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Essential role for fibrillin-2 in zebrafish notochord and vascular morphogenesis.

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 daddygw1 (pfdgw1). Meiotic mapping and cloning reveal that the pfdgw1 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 pfdgw1 phenotype. Furthermore, each aspect of the pfdgw1 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.

Pubmed ID: 18816837


  • Gansner JM
  • Madsen EC
  • Mecham RP
  • Gitlin JD


Developmental dynamics : an official publication of the American Association of Anatomists

Publication Data

October 2, 2008

Associated Grants

  • Agency: NIDDK NIH HHS, Id: DK44464
  • Agency: NIGMS NIH HHS, Id: T32 GM07200
  • Agency: NICHD NIH HHS, Id: T32 HD060554
  • Agency: NICHD NIH HHS, Id: T32 HD060554-01

Mesh Terms

  • Animals
  • Animals, Genetically Modified
  • Embryo, Nonmammalian
  • Gene Expression Regulation, Developmental
  • Microfilament Proteins
  • Microscopy, Electron, Transmission
  • Morphogenesis
  • Mutation
  • Notochord
  • Phenotype
  • Protein-Lysine 6-Oxidase
  • Zebrafish