ArticlePloS one2010
Lrp4 regulates initiation of ureteric budding and is crucial for kidney formation--a mouse model for Cenani-Lenz syndrome.
Article in PloS one, 2010. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 33 papers.
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Who cites it
33 citing papers in PubMed, 56 citations in OpenAlex.
- Precision targeting of autoantigen-specific B cells in muscle-specific tyrosine kinase myasthenia gravis with chimeric autoantibody receptor T cells.Nature biotechnology · 2023Article
- Wnt signaling: Essential roles in osteoblast differentiation, bone metabolism and therapeutic implications for bone and skeletal disorders.Genes & diseases · 2023Review
- Balancing WNT signalling in early forebrain development: The role of LRP4 as a modulator of LRP6 function.Frontiers in cell and developmental biology · 2023Article
- Novel variants in the LRP4 underlying Cenani-Lenz Syndactyly syndrome.Journal of human genetics · 2022Article
- Review
- High Resolution Episcopic Microscopy for Qualitative and Quantitative Data in Phenotyping Altered Embryos and Adult Mice Using the New "Histo3D" System.Biomedicines · 2021Article
- Secreted Signaling Molecules at the Neuromuscular Junction in Physiology and Pathology.International journal of molecular sciences · 2021Review
- LRPs in WNT Signalling.Handbook of experimental pharmacology · 2021Review
- LRP4 promotes migration and invasion of gastric cancer under the regulation of microRNA-140-5p.Cancer biomarkers : section A of Disease markers · 2020Article
- WNT Signaling and Bone: Lessons From Skeletal Dysplasias and Disorders.Frontiers in endocrinology · 2020Review
- Deficiency of lrp4 in zebrafish and human LRP4 mutation induce aberrant activation of Jagged-Notch signaling in fin and limb development.Cellular and molecular life sciences : CMLS · 2019Article
- Fundamental Molecules and Mechanisms for Forming and Maintaining Neuromuscular Synapses.International journal of molecular sciences · 2018Review
- Multiple modes of Lrp4 function in modulation of Wnt/β-catenin signaling during tooth development.Development (Cambridge, England) · 2017Article
- Low-Density Lipoprotein Receptor-Related Proteins in Skeletal Development and Disease.Physiological reviews · 2017Review
- ApoE, ApoE Receptors, and the Synapse in Alzheimer's Disease.Trends in endocrinology and metabolism: TEM · 2017Review
- Functional Roles of the Interaction of APP and Lipoprotein Receptors.Frontiers in molecular neuroscience · 2017Review
- Polyhydramnios in Lrp4 knockout mice with bilateral kidney agenesis: Defects in the pathways of amniotic fluid clearance.Scientific reports · 2016Article
- Advances in the Molecular Genetics of Non-syndromic Syndactyly.Current genomics · 2015Article
- Article
- Disruption of Lrp4 function by genetic deletion or pharmacological blockade increases bone mass and serum sclerostin levels.Proceedings of the National Academy of Sciences of the United States of America · 2014Article
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Authors and funding
9 authors at 3 institutions in 3 countries.
Funding
Abstract
backgroundDevelopment of the kidney is initiated when the ureteric bud (UB) branches from the Wolffian duct and invades the overlying metanephric mesenchyme (MM) triggering the mesenchymal/epithelial interactions that are the basis of organ formation. Multiple signaling pathways must be integrated to ensure proper timing and location of the ureteric bud formation. METHODS AND PRINCIPAL
findingsWe have used gene targeting to create an Lrp4 null mouse line. The mutation results in early embryonic lethality with a subpenetrant phenotype of kidney agenesis. Ureteric budding is delayed with a failure to stimulate the metanephric mesenchyme in a timely manner, resulting in failure of cellular differentiation and resulting absence of kidney formation in the mouse as well as comparable malformations in humans with Cenani-Lenz syndrome.
conclusionLrp4 is a multi-functional receptor implicated in the regulation of several molecular pathways, including Wnt and Bmp signaling. Lrp4(-/-) mice show a delay in ureteric bud formation that results in unilateral or bilateral kidney agenesis. These data indicate that Lrp4 is a critical regulator of UB branching and lack of Lrp4 results in congenital kidney malformations in humans and mice.
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