Literature DB >> 25109986

Aberrant redox homoeostasis and mitochondrial dysfunction in Rett syndrome.

Michael Müller1, Karolina Can1.   

Abstract

RTT (Rett syndrome) is a severe progressive neurodevelopmental disorder with a monogenetic cause, but complex and multifaceted clinical appearance. Compelling evidence suggests that mitochondrial alterations and aberrant redox homoeostasis result in oxidative challenge. Yet, compared with other severe neuropathologies, RTT is not associated with marked neurodegeneration, but rather a chemical imbalance and miscommunication of neuronal elements. Different pharmacotherapies mediate partial improvement of conditions in RTT, and also antioxidants or compounds improving mitochondrial function may be of potential merit. In the present paper, we summarize findings from patients and transgenic mice that point towards the nature of RTT as a mitochondrial disease. Also, open questions are addressed that require clarification to fully understand and successfully target the associated cellular redox imbalance.

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Year:  2014        PMID: 25109986     DOI: 10.1042/BST20140071

Source DB:  PubMed          Journal:  Biochem Soc Trans        ISSN: 0300-5127            Impact factor:   5.407


  14 in total

1.  Aberrant mitochondrial function in patient-derived neural cells from CDKL5 deficiency disorder and Rett syndrome.

Authors:  Smita Jagtap; Jessica M Thanos; Ting Fu; Jennifer Wang; Jasmin Lalonde; Thomas O Dial; Ariel Feiglin; Jeffrey Chen; Isaac Kohane; Jeannie T Lee; Steven D Sheridan; Roy H Perlis
Journal:  Hum Mol Genet       Date:  2019-11-01       Impact factor: 6.150

2.  PTP1B inhibition suggests a therapeutic strategy for Rett syndrome.

Authors:  Navasona Krishnan; Keerthi Krishnan; Christopher R Connors; Meng S Choy; Rebecca Page; Wolfgang Peti; Linda Van Aelst; Stephen D Shea; Nicholas K Tonks
Journal:  J Clin Invest       Date:  2015-07-27       Impact factor: 14.808

3.  Cytokine Dysregulation in MECP2- and CDKL5-Related Rett Syndrome: Relationships with Aberrant Redox Homeostasis, Inflammation, and ω-3 PUFAs.

Authors:  Silvia Leoncini; Claudio De Felice; Cinzia Signorini; Gloria Zollo; Alessio Cortelazzo; Thierry Durand; Jean-Marie Galano; Roberto Guerranti; Marcello Rossi; Lucia Ciccoli; Joussef Hayek
Journal:  Oxid Med Cell Longev       Date:  2015-07-08       Impact factor: 6.543

4.  Systemic Radical Scavenger Treatment of a Mouse Model of Rett Syndrome: Merits and Limitations of the Vitamin E Derivative Trolox.

Authors:  Oliwia A Janc; Marc A Hüser; Katharina Dietrich; Belinda Kempkes; Christiane Menzfeld; Swen Hülsmann; Michael Müller
Journal:  Front Cell Neurosci       Date:  2016-11-15       Impact factor: 5.505

5.  Alterations in the carnitine cycle in a mouse model of Rett syndrome.

Authors:  Sabrina Mucerino; Anna Di Salle; Nicola Alessio; Sabrina Margarucci; Raffaella Nicolai; Mariarosa A B Melone; Umberto Galderisi; Gianfranco Peluso
Journal:  Sci Rep       Date:  2017-02-02       Impact factor: 4.379

6.  Biotin tagging of MeCP2 in mice reveals contextual insights into the Rett syndrome transcriptome.

Authors:  Brian S Johnson; Ying-Tao Zhao; Maria Fasolino; Janine M Lamonica; Yoon Jung Kim; George Georgakilas; Kathleen H Wood; Daniel Bu; Yue Cui; Darren Goffin; Golnaz Vahedi; Tae Hoon Kim; Zhaolan Zhou
Journal:  Nat Med       Date:  2017-09-18       Impact factor: 53.440

Review 7.  Mitochondrial Dysfunction in the Pathogenesis of Rett Syndrome: Implications for Mitochondria-Targeted Therapies.

Authors:  Natalya Shulyakova; Ana C Andreazza; Linda R Mills; James H Eubanks
Journal:  Front Cell Neurosci       Date:  2017-03-14       Impact factor: 5.505

8.  MECP2 Duplication Syndrome: Evidence of Enhanced Oxidative Stress. A Comparison with Rett Syndrome.

Authors:  Cinzia Signorini; Claudio De Felice; Silvia Leoncini; Rikke S Møller; Gloria Zollo; Sabrina Buoni; Alessio Cortelazzo; Roberto Guerranti; Thierry Durand; Lucia Ciccoli; Maurizio D'Esposito; Kirstine Ravn; Joussef Hayek
Journal:  PLoS One       Date:  2016-03-01       Impact factor: 3.240

9.  MeCP2 co-ordinates liver lipid metabolism with the NCoR1/HDAC3 corepressor complex.

Authors:  Stephanie M Kyle; Pradip K Saha; Hannah M Brown; Lawrence C Chan; Monica J Justice
Journal:  Hum Mol Genet       Date:  2016-06-10       Impact factor: 6.150

10.  Increased Mitochondrial Mass and Cytosolic Redox Imbalance in Hippocampal Astrocytes of a Mouse Model of Rett Syndrome: Subcellular Changes Revealed by Ratiometric Imaging of JC-1 and roGFP1 Fluorescence.

Authors:  Dörthe F Bebensee; Karolina Can; Michael Müller
Journal:  Oxid Med Cell Longev       Date:  2017-08-13       Impact factor: 6.543

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