{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,5]],"date-time":"2026-04-05T07:35:27Z","timestamp":1775374527494,"version":"3.50.1"},"reference-count":74,"publisher":"Oxford University Press (OUP)","issue":"1","license":[{"start":{"date-parts":[[2019,9,27]],"date-time":"2019-09-27T00:00:00Z","timestamp":1569542400000},"content-version":"vor","delay-in-days":269,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"funder":[{"name":"ATOZ foundation"},{"DOI":"10.13039\/100016358","name":"Fondation du P\u00e9lican de Mie et Pierre Hippert-Faber","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100016358","id-type":"DOI","asserted-by":"crossref"}]},{"name":"Doctoral School in Systems and Molecular Biomedicine at the University of Luxembourg"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2019,1,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>Mutations in ATP13A2 (PARK9) are causally linked to the rare neurodegenerative disorders Kufor-Rakeb syndrome, hereditary spastic paraplegia and neuronal ceroid lipofuscinosis. This suggests that ATP13A2, a lysosomal cation-transporting ATPase, plays a crucial role in neuronal cells. The heterogeneity of the clinical spectrum of ATP13A2-associated disorders is not yet well understood and currently, these diseases remain without effective treatment. Interestingly, ATP13A2 is widely conserved among eukaryotes, and the yeast model for ATP13A2 deficiency was the first to indicate a role in heavy metal homeostasis, which was later confirmed in human cells. In this study, we show that the deletion of YPK9 (the yeast orthologue of ATP13A2) in Saccharomyces cerevisiae leads to growth impairment in the presence of Zn2+, Mn2+, Co2+ and Ni2+, with the strongest phenotype being observed in the presence of zinc. Using the ypk9\u0394 mutant, we developed a high-throughput growth rescue screen based on the Zn2+ sensitivity phenotype. Screening of two libraries of Food and Drug Administration-approved drugs identified 11 compounds that rescued growth. Subsequently, we generated a zebrafish model for ATP13A2 deficiency and found that both partial and complete loss of atp13a2 function led to increased sensitivity to Mn2+. Based on this phenotype, we confirmed two of the drugs found in the yeast screen to also exert a rescue effect in zebrafish\u2014N-acetylcysteine, a potent antioxidant, and furaltadone, a nitrofuran antibiotic. This study further supports that combining the high-throughput screening capacity of yeast with rapid in vivo drug testing in zebrafish can represent an efficient drug repurposing strategy in the context of rare inherited disorders involving conserved genes. This work also deepens the understanding of the role of ATP13A2 in heavy metal detoxification and provides a new in vivo model for investigating ATP13A2 deficiency.<\/jats:p>","DOI":"10.1093\/braincomms\/fcz019","type":"journal-article","created":{"date-parts":[[2019,9,27]],"date-time":"2019-09-27T13:11:46Z","timestamp":1569589906000},"source":"Crossref","is-referenced-by-count":16,"title":["Phenotypic assays in yeast and zebrafish reveal drugs that rescue ATP13A2 deficiency"],"prefix":"10.1093","volume":"1","author":[{"given":"Ursula","family":"Heins-Marroquin","sequence":"first","affiliation":[{"name":"Luxembourg Centre for Systems Biomedicine, University of Luxembourg, L-4367 Belvaux, Luxembourg"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Paul P","family":"Jung","sequence":"additional","affiliation":[{"name":"Luxembourg Centre for Systems Biomedicine, University of Luxembourg, L-4367 Belvaux, Luxembourg"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Maria Lorena","family":"Cordero-Maldonado","sequence":"additional","affiliation":[{"name":"Luxembourg Centre for Systems Biomedicine, University of Luxembourg, L-4367 Belvaux, Luxembourg"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Alexander D","family":"Crawford","sequence":"additional","affiliation":[{"name":"Luxembourg Centre for Systems Biomedicine, University of Luxembourg, L-4367 Belvaux, Luxembourg"},{"name":"Faculty of Veterinary Medicine, Norwegian University of Life Sciences, 0454 Oslo, Norway"},{"name":"Institute for Orphan Drug Discovery, Bremer Innovations- und Technologiezentrum, 28359 Bremen, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7143-0719","authenticated-orcid":false,"given":"Carole L","family":"Linster","sequence":"additional","affiliation":[{"name":"Luxembourg Centre for Systems Biomedicine, University of Luxembourg, L-4367 Belvaux, Luxembourg"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2019,9,27]]},"reference":[{"key":"2020081108572898300_fcz019-B1","doi-asserted-by":"crossref","first-page":"1515","DOI":"10.1212\/WNL.57.8.1515","article-title":"Controlled trial of N-acetylcysteine for patients with probable Alzheimer\u2019s disease","volume":"57","author":"Adair","year":"2001","journal-title":"Neurology"},{"key":"2020081108572898300_fcz019-B2","doi-asserted-by":"crossref","first-page":"913","DOI":"10.1002\/yea.1502","article-title":"A suite of Gateway cloning vectors for high-throughput genetic analysis in Saccharomyces cerevisiae","volume":"24","author":"Alberti","year":"2007","journal-title":"Yeast"},{"key":"2020081108572898300_fcz019-B3","doi-asserted-by":"crossref","first-page":"1239","DOI":"10.1242\/dmm.016683","article-title":"A zebrafish model of manganism reveals reversible and treatable symptoms that are independent 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