期刊
MATRIX BIOLOGY
卷 27, 期 6, 页码 547-560出版社
ELSEVIER SCIENCE BV
DOI: 10.1016/j.matbio.2008.03.002
关键词
lysyl oxidase; muscle; cartilage; neurogenesis; copper metabolism; Menkes disease
资金
- Centre National de la Recherche Scientifique
- Universite Lyon 1
- Hospices Civils de Lyon
- Association Fran aise contre les Myopathies
- European Community [LSHM-CT-2005-018960]
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. (C) 2008 Elsevier B.V. All rights reserved.
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