Literature DB >> 24576561

Ubiquinol-10 ameliorates mitochondrial encephalopathy associated with CoQ deficiency.

Laura García-Corzo1, Marta Luna-Sánchez1, Carolina Doerrier1, Francisco Ortiz1, Germaine Escames1, Darío Acuña-Castroviejo1, Luis C López2.   

Abstract

Coenzyme Q10 (CoQ10) deficiency (MIM 607426) causes a mitochondrial syndrome with variability in the clinical presentations. Patients with CoQ10 deficiency show inconsistent responses to oral ubiquinone-10 supplementation, with the highest percentage of unsuccessful results in patients with neurological symptoms (encephalopathy, cerebellar ataxia or multisystemic disease). Failure in the ubiquinone-10 treatment may be the result of its poor absorption and bioavailability, which may be improved by using different pharmacological formulations. In a mouse model (Coq9(X/X)) of mitochondrial encephalopathy due to CoQ deficiency, we have evaluated oral supplementation with water-soluble formulations of reduced (ubiquinol-10) and oxidized (ubiquinone-10) forms of CoQ10. Our results show that CoQ10 was increased in all tissues after supplementation with ubiquinone-10 or ubiquinol-10, with the tissue levels of CoQ10 with ubiquinol-10 being higher than with ubiquinone-10. Moreover, only ubiquinol-10 was able to increase the levels of CoQ10 in mitochondria from cerebrum of Coq9(X/X) mice. Consequently, ubiquinol-10 was more efficient than ubiquinone-10 in increasing the animal body weight and CoQ-dependent respiratory chain complex activities, and reducing the vacuolization, astrogliosis and oxidative damage in diencephalon, septum-striatum and, to a lesser extent, in brainstem. These results suggest that water-soluble formulations of ubiquinol-10 may improve the efficacy of CoQ10 therapy in primary and secondary CoQ10 deficiencies, other mitochondrial diseases and neurodegenerative diseases.
Copyright © 2014 Elsevier B.V. All rights reserved.

Entities:  

Keywords:  CoQ(10) deficiency; Mitochondrial encephalopathy; Mouse model; Ubiquinol-10

Mesh:

Substances:

Year:  2014        PMID: 24576561     DOI: 10.1016/j.bbadis.2014.02.008

Source DB:  PubMed          Journal:  Biochim Biophys Acta        ISSN: 0006-3002


  25 in total

Review 1.  Biochemistry of Mitochondrial Coenzyme Q Biosynthesis.

Authors:  Jonathan A Stefely; David J Pagliarini
Journal:  Trends Biochem Sci       Date:  2017-09-17       Impact factor: 13.807

Review 2.  Genetic bases and clinical manifestations of coenzyme Q10 (CoQ 10) deficiency.

Authors:  Maria Andrea Desbats; Giada Lunardi; Mara Doimo; Eva Trevisson; Leonardo Salviati
Journal:  J Inherit Metab Dis       Date:  2014-08-05       Impact factor: 4.982

3.  The clinical heterogeneity of coenzyme Q10 deficiency results from genotypic differences in the Coq9 gene.

Authors:  Marta Luna-Sánchez; Elena Díaz-Casado; Emanuele Barca; Miguel Ángel Tejada; Ángeles Montilla-García; Enrique Javier Cobos; Germaine Escames; Dario Acuña-Castroviejo; Catarina M Quinzii; Luis Carlos López
Journal:  EMBO Mol Med       Date:  2015-05       Impact factor: 12.137

4.  Rescue of primary ubiquinone deficiency due to a novel COQ7 defect using 2,4-dihydroxybensoic acid.

Authors:  Christoph Freyer; Henrik Stranneheim; Karin Naess; Arnaud Mourier; Andrea Felser; Camilla Maffezzini; Nicole Lesko; Helene Bruhn; Martin Engvall; Rolf Wibom; Michela Barbaro; Yvonne Hinze; Måns Magnusson; Robin Andeer; Rolf H Zetterström; Ulrika von Döbeln; Anna Wredenberg; Anna Wedell
Journal:  J Med Genet       Date:  2015-06-17       Impact factor: 6.318

5.  CoQ deficiency causes disruption of mitochondrial sulfide oxidation, a new pathomechanism associated with this syndrome.

Authors:  Marta Luna-Sánchez; Agustín Hidalgo-Gutiérrez; Tatjana M Hildebrandt; Julio Chaves-Serrano; Eliana Barriocanal-Casado; Ángela Santos-Fandila; Miguel Romero; Ramy Ka Sayed; Juan Duarte; Holger Prokisch; Markus Schuelke; Felix Distelmaier; Germaine Escames; Darío Acuña-Castroviejo; Luis C López
Journal:  EMBO Mol Med       Date:  2017-01       Impact factor: 12.137

Review 6.  Impact of Chemical Analogs of 4-Hydroxybenzoic Acid on Coenzyme Q Biosynthesis: From Inhibition to Bypass of Coenzyme Q Deficiency.

Authors:  Fabien Pierrel
Journal:  Front Physiol       Date:  2017-06-22       Impact factor: 4.566

7.  CoQ10 supplementation rescues nephrotic syndrome through normalization of H2S oxidation pathway.

Authors:  Giulio Kleiner; Emanuele Barca; Marcello Ziosi; Valentina Emmanuele; Yimeng Xu; Agustin Hidalgo-Gutierrez; Changhong Qiao; Saba Tadesse; Estela Area-Gomez; Luis C Lopez; Catarina M Quinzii
Journal:  Biochim Biophys Acta Mol Basis Dis       Date:  2018-09-06       Impact factor: 6.633

8.  Coenzyme Q10 defects may be associated with a deficiency of Q10-independent mitochondrial respiratory chain complexes.

Authors:  Konstantina Fragaki; Annabelle Chaussenot; Jean-François Benoist; Samira Ait-El-Mkadem; Sylvie Bannwarth; Cécile Rouzier; Charlotte Cochaud; Véronique Paquis-Flucklinger
Journal:  Biol Res       Date:  2016-01-08       Impact factor: 5.612

Review 9.  Towards Personalized Intervention for Alzheimer's Disease.

Authors:  Xing Peng; Peiqi Xing; Xiuhui Li; Ying Qian; Fuhai Song; Zhouxian Bai; Guangchun Han; Hongxing Lei
Journal:  Genomics Proteomics Bioinformatics       Date:  2016-09-28       Impact factor: 7.691

10.  Reduction in the levels of CoQ biosynthetic proteins is related to an increase in lifespan without evidence of hepatic mitohormesis.

Authors:  María Rodríguez-Hidalgo; Marta Luna-Sánchez; Agustín Hidalgo-Gutiérrez; Eliana Barriocanal-Casado; Cristina Mascaraque; Darío Acuña-Castroviejo; Margarita Rivera; Germaine Escames; Luis C López
Journal:  Sci Rep       Date:  2018-09-18       Impact factor: 4.379

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