title: A genetic screen for zebrafish mutants with hepatic steatosis identifies a locus required for larval growth.
abstract: In a screen for zebrafish larval mutants with excessive liver lipid accumulation (hepatic steatosis), we identified harvest moon (hmn). Cytoplasmic lipid droplets, surrounded by multivesicular structures and mitochondria whose cristae appeared swollen, are seen in hmn mutant hepatocytes. Whole body triacylglycerol is increased in hmn mutant larvae. When we attempted to raise mutants, which were morphologically normal at the developmental stage that the screen was conducted, to adulthood, we observed that most hmn mutants do not survive to the juvenile period when raised. An arrest in growth occurs in the late larval period without obvious organ defects. Maternal zygotic mutants have no additional defects, suggesting that the mutation affects a late developmental process. The developmental window between embryogenesis and the metamorphosis remains under-studied, and hmn mutants might be useful for exploring the molecular and anatomic processes occurring during this transition period.
authors.affiliation: University of Utah Molecular Medicine (U2M2) Program, University of Utah School of Medicine, Salt Lake City, UT, USA.|Division of Endocrinology, Metabolism and Diabetes, Department of Internal Medicine, University of Utah School of Medicine, Salt Lake City, UT, USA.
authors.forename: Sarah E
authors.initials: SE
authors.lastname: Hugo
authors.affiliation: University of Utah Molecular Medicine (U2M2) Program, University of Utah School of Medicine, Salt Lake City, UT, USA.|Division of Endocrinology, Metabolism and Diabetes, Department of Internal Medicine, University of Utah School of Medicine, Salt Lake City, UT, USA.|Department of Biochemistry, University of Utah School of Medicine, Salt Lake City, UT, USA.|Department of Nutrition and Integrative Physiology, College of Health, University of Utah, Salt Lake City, UT, USA.
authors.forename: Amnon
authors.identifier: 0000-0003-4060-2252
authors.initials: A
authors.lastname: Schlegel
mesh_terms: D000818:Animals; D004195:Disease Models, Animal; D005234:Fatty Liver / Q000235:genetics*; D007814:Larva / Q000254:growth & development*; D008675:Metamorphosis, Biological / Q000502:physiology; D046529:Microscopy, Electron, Transmission; D009154:Mutation; D013217:Starvation / Q000235:genetics*; D015027:Zebrafish / Q000196:embryology* / Q000235:genetics*; D029961:Zebrafish Proteins / Q000235:genetics
chemical_list: D029961:Zebrafish Proteins
keywords: genetic screen; hepatic steatosis; larval staging; zebrafish