ArticleProceedings of the National Academy of Sciences of the United States of America2006
Essential role of RGS-PX1/sorting nexin 13 in mouse development and regulation of endocytosis dynamics.
Article in Proceedings of the National Academy of Sciences of the United States of America, 2006. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 26 papers.
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Who cites it
26 citing papers in PubMed, 62 citations in OpenAlex.
- Survey of nucleotide-specific Rab GTPase interactions reveals multiple Rab effectors.bioRxiv : the preprint server for biology · 2026Article
- The role of endothelial-derived factors in neural tube development: implications for organoid models.Open biology · 2025Review
- Maternal Vitamin C Deficiency and Genetic Risk Factors Contribute to Congenital Defects through Dysregulation of DNA Methylation.bioRxiv : the preprint server for biology · 2025Article
- Epigenetic regulation by TET1 in gene-environmental interactions influencing susceptibility to congenital malformations.bioRxiv : the preprint server for biology · 2024Article
- Endocytosis at the maternal-fetal interface: balancing nutrient transport and pathogen defense.Frontiers in immunology · 2024Review
- Identification and analysis of short open reading frame-encoded peptides in different regions of mouse brain.iScience · 2023Article
- Sorting nexins: role in the regulation of blood pressure.The FEBS journal · 2023Review
- Structural Predictions of the SNX-RGS Proteins Suggest They Belong to a New Class of Lipid Transfer Proteins.Frontiers in cell and developmental biology · 2022Article
- Sorting Out Sorting Nexins Functions in the Nervous System in Health and Disease.Molecular neurobiology · 2021Review
- SNX-PXA-RGS-PXC Subfamily of SNXs in the Regulation of Receptor-Mediated Signaling and Membrane Trafficking.International journal of molecular sciences · 2021Review
- Rab7-Mediated Endocytosis Establishes Patterning of Wnt Activity through Inactivation of Dkk Antagonism.Cell reports · 2020Article
- Loss of Cubilin, the intrinsic factor-vitamin B12 receptor, impairs visceral endoderm endocytosis and endodermal patterning in the mouse.Scientific reports · 2019Article
- In vivo identification of GTPase interactors by mitochondrial relocalization and proximity biotinylation.eLife · 2019Article
- Discovery and Roles of ER-Endolysosomal Contact Sites in Disease.Advances in experimental medicine and biology · 2017Review
- Mdm1/Snx13 is a novel ER-endolysosomal interorganelle tethering protein.The Journal of cell biology · 2015Article
- Ubiquitinated proteins in exosomes secreted by myeloid-derived suppressor cells.Journal of proteome research · 2014Article
- Mutations in SNX14 cause a distinctive autosomal-recessive cerebellar ataxia and intellectual disability syndrome.American journal of human genetics · 2014Article
- Structural basis for different phosphoinositide specificities of the PX domains of sorting nexins regulating G-protein signaling.The Journal of biological chemistry · 2014Article
- Sorting nexin 5 of a new diagnostic marker of papillary thyroid carcinoma regulates Caspase-2.Cancer science · 2012Article
- A SNX10/V-ATPase pathway regulates ciliogenesis in vitro and in vivo.Cell research · 2012Article
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Authors and funding
9 authors at 2 institutions in 2 countries.
Funding
Abstract
RGS-PX1 (also known as sorting nexin 13) is a member of both the regulator of G protein signaling (RGS) and sorting nexin (SNX) protein families. Biochemical and cell culture studies have shown that RGS-PX1/SNX13 attenuates Galphas-mediated signaling through its RGS domain and regulates endocytic trafficking and degradation of the epidermal growth factor receptor. To understand the functions of RGS-PX1/SNX13 in vivo, we generated mice carrying targeted mutations of Snx13 and found that systemic Snx13-null mice were embryonic lethal around midgestation. Snx13-null embryos had significant overall growth retardation and defects in neural tube closure, blood vessel formation, and the formation of the placental labyrinthine layer. Moreover, the Snx13-null visceral yolk sac endoderm cells showed dramatic changes in the organization of endocytic compartments, abundant autophagic vacuoles, and abnormal localization of several endocytic markers, including megalin, a receptor for nutrients and proteins; ARH, a coat protein that binds megalin; LAMP2; and LC3. These changes suggest that Snx13-null embryos are defective in nutrient uptake and transport, which may contribute to the other developmental abnormalities observed. Taken together, our findings demonstrate an essential role for RGS-PX1/SNX13 in mouse development and provide previously undescribed insights into its cellular function in the regulation of endocytosis dynamics.
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