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Caroli's syndrome: a heterogeneous entity.

R K Tandon1, H Grewal, A C Anand

  • 1Department of Gastroenterology, All India Institute of Medical Sciences, New Delhi.

The American Journal of Gastroenterology
|February 1, 1990
PubMed
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Caroli's syndrome, a rare liver condition, often presents in young adults with recurrent abdominal pain. Surgical intervention for associated biliary cysts yielded satisfactory outcomes in this patient cohort.

Area of Science:

  • Hepatology
  • Gastroenterology
  • Surgical Management

Background:

  • Caroli's syndrome is a rare congenital disorder characterized by cystic dilatation of the intrahepatic bile ducts.
  • It often presents in young adults with symptoms like recurrent abdominal pain and mild jaundice.
  • Associated biliary anomalies, such as extrahepatic biliary cysts, are common.

Purpose of the Study:

  • To describe the clinical experience with adult patients diagnosed with Caroli's syndrome.
  • To evaluate the diagnostic utility of imaging modalities.
  • To assess the outcomes of surgical management for associated biliary abnormalities.

Main Methods:

  • Retrospective review of nine adult patients with Caroli's syndrome.
  • Clinical presentation, laboratory findings, and imaging results (ultrasound, cholangiography) were analyzed.

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  • Surgical procedures and their outcomes were documented.

    • Main Results:

    • The mean age of patients was 27.0 years, with a mean diagnostic delay of 12.1 years.
    • Recurrent abdominal pain was the predominant symptom (8/9 patients).
    • Six out of nine patients had associated extrahepatic biliary cysts (type IVa); surgical management involving cyst excision and biliary drainage showed satisfactory results.

    Conclusions:

    • Caroli's syndrome requires a high index of suspicion, especially in young adults with unexplained abdominal pain.
    • Ultrasound is a useful screening tool, but cholangiography is essential for diagnosis confirmation.
    • Surgical management of associated extrahepatic biliary cysts can lead to favorable outcomes.