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Loss of Lrp2 in zebrafish disrupts pronephric tubular clearance but not forebrain development

Item Type:Article
Title:Loss of Lrp2 in zebrafish disrupts pronephric tubular clearance but not forebrain development
Creators Name: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.
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.
Keywords:Megalin, Forebrain Development, LDL Receptor-Related Proteins, Pronephos, Animals, Zebrafish
Source:Developmental Dynamics
ISSN:1058-8388
Publisher:Wiley-Blackwell
Volume:240
Number:6
Page Range:1567-1577
Date:June 2011
Official Publication:https://doi.org/10.1002/dvdy.22624
PubMed:View item in PubMed

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