ArticleOxidative medicine and cellular longevity2015
A Yeast/Drosophila Screen to Identify New Compounds Overcoming Frataxin Deficiency.
Article in Oxidative medicine and cellular longevity, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 10 papers.
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Who cites it
10 citing papers in PubMed.
- Hypomagnetic Fields Influence the Developmental Duration, Fecundity and Temperature Stress Resistance ofBiology · 2026Article
- Wings of Discovery: UsingCells · 2025Review
- Animal Models of Neurodegenerative Disease: Recent Advances in Fly Highlight Innovative Approaches to Drug Discovery.Frontiers in molecular neuroscience · 2022Review
- Delayed Infliximab Treatment Affects the Outcomes of Patients With Crohn's Disease During the COVID-19 Epidemic in China: A Propensity Score-Matched Analysis.Frontiers in medicine · 2021Article
- Drug screening in Drosophila; why, when, and when not?Wiley interdisciplinary reviews. Developmental biology · 2019Review
- Nutrigenomics as a tool to study the impact of diet on aging and age-related diseases: theGenes & nutrition · 2019Review
- Identification of cardioprotective drugs by medium-scaleDisease models & mechanisms · 2018Article
- Impact ofInternational journal of molecular sciences · 2018Review
- Review
- Drug Discovery in Fish, Flies, and Worms.ILAR journal · 2016Review
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Authors and funding
9 authors.
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No grant is acknowledged in the PubMed record.
Abstract
Friedreich's ataxia (FA) is a rare neurodegenerative disease which is very debilitating for the patients who progressively lose their autonomy. The lack of efficient therapeutic treatment of the disease strongly argues for urgent need to search for new active compounds that may stop the progression of the disease or prevent the appearance of the symptoms when the genetic defect is diagnosed early enough. In the present study, we used a yeast strain with a deletion of the frataxin homologue gene as a model of FA cells in a primary screen of two chemical libraries, a fraction of the French National Chemical Library (5500 compounds) and the Prestwick collection (880 compounds). We ran a secondary screen on Drosophila melanogaster flies expressing reduced levels of frataxin during larval development. Half of the compounds selected in yeast appeared to be active in flies in this developmental paradigm, and one of the two compounds with highest activities in this assay partially rescued the heart dilatation phenotype resulting from heart specific depletion of frataxin. The unique complementarity of these two frataxin-deficient models, unicellular and multicellular, appears to be very efficient to select new compounds with improved selectivity, bringing significant perspectives towards improvements in FA therapy.
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