atypical phenotypes.

Authors:  Michael H Parkinson; Sylvia Boesch; Wolfgang Nachbauer; Caterina Mariotti; Paola Giunti
Journal:  J Neurochem       Date:  2013-08       Impact factor: 5.372

Review 4.  Therapeutic strategies in Friedreich's ataxia.

Authors:  Timothy E Richardson; Heather N Kelly; Amanda E Yu; James W Simpkins
Journal:  Brain Res       Date:  2013-04-13       Impact factor: 3.252

5.  Functional genomic analysis of frataxin deficiency reveals tissue-specific alterations and identifies the PPARgamma pathway as a therapeutic target in Friedreich's ataxia.

Authors:  Giovanni Coppola; Daniele Marmolino; Daning Lu; Qing Wang; Miriam Cnop; Myriam Rai; Fabio Acquaviva; Sergio Cocozza; Massimo Pandolfo; Daniel H Geschwind
Journal:  Hum Mol Genet       Date:  2009-04-17       Impact factor: 6.150

6.  Real time PCR quantification of frataxin mRNA in the peripheral blood leucocytes of Friedreich ataxia patients and carriers.

Authors:  L Pianese; M Turano; M S Lo Casale; I De Biase; M Giacchetti; A Monticelli; C Criscuolo; A Filla; S Cocozza
Journal:  J Neurol Neurosurg Psychiatry       Date:  2004-07       Impact factor: 10.154

7.  Elucidation of the mechanism of mitochondrial iron loading in Friedreich's ataxia by analysis of a mouse mutant.

Authors:  Michael Li-Hsuan Huang; Erika M Becker; Megan Whitnall; Yohan Suryo Rahmanto; Prem Ponka; Des R Richardson
Journal:  Proc Natl Acad Sci U S A       Date:  2009-09-04       Impact factor: 11.205

8.  A combined nucleic acid and protein analysis in Friedreich ataxia: implications for diagnosis, pathogenesis and clinical trial design.

Authors:  Francesco Saccà; Giorgia Puorro; Antonella Antenora; Angela Marsili; Alessandra Denaro; Raffaele Piro; Pierpaolo Sorrentino; Chiara Pane; Alessandra Tessa; Vincenzo Brescia Morra; Sergio Cocozza; Giuseppe De Michele; Filippo M Santorelli; Alessandro Filla
Journal:  PLoS One       Date:  2011-03-11       Impact factor: 3.240

9.  Decreased expression of genes involved in sulfur amino acid metabolism in frataxin-deficient cells.

Authors:  Guolin Tan; Eleonora Napoli; Franco Taroni; Gino Cortopassi
Journal:  Hum Mol Genet       Date:  2003-07-15       Impact factor: 6.150

10.  Gene expression profiling of mitochondrial oxidative phosphorylation (OXPHOS) complex I in Friedreich ataxia (FRDA) patients.

Authors:  Mohammad Hossein Salehi; Behnam Kamalidehghan; Massoud Houshmand; Goh Yong Meng; Majid Sadeghizadeh; Omid Aryani; Shahriar Nafissi
Journal:  PLoS One       Date:  2014-04-04       Impact factor: 3.240

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