{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,23]],"date-time":"2026-09-23T15:21:04Z","timestamp":1790176864235,"version":"4.1.0"},"reference-count":57,"publisher":"The Company of Biologists","license":[{"start":{"date-parts":[[2021,5,18]],"date-time":"2021-05-18T00:00:00Z","timestamp":1621296000000},"content-version":"am","delay-in-days":868,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/501100004794","name":"Centre National de la Recherche Scientifique","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100004794","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100009160","name":"Universit? Toulouse III - Paul Sabatier","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100009160","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100004431","name":"Fondation de France","doi-asserted-by":"publisher","award":["20140004817"],"award-info":[{"award-number":["20140004817"]}],"id":[{"id":"10.13039\/501100004431","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"abstract":"<jats:p>Mitochondria continually move, fuse and divide and these dynamics are essential for the proper function of these organelles. Indeed, the dynamic balance of fusion and fission of mitochondria determines their morphology and allows their immediate adaptation to energetic needs as well as preserving their integrity. As a consequence, mitochondrial fusion and fission dynamics and the proteins that control these processes, which are conserved from yeast to human, are essential and their disturbances are associated with severe human disorders, including neurodegenerative diseases. For example, mutations in OPA1, that encodes a conserved factor essential for mitochondrial fusion, lead to Optic Atrophy 1, a neurodegeneration that affects the optic nerve eventually leading to blindness. Here, by screening a collection of \u223c1,600 repurposed drugs on a fission yeast model, we identified five compounds able to efficiently prevent the lethality associated to the loss of Msp1p, the fission yeast orthologue of OPA1. One compound, hexestrol, was able to rescue both the mitochondrial fragmentation and mtDNA depletion induced by the loss of Msp1p, whereas the second, clomifene, only suppressed the mtDNA defect. Since yeast was already successfully used to identify candidate drugs to treat inherited mitochondrial diseases, this work may provide useful leads for the treatment of optic atrophies such as Optic Atrophy 1 or Leber Hereditary Optic Neuropathy.<\/jats:p>","DOI":"10.1242\/dmm.036558","type":"journal-article","created":{"date-parts":[[2019,1,18]],"date-time":"2019-01-18T16:39:11Z","timestamp":1547829551000},"source":"Crossref","is-referenced-by-count":19,"title":["A yeast-based screening assay identifies repurposed drugs that suppress mitochondrial fusion and mtDNA maintenance defects"],"prefix":"10.1242","author":[{"given":"Thomas","family":"Delerue","sequence":"first","affiliation":[{"name":"Research Center on Animal Cognition (CRCA) and Center of Developmental Biology (CBD), Center for Integrative Biology (CBI), Toulouse University, CNRS, UPS, France"},{"name":"Present address: Laboratory of Molecular Biology, National Cancer Institute, National Institutes of Health, Bethesda, MD 20892, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"D\u00e9borah","family":"Tribouillard-Tanvier","sequence":"additional","affiliation":[{"name":"Institut National de la Sant\u00e9 et de la Recherche M\u00e9dicale UMR1078; Universit\u00e9 de Bretagne Occidentale; Etablissement Fran\u00e7ais du Sang Bretagne; CHRU Brest, H\u00f4pital Morvan, Laboratoire de G\u00e9n\u00e9tique Mol\u00e9culaire, Brest, France"},{"name":"Institut de Biochimie et G\u00e9n\u00e9tique Cellulaires, CNRS UMR 5095, Universit\u00e9 de Bordeaux, 1 rue Camille Saint-Sa\u00ebns, 33077 Bordeaux cedex, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marl\u00e8ne","family":"Daloyau","sequence":"additional","affiliation":[{"name":"Research Center on Animal Cognition (CRCA) and Center of Developmental Biology (CBD), Center for Integrative Biology (CBI), Toulouse University, CNRS, UPS, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Farnoosh","family":"Khosrobakhsh","sequence":"additional","affiliation":[{"name":"Research Center on Animal Cognition (CRCA) and Center of Developmental Biology (CBD), Center for Integrative Biology (CBI), Toulouse University, CNRS, UPS, France"},{"name":"Present address: Department of Biological Science, Faculty of Science, University of Kurdistan, Sanandaj, Iran"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laurent Jean","family":"Emorine","sequence":"additional","affiliation":[{"name":"Research Center on Animal Cognition (CRCA) and Center of Developmental Biology (CBD), Center for Integrative Biology (CBI), Toulouse University, CNRS, UPS, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ga\u00eblle","family":"Friocourt","sequence":"additional","affiliation":[{"name":"Institut National de la Sant\u00e9 et de la Recherche M\u00e9dicale UMR1078; Universit\u00e9 de Bretagne Occidentale; Etablissement Fran\u00e7ais du Sang Bretagne; CHRU Brest, H\u00f4pital Morvan, Laboratoire de G\u00e9n\u00e9tique Mol\u00e9culaire, Brest, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0229-5554","authenticated-orcid":false,"given":"Pascale","family":"Belenguer","sequence":"additional","affiliation":[{"name":"Research Center on Animal Cognition (CRCA) and Center of Developmental Biology (CBD), Center for Integrative Biology (CBI), Toulouse University, CNRS, UPS, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marc","family":"Blondel","sequence":"additional","affiliation":[{"name":"Institut National de la Sant\u00e9 et de la Recherche M\u00e9dicale UMR1078; Universit\u00e9 de Bretagne Occidentale; Etablissement Fran\u00e7ais du Sang Bretagne; CHRU Brest, H\u00f4pital Morvan, Laboratoire de G\u00e9n\u00e9tique Mol\u00e9culaire, Brest, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laetitia","family":"Arnaun\u00e9-Pelloquin","sequence":"additional","affiliation":[{"name":"Research Center on Animal Cognition (CRCA) and Center of Developmental Biology (CBD), Center for Integrative Biology (CBI), Toulouse University, CNRS, UPS, France"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2019,1,1]]},"reference":[{"key":"2021042513033626400_DMM036558C1","doi-asserted-by":"crossref","first-page":"926","DOI":"10.1038\/ng.3354","article-title":"Mutations in SLC25A46, encoding a UGO1-like protein, cause an optic atrophy spectrum disorder","volume":"47","author":"Abrams","year":"2015","journal-title":"Nat. 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