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Yeast as a Tool for Deeper Understanding of Human Manganese-Related Diseases

Louvain Institute of Biomolecular Science and Technology, Université catholique de Louvain, B-1348 Louvain-la-Neuve, Belgium
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Genes 2019, 10(7), 545; https://doi.org/10.3390/genes10070545
Received: 12 June 2019 / Revised: 11 July 2019 / Accepted: 15 July 2019 / Published: 17 July 2019
(This article belongs to the Special Issue Humanized Yeast Models)
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Abstract

The biological importance of manganese lies in its function as a key cofactor for numerous metalloenzymes and as non-enzymatic antioxidant. Due to these two essential roles, it appears evident that disturbed manganese homeostasis may trigger the development of pathologies in humans. In this context, yeast has been extensively used over the last decades to gain insight into how cells regulate intra-organellar manganese concentrations and how human pathologies may be related to disturbed cellular manganese homeostasis. This review first summarizes how manganese homeostasis is controlled in yeast cells and how this knowledge can be extrapolated to human cells. Several manganese-related pathologies whose molecular mechanisms have been studied in yeast are then presented in the light of the function of this cation as a non-enzymatic antioxidant or as a key cofactor of metalloenzymes. In this line, we first describe the Transmembrane protein 165-Congenital Disorder of Glycosylation (TMEM165-CDG) and Friedreich ataxia pathologies. Then, due to the established connection between manganese cations and neurodegeneration, the Kufor–Rakeb syndrome and prion-related diseases are finally presented. View Full-Text
Keywords: yeast; manganese; disease; antioxidant; cofactor; TMEM165-CDG; Friedreich ataxia; neurodegeneration; Kufor-Rakeb; prion diseases yeast; manganese; disease; antioxidant; cofactor; TMEM165-CDG; Friedreich ataxia; neurodegeneration; Kufor-Rakeb; prion diseases
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This is an open access article distributed under the Creative Commons Attribution License which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited (CC BY 4.0).
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Thines, L.; Deschamps, A.; Stribny, J.; Morsomme, P. Yeast as a Tool for Deeper Understanding of Human Manganese-Related Diseases. Genes 2019, 10, 545.

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