ArticlePloS one2015
A post-developmental genetic screen for zebrafish models of inherited liver disease.
Article in PloS one, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 24 papers.
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Who cites it
24 citing papers in PubMed, 39 citations in OpenAlex.
- General transcription factor IIIAa regulates the transcription of somatic 5S rRNA and influences embryonic organ development in zebrafish.Marine life science & technology · 2026Article
- Optimizing gRNA selection for high-penetrance F0 CRISPR screening for interrogating disease gene function.Nucleic acids research · 2025Article
- dldhcri3 zebrafish exhibit altered mitochondrial ultrastructure, morphology, and dysfunction partially rescued by probucol or thiamine.JCI insight · 2024Article
- Zebrafish ApoB-Containing Lipoprotein Metabolism: A Closer Look.Arteriosclerosis, thrombosis, and vascular biology · 2024Review
- Zebrafish as a Model for Cardiovascular and Metabolic Disease: The Future of Precision Medicine.Biomedicines · 2024Review
- WRN loss accelerates abnormal adipocyte metabolism in Werner syndrome.Cell & bioscience · 2024Article
- The lysosomal trafficking regulator "LYST": an 80-year traffic jam.Frontiers in immunology · 2024Review
- Local fat content determines global and local stiffness in livers with simple steatosis.FASEB bioAdvances · 2023Article
- Article
- Multi-omics analyses of early liver injury reveals cell-type-specific transcriptional and epigenomic shift.BMC genomics · 2021Article
- Transgenic fluorescent zebrafish lines that have revolutionized biomedical research.Laboratory animal research · 2021Review
- Postembryonic screen for mutations affecting spine development in zebrafish.Developmental biology · 2021Article
- Zebrafish as an Emerging Model for Dyslipidemia and Associated Diseases.Journal of lipid and atherosclerosis · 2021Review
- Genome mapping of aProceedings of the National Academy of Sciences of the United States of America · 2020Article
- Modeling Lysosomal Storage Diseases in the Zebrafish.Frontiers in molecular biosciences · 2020Review
- Primary Mammary Organoid Model of Lactation and Involution.Frontiers in cell and developmental biology · 2020Article
- On Zebrafish Disease Models and Matters of the Heart.Biomedicines · 2019Review
- 3-ketodihydrosphingosine reductase mutation induces steatosis and hepatic injury in zebrafish.Scientific reports · 2019Article
- Modeling Niemann-Pick disease type C1 in zebrafish: a robust platform forDisease models & mechanisms · 2018Article
- Zebrafish as a Model for Obesity and Diabetes.Frontiers in cell and developmental biology · 2018Review
Corrections and comments
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Authors and funding
12 authors at 3 institutions in 1 country.
Funding
Abstract
Nonalcoholic fatty liver disease (NAFLD) is one of the most common causes of chronic liver disease such as simple steatosis, nonalcoholic steatohepatitis (NASH), cirrhosis and fibrosis. However, the molecular pathogenesis and genetic variations causing NAFLD are poorly understood. The high prevalence and incidence of NAFLD suggests that genetic variations on a large number of genes might be involved in NAFLD. To identify genetic variants causing inherited liver disease, we used zebrafish as a model system for a large-scale mutant screen, and adopted a whole genome sequencing approach for rapid identification of mutated genes found in our screen. Here, we report on a forward genetic screen of ENU mutagenized zebrafish. From 250 F2 lines of ENU mutagenized zebrafish during post-developmental stages (5 to 8 days post fertilization), we identified 19 unique mutant zebrafish lines displaying visual evidence of hepatomegaly and/or steatosis with no developmental defects. Histological analysis of mutants revealed several specific phenotypes, including common steatosis, micro/macrovesicular steatosis, hepatomegaly, ballooning, and acute hepatocellular necrosis. This work has identified multiple post-developmental mutants and establishes zebrafish as a novel animal model for post-developmental inherited liver disease.
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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.