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Loss of Lrp2 in zebrafish disrupts pronephric tubular clearance but not forebrain development. Dev Dyn 2011 Jun;240(6):1567-77

Date

04/02/2011

Pubmed ID

21455927

Pubmed Central ID

PMC3278082

DOI

10.1002/dvdy.22624

Scopus ID

2-s2.0-79955720762   22 Citations

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.

Author List

Kur E, Christa A, Veth KN, Gajera CR, Andrade-Navarro MA, Zhang J, Willer JR, Gregg RG, Abdelilah-Seyfried S, Bachmann S, Link BA, Hammes A, Willnow TE

Author

Brian A. Link PhD Professor in the Cell Biology, Neurobiology and Anatomy department at Medical College of Wisconsin




MESH terms used to index this publication - Major topics in bold

Animals
Animals, Genetically Modified
Body Patterning
Gene Deletion
Gene Expression Regulation, Developmental
Kidney Tubules
Low Density Lipoprotein Receptor-Related Protein-2
Metabolic Clearance Rate
Models, Biological
Phylogeny
Prosencephalon
Protein Structure, Tertiary
Signal Transduction
Zebrafish
jenkins-FCD Prod-486 e3098984f26de787f5ecab75090d0a28e7f4f7c0