ArticleInternational journal of molecular sciences2021
A 3D Renal Proximal Tubule on Chip Model Phenocopies Lowe Syndrome and Dent II Disease Tubulopathy.
Article in International journal of molecular sciences, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 11 papers, 1 of them a synthesis that pooled it.
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Who cites it
11 citing papers in PubMed, 1 synthesis or guideline pooled it, 20 citations in OpenAlex.
- A systematic review of kidney-on-a-chip-based models to study human renal (patho-)physiology.Disease models & mechanisms · 2023Pooled it
- Synergistic innovation in organ-on-a-chip and organoid technologies: reshaping the future of disease modeling, drug development, and precision medicine.Protein & cell · 2026Review
- Bridging the Gap Between Static Histology and Dynamic Organ-on-a-Chip Models.Pathophysiology : the official journal of the International Society for Pathophysiology · 2026Review
- Microfluidic Systems to Mimic the Blood-Brain Barrier: from Market to Engineering Challenges and Perspectives.ACS biomaterials science & engineering · 2025Review
- Organoids-on-Chips Technology: Unveiling New Perspectives in Rare-Disease Research.International journal of molecular sciences · 2025Review
- My cells, my model: immune-competent autologous organ-on-chip systems as a new paradigm in precision medicine.Frontiers in immunology · 2025Review
- Kidney Disease Modeling with Organoids and Organs-on-Chips.Annual review of biomedical engineering · 2024Review
- Revolutionizing nephrology research: expanding horizons with kidney-on-a-chip and beyond.Frontiers in bioengineering and biotechnology · 2024Review
- Analysis of reproducibility and robustness of a renal proximal tubule microphysiological system OrganoPlate 3-lane 40 for in vitro studies of drug transport and toxicity.Toxicological sciences : an official journal of the Society of Toxicology · 2023Article
- Organs-on-chip technology: a tool to tackle genetic kidney diseases.Pediatric nephrology (Berlin, Germany) · 2022Review
- Human organs-on-chips for disease modelling, drug development and personalized medicine.Nature reviews. Genetics · 2022Review
Corrections and comments
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Authors and funding
10 authors at 4 institutions in 3 countries.
Funding
Abstract
Lowe syndrome and Dent II disease are X-linked monogenetic diseases characterised by a renal reabsorption defect in the proximal tubules and caused by mutations in the OCRL gene, which codes for an inositol-5-phosphatase. The life expectancy of patients suffering from Lowe syndrome is largely reduced because of the development of chronic kidney disease and related complications. There is a need for physiological human in vitro models for Lowe syndrome/Dent II disease to study the underpinning disease mechanisms and to identify and characterise potential drugs and drug targets. Here, we describe a proximal tubule organ on chip model combining a 3D tubule architecture with fluid flow shear stress that phenocopies hallmarks of Lowe syndrome/Dent II disease. We demonstrate the high suitability of our in vitro model for drug target validation. Furthermore, using this model, we demonstrate that proximal tubule cells lacking OCRL expression upregulate markers typical for epithelial-mesenchymal transition (EMT), including the transcription factor SNAI2/Slug, and show increased collagen expression and deposition, which potentially contributes to interstitial fibrosis and disease progression as observed in Lowe syndrome and Dent II disease.
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Registered trials
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