PUBLICATION

A genetic screen for zebrafish mutants with hepatic steatosis identifies a locus required for larval growth

Authors
Hugo, S.E., Schlegel, A.
ID
ZDB-PUB-161216-3
Date
2017
Source
Journal of anatomy   230(3): 407-413 (Journal)
Registered Authors
Hugo, Sarah, Schlegel, Amnon
Keywords
genetic screen, hepatic steatosis, larval staging, zebrafish
MeSH Terms
  • Animals
  • Disease Models, Animal
  • Fatty Liver/genetics*
  • Larva/growth & development*
  • Metamorphosis, Biological/physiology
  • Microscopy, Electron, Transmission
  • Mutation
  • Starvation/genetics*
  • Zebrafish/embryology*
  • Zebrafish/genetics*
  • Zebrafish Proteins/genetics
PubMed
27976367 Full text @ J. Anat.
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.
Genes / Markers
Figures
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Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Antibodies
Orthology
Engineered Foreign Genes
Mapping