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- Title
Essential role for fibrillin-2 in zebrafish notochord and vascular morphogenesis.
- Authors
Gansner, John M.; Madsen, Erik C.; Mecham, Robert P.; Gitlin, Jonathan D.
- Abstract
Recent studies demonstrate that lysyl oxidase cuproenzymes are critical for zebrafish notochord formation, but the molecular mechanisms of copper-dependent notochord morphogenesis are incompletely understood. We, therefore, conducted a forward genetic screen for zebrafish mutants that exhibit notochord sensitivity to lysyl oxidase inhibition, yielding a mutant with defects in notochord and vascular morphogenesis, puff daddy gw1 ( pfd gw1). Meiotic mapping and cloning reveal that the pfd gw1 phenotype results from disruption of the gene encoding the extracellular matrix protein fibrillin-2, and the spatiotemporal expression of fibrillin-2 is consistent with the pfd gw1 phenotype. Furthermore, each aspect of the pfd gw1 phenotype is recapitulated by morpholino knockdown of fibrillin-2. Taken together, the data reveal a genetic interaction between fibrillin-2 and the lysyl oxidases in notochord formation and demonstrate the importance of fibrillin-2 in specific early developmental processes in zebrafish. Developmental Dynamics 237:2844-2861, 2008. © 2008 Wiley-Liss, Inc.
- Publication
Developmental Dynamics, 2008, Vol 237, Issue 10, p2844
- ISSN
1058-8388
- Publication type
Article
- DOI
10.1002/dvdy.21705