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Published online April 21, 2008
doi:10.1083/jcb.200708022
The Journal of Cell Biology, Vol. 181, No. 2, 381-394
The Rockefeller University Press, 0021-9525 $30.00
© 2008 Zoeller et al.
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Article

 A central function for perlecan in skeletal muscle and cardiovascular development

Jason J. Zoeller1,2, Angela McQuillan1,2, John Whitelock5, Shiu-Ying Ho3,4, and Renato V. Iozzo1,2

1 Department of Pathology, Anatomy, and Cell Biology, 2 Cancer Cell Biology and Signaling Program, 3 Department of Biochemistry and Molecular Biology, and 4 Kimmel Cancer Center, Thomas Jefferson University, Philadelphia, PA 19107
5 Graduate School of Biomedical Engineering, University of New South Wales, Sydney 2052, Australia

Correspondence to Renato V. Iozzo: iozzo{at}mail.jci.tju.edu

Perlecan's developmental functions are difficult to dissect in placental animals because perlecan disruption is embryonic lethal. In contrast to mammals, cardiovascular function is not essential for early zebrafish development because the embryos obtain adequate oxygen by diffusion. In this study, we use targeted protein depletion coupled with protein-based rescue experiments to investigate the involvement of perlecan and its C-terminal domain V/endorepellin in zebrafish development. The perlecan morphants show a severe myopathy characterized by abnormal actin filament orientation and disorganized sarcomeres, suggesting an involvement of perlecan in myopathies. In the perlecan morphants, primary intersegmental vessel sprouts, which develop through angiogenesis, fail to extend and show reduced protrusive activity. Live videomicroscopy confirms the abnormal swimming pattern caused by the myopathy and anomalous head and trunk vessel circulation. The phenotype is partially rescued by microinjection of human perlecan or endorepellin. These findings indicate that perlecan is essential for the integrity of somitic muscle and developmental angiogenesis and that endorepellin mediates most of these biological activities.

Abbreviations used in this paper: AChE, acetylcholine esterase; AChR, acetylcholine receptor; DA, dorsal aorta; DIC, differential interference contrast; DLAV, dorsal longitudinal anastomotic vessel; dpf, days postfertilization; hpf, hours postfertilization; HSPG, heparan sulfate proteoglycan; ISH, in situ hybridization; ISV, intersegmental vessel; PCV, posterior cardinal vein; SIV, subintestinal vessel.


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