ArticleDisease models & mechanisms
Combined zebrafish-yeast chemical-genetic screens reveal gene-copper-nutrition interactions that modulate melanocyte pigmentation.
Article in Disease models & mechanisms. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 27 papers.
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Who cites it
27 citing papers in PubMed, 42 citations in OpenAlex.
- Circular RNA profiling uncovers regulators of early body color transition in juvenile leopard coral grouper (Plectropomus leopardus).BMC genomics · 2026Article
- Small molecule inhibitors of fungal Δ(9) fatty acid desaturase as antifungal agents againstFrontiers in cellular and infection microbiology · 2024Article
- The ionophore thiomaltol induces rapid lysosomal accumulation of copper and apoptosis in melanoma.Metallomics : integrated biometal science · 2022Article
- Role of adaptin protein complexes in intracellular trafficking and their impact on diseases.Bioengineered · 2021Article
- Zebrafish disease models in drug discovery: from preclinical modelling to clinical trials.Nature reviews. Drug discovery · 2021Review
- An FDA-Approved Drug Screen for Compounds Influencing Craniofacial Skeletal Development and Craniosynostosis.Molecular syndromology · 2019Article
- MEDNIK syndrome with a frame shift causing mutation in AP1S1 gene and literature review of the clinical features.Metabolic brain disease · 2018Review
- A phenotypic small-molecule screen identifies halogenated salicylanilides as inhibitors of fungal morphogenesis, biofilm formation and host cell invasion.Scientific reports · 2018Article
- How Surrogate and Chemical Genetics in Model Organisms Can Suggest Therapies for Human Genetic Diseases.Genetics · 2018Review
- Highly efficient and energy-conserved flocculation of copper in wastewater by pulse-alternating current.Environmental science and pollution research international · 2017Article
- Chemical screening in zebrafish for novel biological and therapeutic discovery.Methods in cell biology · 2017Review
- Systematic chemical-genetic and chemical-chemical interaction datasets for prediction of compound synergism.Scientific data · 2016Article
- Article
- Zebrafish as tools for drug discovery.Nature reviews. Drug discovery · 2015Review
- COMMD1 is linked to the WASH complex and regulates endosomal trafficking of the copper transporter ATP7A.Molecular biology of the cell · 2015Article
- 3D imaging of transition metals in the zebrafish embryo by X-ray fluorescence microtomography.Metallomics : integrated biometal science · 2014Article
- A functional screen for copper homeostasis genes identifies a pharmacologically tractable cellular system.BMC genomics · 2014Article
- Zebrafish in the sea of mineral (iron, zinc, and copper) metabolism.Frontiers in pharmacology · 2014Review
- Functional toxicology: tools to advance the future of toxicity testing.Frontiers in genetics · 2014Review
- Juxtaposition of chemical and mutation-induced developmental defects in zebrafish reveal a copper-chelating activity for kalihinol F.Chemistry & biology · 2013Article
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Authors and funding
12 authors at 7 institutions in 3 countries.
Funding
Abstract
Hypopigmentation is a feature of copper deficiency in humans, as caused by mutation of the copper (Cu(2+)) transporter ATP7A in Menkes disease, or an inability to absorb copper after gastric surgery. However, many causes of copper deficiency are unknown, and genetic polymorphisms might underlie sensitivity to suboptimal environmental copper conditions. Here, we combined phenotypic screens in zebrafish for compounds that affect copper metabolism with yeast chemical-genetic profiles to identify pathways that are sensitive to copper depletion. Yeast chemical-genetic interactions revealed that defects in intracellular trafficking pathways cause sensitivity to low-copper conditions; partial knockdown of the analogous Ap3s1 and Ap1s1 trafficking components in zebrafish sensitized developing melanocytes to hypopigmentation in low-copper environmental conditions. Because trafficking pathways are essential for copper loading into cuproproteins, our results suggest that hypomorphic alleles of trafficking components might underlie sensitivity to reduced-copper nutrient conditions. In addition, we used zebrafish-yeast screening to identify a novel target pathway in copper metabolism for the small-molecule MEK kinase inhibitor U0126. The zebrafish-yeast screening method combines the power of zebrafish as a disease model with facile genome-scale identification of chemical-genetic interactions in yeast to enable the discovery and dissection of complex multigenic interactions in disease-gene networks.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.