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ZIRC
ZFIN ID: ZDB-PUB-110503-4
Loss of Lrp2 in zebrafish disrupts pronephric tubular clearance but not forebrain development
Kur, E., Christa, A., Veth, K.N., Gajera, C.R., Andrade-Navarro, M.A., Zhang, J., Willer, J.R., Gregg, R.G., Abdelilah-Seyfried, S., Bachmann, S., Link, B.A., Hammes, A., and Willnow, T.E.
Date: 2011
Source: Developmental dynamics : an official publication of the American Association of Anatomists   240(6): 1567-1577 (Journal)
Registered Authors: Abdelilah-Seyfried, Salim, Gregg, Ronald G., Link, Brian, Veth, Kerry, Willer, Jason
Keywords: megalin, forebrain development, LDL receptor-related proteins, pronephros
MeSH Terms:
  • Animals
  • Animals, Genetically Modified
  • Body Patterning/genetics
  • Body Patterning/physiology
  • Gene Deletion
  • Gene Expression Regulation, Developmental
  • Kidney Tubules/embryology
  • Kidney Tubules/metabolism*
  • Low Density Lipoprotein Receptor-Related Protein-2/chemistry
  • Low Density Lipoprotein Receptor-Related Protein-2/genetics*
  • Low Density Lipoprotein Receptor-Related Protein-2/metabolism
  • Low Density Lipoprotein Receptor-Related Protein-2/physiology
  • Metabolic Clearance Rate/genetics*
  • Models, Biological
  • Phylogeny
  • Prosencephalon/embryology*
  • Prosencephalon/metabolism
  • Protein Structure, Tertiary
  • Signal Transduction/genetics
  • Signal Transduction/physiology
  • Zebrafish/embryology*
  • Zebrafish/genetics
  • Zebrafish/metabolism
  • Zebrafish/physiology
PubMed: 21455927 Full text @ Dev. Dyn.
FIGURES
ABSTRACT
Low-density lipoprotein receptor-related protein 2 (LRP2) is a multifunctional cell surface receptor conserved from nematodes to humans. In mammals, it acts as regulator of sonic hedgehog and bone morphogenetic protein pathways in patterning of the embryonic forebrain and as a clearance receptor in the adult kidney. Little is known about activities of this LRP in other phyla. Here, we extend the functional elucidation of LRP2 to zebrafish as a model organism of receptor (dys)function. We demonstrate that expression of Lrp2 in embryonic and larval fish recapitulates the patterns seen in mammalian brain and kidney. Furthermore, we studied the consequence of receptor deficiencies in lrp2 and in lrp2b, a homologue unique to fish, using ENU mutagenesis or morpholino knockdown. While receptor-deficient zebrafish suffer from overt renal resorption deficiency, their brain development proceeds normally, suggesting evolutionary conservation of receptor functions in pronephric duct clearance but not in patterning of the teleost forebrain.
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