ArticleClinical journal of gastroenterology2019
Caroli syndrome: a clinical case with detailed histopathological analysis.
Article in Clinical journal of gastroenterology, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers.
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Who cites it
6 citing papers in PubMed, 11 citations in OpenAlex.
- A case of pregnancy complicated by caroli disease.BMC pregnancy and childbirth · 2025Article
- Article
- Rare variants in PKHD1 associated with Caroli syndrome: Two case reports.Molecular genetics & genomic medicine · 2022Article
- Review
- JNK signaling prevents biliary cyst formation through a CASPASE-8-dependent function of RIPK1 during aging.Proceedings of the National Academy of Sciences of the United States of America · 2021Article
- Management of Hepatolithiasis: Review of the Literature.Current gastroenterology reports · 2020Review
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Authors and funding
12 authors at 6 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Herein we present a clinical case of the Caroli syndrome caused by the compound heterozygous mutation in the PKHD1 gene. Histopathological assessment of liver detected biliary cirrhosis, numerous dilated bile ducts of various sizes, hyperplastic cholangiocytes containing a large amount of acid mucopolysaccharides, decreased ß-tubulin expression and increased proliferation of cholangiocytes. A significant proportion of hepatic tissue was composed of giant cysts lined with a single layer of cholangiocytes, containing pus and bile in its lumen and surrounded by granulation tissue. An accumulation of neutrophils in the lumen of the bile ducts was observed, as well as an infiltration of the ducts and cysts surrounding connective tissue by CD4
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