ArticlePediatric nephrology (Berlin, Germany)1993
Renal and biliary abnormalities in a new murine model of autosomal recessive polycystic kidney disease.
Article in Pediatric nephrology (Berlin, Germany), 1993. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 30 papers.
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Who cites it
30 citing papers in PubMed, 120 citations in OpenAlex.
- Article
- Ameliorating liver disease in an autosomal recessive polycystic kidney disease mouse model.American journal of physiology. Gastrointestinal and liver physiology · 2023Article
- Cystin genetic variants cause autosomal recessive polycystic kidney disease associated with altered Myc expression.Scientific reports · 2021Article
- Molecular Pathophysiology of Autosomal Recessive Polycystic Kidney Disease.International journal of molecular sciences · 2021Review
- Multi-parametric MRI of kidney disease progression for autosomal recessive polycystic kidney disease: mouse model and initial patient results.Pediatric research · 2021Article
- Disease Modeling To Understand the Pathomechanisms of Human Genetic Kidney Disorders.Clinical journal of the American Society of Nephrology : CJASN · 2020Review
- Tesevatinib ameliorates progression of polycystic kidney disease in rodent models of autosomal recessive polycystic kidney disease.World journal of nephrology · 2017Article
- Bicc1 Polymerization Regulates the Localization and Silencing of Bound mRNA.Molecular and cellular biology · 2015Article
- Article
- Translational research in ADPKD: lessons from animal models.Nature reviews. Nephrology · 2014Review
- The role of the cilium in normal and abnormal cell cycles: emphasis on renal cystic pathologies.Cellular and molecular life sciences : CMLS · 2013Review
- Role of genetic modifiers in an orthologous rat model of ARPKD.Physiological genomics · 2012Article
- Polycystin-1 regulates STAT activity by a dual mechanism.Proceedings of the National Academy of Sciences of the United States of America · 2011Article
- Autosomal recessive polycystic kidney disease epithelial cell model reveals multiple basolateral epidermal growth factor receptor sorting pathways.Molecular biology of the cell · 2010Article
- Polycystic liver diseases.Digestive and liver disease : official journal of the Italian Society of Gastroenterology and the Italian Association for the Study of the Liver · 2010Review
- The RNA-binding protein bicaudal C regulates polycystin 2 in the kidney by antagonizing miR-17 activity.Development (Cambridge, England) · 2010Article
- Hepato-renal pathology in pkd2ws25/- mice, an animal model of autosomal dominant polycystic kidney disease.The American journal of pathology · 2010Article
- Chronic blockade of 20-HETE synthesis reduces polycystic kidney disease in an orthologous rat model of ARPKD.American journal of physiology. Renal physiology · 2009Article
- Src inhibition ameliorates polycystic kidney disease.Journal of the American Society of Nephrology : JASN · 2008Article
- Xenopus Bicaudal-C is required for the differentiation of the amphibian pronephros.Developmental biology · 2007Article
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Authors and funding
5 authors at 2 institutions in 2 countries.
Funding
Abstract
Current models of autosomal recessive polycystic kidney disease (ARPKD) fail to demonstrate biliary abnormalities in association with renal cysts. We therefore studied a new murine model of ARPKD in which dual renal tubular and biliary epithelial abnormalities are present. Affected homozygous animals typically die 1 month postnatally in renal failure with progressively enlarged kidneys. Renal cysts shift in site from inner cortical proximal tubules at birth to collecting tubules 20 days later, as determined by segment-specific lectin binding. Increased numbers of mitosis were demonstrated in proximal and collecting tubular cysts. In addition, epithelial hyperplasia was demonstrated morphometrically in the intra- and extrahepatic biliary tract of affected animals. The number of intrahepatic biliary epithelial cells was increased by 50% on postnatal day 5 and by 100% on postnatal day 25 (P < 0.01). Despite an increased frequency of "chaotic" portal areas in mice with renal cysts, no intrahepatic cysts or shape abnormalities of the biliary lumen were detected using biliary casts and morphometry. Additionally there was nonobstructive hyperplastic dilatation of the extrahepatic biliary tract which was linked in all animals to the presence of renal cysts. The hyperplastic abnormalities in both renal and biliary epithelium make this new mouse strain a good model for the study of the dual organ cellular pathophysiology of ARPKD.
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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.