PUBLICATION
Fer1l6 Is Essential for the Development of Vertebrate Muscle Tissue in Zebrafish
- Authors
- Bonventre, J.A., Holman, C., Manchanda, A., Codding, S.J., Chau, T., Huegel, J., Barton, C., Tanguay, R., Johnson, C.P.
- ID
- ZDB-PUB-181206-11
- Date
- 2018
- Source
- Molecular biology of the cell 30(3): 293-301 (Journal)
- Registered Authors
- Barton, Carrie, Tanguay, Robyn L.
- Keywords
- none
- MeSH Terms
-
- Animals
- Cells, Cultured
- Gene Expression Regulation, Developmental/drug effects
- Membrane Lipids/metabolism
- Mice
- Morpholinos/pharmacology
- Muscle Development*/drug effects
- Muscle Development*/genetics
- Muscles/embryology*
- Muscles/metabolism*
- Muscles/ultrastructure
- Mutation/genetics
- Qa-SNARE Proteins/metabolism
- Transcription, Genetic/drug effects
- Vesicular Transport Proteins/genetics
- Vesicular Transport Proteins/physiology*
- Vinculin/metabolism
- Zebrafish/embryology*
- Zebrafish/genetics
- Zebrafish Proteins/genetics
- Zebrafish Proteins/physiology*
- PubMed
- 30516436 Full text @ Mol. Biol. Cell
Citation
Bonventre, J.A., Holman, C., Manchanda, A., Codding, S.J., Chau, T., Huegel, J., Barton, C., Tanguay, R., Johnson, C.P. (2018) Fer1l6 Is Essential for the Development of Vertebrate Muscle Tissue in Zebrafish. Molecular biology of the cell. 30(3):293-301.
Abstract
The precise spatial and temporal expression of genes is essential for proper organismal development. Despite their importance however, many developmental genes have yet to be identified. We have determined that Fer1L6, a member of the ferlin family of genes, is a novel factor in zebrafish development. We find that Fer1L6 is expressed broadly in the trunk and head of zebrafish larvae and is more restricted to gills and female gonads in adult zebrafish. Using both genetic mutant and morpholino knockdown models, we found that loss of Fer1L6 led to deformation of striated muscle tissues, delayed development of the heart, and high morbidity. Further, expression of genes associated with muscle cell proliferation and differentiation were affected. Fer1L6 was also detected in the C2C12 cell line, and unlike other ferlin homologues, we found Fer1L6 expression was independent of the myoblast-to-myotube transition. Lastly, analysis of cell and recombinant protein-based assays indicate that Fer1L6 colocalizes with syntaxin 4 and vinculin, and that the putative C2 domains interact with lipid membranes. We conclude that Fer1L6 has diverged from other vertebrate ferlins to play an essential role in zebrafish skeletal and cardiac muscle development. Movie S1 Movie S1 Representative video of z-slices of sarcomere myosin labeled 4 dpf heart from control sample. Movie S2 Movie S2 Representative video of z-slices of sarcomere myosin labeled 4 dpf heart from Fer1L6 morphant sample.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping