ArticleDevelopment (Cambridge, England)2022
Human pluripotent stem cell-derived kidney organoids for personalized congenital and idiopathic nephrotic syndrome modeling.
Article in Development (Cambridge, England), 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 32 papers.
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Who cites it
32 citing papers in PubMed, 43 citations in OpenAlex.
- Kidney organoids as a platform to study plasminogen-induced injury and uPA-based therapies.Stem cells translational medicine · 2026Article
- Kidney organoids as models for hereditary kidney diseases: Toward precision medicine (Review).Experimental and therapeutic medicine · 2026Review
- Splice modulation of COL4A5 reinstates collagen IV assembly in an organoid model of Alport syndrome.JCI insight · 2026Article
- Vascularization and maturation of kidney organoids: a comprehensive review of technological trends and challenges.Journal of artificial organs : the official journal of the Japanese Society for Artificial Organs · 2026Review
- Urinary Podocyte Loss is Associated With Treatment Response in Patients With Primary Nephrotic Syndrome.Kidney international reports · 2026Article
- New Dimensions in Diabetic Kidney Disease Research: Advanced Organoids and Organs-on-a-Chip.BME frontiers · 2026Review
- Modeling kidney fibrosis and tubular regeneration in iPSC-derived kidney organoids.Stem cell research & therapy · 2025Article
- CellPhoneDB v5: inferring cell-cell communication from single-cell multiomics data.Nature protocols · 2025Review
- Platelets induce epithelial to mesenchymal transition in renal proximal tubular epithelial cells through TGF-β signaling pathway.Molecular medicine (Cambridge, Mass.) · 2025Article
- Kidney Organoids: Current Advances and Applications.Life (Basel, Switzerland) · 2025Review
- Podocytes in health and disease: from development to regeneration.Human cell · 2025Review
- Personalized disease recurrence modeling using iPSC-derived podocytes in patients with idiopathic nephrotic syndrome.Nephrology, dialysis, transplantation : official publication of the European Dialysis and Transplant Association - European Renal Association · 2025Article
- A kidney organoid-based readout to assess disease activity in primary and recurrent focal segmental glomerulosclerosis.Kidney international · 2025Article
- Mechanisms of podocyte injury in genetic kidney disease.Pediatric nephrology (Berlin, Germany) · 2025Review
- Calycosin ameliorates albuminuria in nephrotic syndrome by targeting Notch1/Snail pathway.BMC nephrology · 2025Article
- A genetically inducible endothelial niche enables vascularization of human kidney organoids with multilineage maturation and emergence of renin expressing cells.Kidney international · 2024Article
- Proximal tubule cell maturation rate and function are controlled by PPARα signaling in kidney organoids.Communications biology · 2024Article
- KCNJ16-depleted kidney organoids recapitulate tubulopathy and lipid recovery upon statins treatment.Stem cell research & therapy · 2024Article
- Review
- Kidney Disease Modeling with Organoids and Organs-on-Chips.Annual review of biomedical engineering · 2024Review
Corrections and comments
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Authors and funding
32 authors at 5 institutions in 3 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Nephrotic syndrome (NS) is characterized by severe proteinuria as a consequence of kidney glomerular injury due to podocyte damage. In vitro models mimicking in vivo podocyte characteristics are a prerequisite to resolve NS pathogenesis. The detailed characterization of organoid podocytes resulting from a hybrid culture protocol showed a podocyte population that resembles adult podocytes and was superior compared with 2D counterparts, based on single-cell RNA sequencing, super-resolution imaging and electron microscopy. In this study, these next-generation podocytes in kidney organoids enabled personalized idiopathic nephrotic syndrome modeling, as shown by activated slit diaphragm signaling and podocyte injury following protamine sulfate, puromycin aminonucleoside treatment and exposure to NS plasma containing pathogenic permeability factors. Organoids cultured from cells of a patient with heterozygous NPHS2 mutations showed poor NPHS2 expression and aberrant NPHS1 localization, which was reversible after genetic correction. Repaired organoids displayed increased VEGFA pathway activity and transcription factor activity known to be essential for podocyte physiology, as shown by RNA sequencing. This study shows that organoids are the preferred model of choice to study idiopathic and congenital podocytopathies.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.