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Fishing in the Cell Powerhouse: Zebrafish as A Tool for Exploration of Mitochondrial Defects Affecting the Nervous System

Molecular Medicine, IRCCS Stella Maris, Via dei Giacinti 2, 56028 Pisa, Italy
Laboratory of General Physiology, Department of Biological and Environmental Sciences and Technologies, University of Salento, Via Provinciale Lecce-Monteroni, 73100 Lecce, Italy
Department of Basic Medical Sciences, Neurosciences and Sense Organs, University of Bari ‘Aldo Moro’, Piazza Giulio Cesare 11, 70124 Bari, Italy
Department of Veterinary Sciences, University of Pisa, viale delle Piagge 2, 56124 Pisa, Italy
Authors to whom correspondence should be addressed.
These authors contributed equally to this work
Int. J. Mol. Sci. 2019, 20(10), 2409;
Received: 16 April 2019 / Revised: 11 May 2019 / Accepted: 13 May 2019 / Published: 15 May 2019
PDF [727 KB, uploaded 15 May 2019]


The zebrafish (Danio rerio) is a small vertebrate ideally suited to the modeling of human diseases. Large numbers of genetic alterations have now been modeled and could be used to study organ development by means of a genetic approach. To date, limited attention has been paid to the possible use of the zebrafish toolbox in studying human mitochondrial disorders affecting the nervous system. Here, we review the pertinent scientific literature discussing the use of zebrafish in modeling gene mutations involved in mitochondria-related neurological human diseases. A critical analysis of the literature suggests that the zebrafish not only lends itself to exploration of the pathological consequences of mitochondrial energy output on the nervous system but could also serve as an attractive platform for future drugs in an as yet untreatable category of human disorders. View Full-Text
Keywords: zebrafish; mitochondria; nervous system development; neurodegenerative conditions zebrafish; mitochondria; nervous system development; neurodegenerative conditions

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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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Fichi, G.; Naef, V.; Barca, A.; Longo, G.; Fronte, B.; Verri, T.; Santorelli, F.M.; Marchese, M.; Petruzzella, V. Fishing in the Cell Powerhouse: Zebrafish as A Tool for Exploration of Mitochondrial Defects Affecting the Nervous System. Int. J. Mol. Sci. 2019, 20, 2409.

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