PUBLICATION
delta-sarcoglycan is necessary for early heart and muscle development in zebrafish
- Authors
- Cheng, L., Guo, X.F., Yang, X.Y., Chong, M., Cheng, J., Li, G., Gui, Y.H., and Lu, D.R.
- ID
- ZDB-PUB-060508-5
- Date
- 2006
- Source
- Biochemical and Biophysical Research Communications 344(4): 1290-1299 (Journal)
- Registered Authors
- Yang, Xueyan
- Keywords
- Sarcoglycan, Heart, Muscle, Development, Zebrafish
- MeSH Terms
-
- Animals
- Embryo, Nonmammalian/drug effects
- Embryo, Nonmammalian/metabolism
- Heart/drug effects
- Heart/embryology*
- Muscle Development/drug effects
- Muscle Development/genetics*
- Muscles/embryology
- Muscles/metabolism
- Oligodeoxyribonucleotides, Antisense/genetics
- Oligodeoxyribonucleotides, Antisense/pharmacology
- Sarcoglycans/antagonists & inhibitors
- Sarcoglycans/genetics
- Sarcoglycans/physiology*
- Somites/metabolism
- Zebrafish/embryology*
- Zebrafish/genetics
- Zebrafish/metabolism
- Zebrafish Proteins/antagonists & inhibitors
- Zebrafish Proteins/genetics
- Zebrafish Proteins/physiology*
- PubMed
- 16650823 Full text @ Biochem. Biophys. Res. Commun.
Citation
Cheng, L., Guo, X.F., Yang, X.Y., Chong, M., Cheng, J., Li, G., Gui, Y.H., and Lu, D.R. (2006) delta-sarcoglycan is necessary for early heart and muscle development in zebrafish. Biochemical and Biophysical Research Communications. 344(4):1290-1299.
Abstract
delta-sarcoglycan, one member of the sarcoglycan complex, is a very conservative muscle-specific protein exclusively expressed in the skeletal and cardiac muscles of vertebrates. Mutations in sarcoglycans are known to be involved in limb-girdle muscular dystrophy (LGMD) and dilated cardiomyopathy (DCM) in humans. To address the role of delta-sarcoglycan gene in zebrafish development, we have studied expression pattern of delta-sarcoglycan in zebrafish embryos and examined the role of delta-sarcoglycan in zebrafish embryonic development by morpholino. Strong expression of delta-sarcoglycan was observed in various muscles including those of the segment, heart, eye, jaw, pectoral fin, branchial arches, and swim bladder in zebrafish embryo. delta-sarcoglycan was also expressed in midbrain and retina. Knockdown of delta-sarcoglycan resulted in severe abnormality in both the cardiac and skeletal muscles. Some severe ones displayed serious morphological abnormality such as hypoplastic head, linear heart, very weak heartbeats, and runtish trunk, all dead within 5dpf. Whole-mount in situ hybridization analysis showed that adaxial cells and muscle pioneers were affected in delta-sarcoglycan knockdown embryos. In addition, absence of delta-sarcoglycan protein severely delayed the cardiac development and influenced the differentiation of cardiac muscle, and the cardiac left-right asymmetry was dramatically changed in morpholino-treated embryos. These data together suggest that delta-sarcoglycan plays an important role in early heart and muscle development.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping