ArticleDevelopmental dynamics : an official publication of the American Association of Anatomists2016
Ablation of the epithelial-specific splicing factor Esrp1 results in ureteric branching defects and reduced nephron number.
Article in Developmental dynamics : an official publication of the American Association of Anatomists, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 13 papers.
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13 citing papers in PubMed, 22 citations in OpenAlex.
- MRAS: Master Regulator Analysis of Alternative Splicing.Advanced science (Weinheim, Baden-Wurttemberg, Germany) · 2025Article
- The Role of the ADAMTS18 Gene-Induced Immune Microenvironment in Mouse Kidney Development.Biomedicines · 2024Article
- Netrin 1 directs vascular patterning and maturity in the developing kidney.Development (Cambridge, England) · 2023Article
- The discovery, function, and regulation of epithelial splicing regulatory proteins (ESRP) 1 and 2.Biochemical Society transactions · 2023Article
- Characterization of alternative mRNA splicing in cultured cell populations representing progressive stages of human fetal kidney development.Scientific reports · 2022Article
- Underlying mechanisms of epithelial splicing regulatory proteins in cancer progression.Journal of molecular medicine (Berlin, Germany) · 2022Review
- New developments in the biology of fibroblast growth factors.WIREs mechanisms of disease · 2022Review
- The epithelial splicing regulator ESRP2 is epigenetically repressed by DNA hypermethylation in Wilms tumour and acts as a tumour suppressor.Molecular oncology · 2022Article
- AnDevelopment (Cambridge, England) · 2020Article
- Single-Cell RNA Sequencing Reveals mRNA Splice Isoform Switching during Kidney Development.Journal of the American Society of Nephrology : JASN · 2020Article
- Esrp1-Regulated Splicing of Arhgef11 Isoforms Is Required for Epithelial Tight Junction Integrity.Cell reports · 2018Article
- Evolutionary recruitment of flexible Esrp-dependent splicing programs into diverse embryonic morphogenetic processes.Nature communications · 2017Article
- ESRP1 Mutations Cause Hearing Loss due to Defects in Alternative Splicing that Disrupt Cochlear Development.Developmental cell · 2017Article
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Authors and funding
8 authors at 5 institutions in 1 country.
Funding
Abstract
backgroundAbnormalities in ureteric bud (UB) branching morphogenesis lead to congenital anomalies of the kidney and reduced nephron numbers associated with chronic kidney disease (CKD) and hypertension. Previous studies showed that the epithelial fibroblast growth factor receptor 2 (Fgfr2) IIIb splice variant supports ureteric morphogenesis in response to ligands from the metanephric mesenchyme during renal organogenesis. The epithelial-specific splicing regulator Esrp1 is required for expression of Fgfr2-IIIb and other epithelial-specific splice variants. Our objective was to determine whether Esrp1 is required for normal kidney development.
resultsAblation of Esrp1 in mice, alone or together with its paralog Esrp2, was associated with reduced kidney size and increased incidence of renal aplasia. Three-dimensional imaging showed that embryonic Esrp1 knockout (KO) kidneys had fewer ureteric tips and reduced nephron numbers. Analysis of alternative splicing in Esrp-null ureteric epithelial cells by RNA-Seq confirmed a splicing switch in Fgfr2 as well as numerous other transcripts.
conclusionsOur findings reveal that Esrp1-regulated splicing in ureteric epithelial cells plays an important role in renal development. Defects in Esrp1 KO kidneys likely reflect reduced and/or absent ureteric branching, leading to decreased nephron induction secondary to incorrect Fgfr2 splicing and other splicing alterations. Developmental Dynamics 245:991-1000, 2016. © 2016 The Authors. Developmental Dynamics published by Wiley Periodicals, Inc. on behalf of American Association of Anatomists.
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