ArticleInternational journal of hepatology2012
Caroli's Disease: Current Knowledge of Its Biliary Pathogenesis Obtained from an Orthologous Rat Model.
Article in International journal of hepatology, 2012. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 10 papers.
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Who cites it
10 citing papers in PubMed, 35 citations in OpenAlex.
- A new clinical classification of congenital biliary dilatation - HUAXI CBD classification.BMC pediatrics · 2024Article
- Laparoscopic hepatectomy for the treatment of Caroli's disease: a case report.Annals of surgical treatment and research · 2018Article
- Caroli's disease misdiagnosed as intraductal papillary neoplasm of the bile duct.Clinical and molecular hepatology · 2015Article
- Clinical classification of Caroli's disease: an analysis of 30 patients.HPB : the official journal of the International Hepato Pancreato Biliary Association · 2015Article
- Intrahepatic cholangiocarcinoma arising in Caroli's disease.Clinical and molecular hepatology · 2014Article
- Activation of the PI3K/mTOR pathway is involved in cystic proliferation of cholangiocytes of the PCK rat.PloS one · 2014Article
- MALDI imaging MS reveals candidate lipid markers of polycystic kidney disease.Journal of lipid research · 2013Article
- Invasive biliary mucinous cystic neoplasm: a review.HPB : the official journal of the International Hepato Pancreato Biliary Association · 2012Review
- A complex case of congenital cystic renal disease.BMJ case reports · 2012Article
- A Rare Case of Caroli's Syndrome.International journal of applied & basic medical researchArticle
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Authors and funding
3 authors at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Caroli's disease belongs to a group of hepatic fibropolycystic diseases and is a hepatic manifestation of autosomal recessive polycystic kidney disease (ARPKD). It is a congenital disorder characterized by segmental saccular dilatations of the large intrahepatic bile duct and is frequently associated with congenital hepatic fibrosis (CHF). The most viable theory explaining its pathogenesis suggests that it is related to ductal plate malformation. The development of the polycystic kidney (PCK) rat, an orthologous rodent model of Caroli's disease with CHF as well as ARPKD, has allowed the molecular pathogenesis of the disease and the therapeutic options for its treatment to be examined. The relevance of the findings of studies using PCK rats and/or the cholangiocyte cell line derived from them to the pathogenesis of human Caroli's disease is currently being analyzed. Fibrocystin/polyductin, the gene product responsible for ARPKD, is normally localized to primary cilia, and defects in the fibrocystin from primary cilia are observed in PCK cholangiocytes. Ciliopathies involving PCK cholangiocytes (cholangiociliopathies) appear to be associated with decreased intracellular calcium levels and increased cAMP concentrations, causing cholangiocyte hyperproliferation, abnormal cell matrix interactions, and altered fluid secretion, which ultimately result in bile duct dilatation. This article reviews the current knowledge about the pathogenesis of Caroli's disease with CHF, particularly focusing on studies of the mechanism responsible for the biliary dysgenesis observed in PCK rats.
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