PUBLICATION
Morpholino knockdown of lysyl oxidase impairs zebrafish development, and reflects some aspects of copper metabolism disorders
- Authors
- Reynaud, C., Baas, D., Gleyzal, C., Le Guellec, D., and Sommer, P.
- ID
- ZDB-PUB-080512-9
- Date
- 2008
- Source
- Matrix biology : journal of the International Society for Matrix Biology 27(6): 547-560 (Journal)
- Registered Authors
- Baas, Dominique, Le Guellec, Dominique
- Keywords
- Lysyl oxidase, Muscle, Cartilage, Neurogenesis, Copper metabolism, Menkes disease
- MeSH Terms
-
- Amino Acid Sequence
- Animals
- Cell Line
- Copper/metabolism*
- Humans
- In Situ Hybridization
- Metabolic Diseases/enzymology*
- Molecular Sequence Data
- Neural Crest/cytology
- Oligonucleotides, Antisense/genetics
- Oligonucleotides, Antisense/metabolism*
- Protein-Lysine 6-Oxidase/genetics
- Protein-Lysine 6-Oxidase/metabolism*
- Sequence Alignment
- Zebrafish/anatomy & histology
- Zebrafish/embryology*
- Zebrafish/genetics
- Zebrafish/metabolism*
- PubMed
- 18467084 Full text @ Matrix Biol.
Citation
Reynaud, C., Baas, D., Gleyzal, C., Le Guellec, D., and Sommer, P. (2008) Morpholino knockdown of lysyl oxidase impairs zebrafish development, and reflects some aspects of copper metabolism disorders. Matrix biology : journal of the International Society for Matrix Biology. 27(6):547-560.
Abstract
Lysyl oxidase (LOX), a copper-dependent amine oxidase known in mammals to catalyze the cross-linking of collagen and elastin in the extracellular matrix, is a member of a multigenic family. Eight genes encoding lysyl oxidase isoforms have been identified in zebrafish. Recent studies have revealed a critical role for two zebrafish lysyl oxidases-like in the formation of the notochord. We now present the role of Lox in zebrafish development. lox morpholino-mediated knockdown results in a mildly undulated notochord, truncated anterior-posterior axis, tail bending and smaller head. Analyses of morphants show a complete disorganization of muscle somites and neural defects, in accordance with the lox expression pattern. Lox inhibition also induces pigment defects and pharyngeal arch deformities consistent with neural crest dysfunction. Taken together, these data reveal a role for Lox in early morphogenesis, especially in muscle development and neurogenesis, and resume some aspects of physiopathology of copper metabolism.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping