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Ann Pathol. 2009 Feb;29(1):24-7. doi: 10.1016/j.annpat.2008.09.048. Epub 2009 Feb 5.

[Cholangiocarcinomas developing on multiple bile duct hamartomas syndrome].

[Article in French]

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  • 1Service d'anatomie pathologique, de chirurgie digestive et de gastroentérologie et hépatologie, hôpital Nord-Laënnec, CHU de Nantes, boulevard Jacques-Monod, 44800 Saint-Herblain, France.


Two cases of cholangiocarcinoma arising in a background of multiple bile duct hamartomas are reported. In each case, the clinical and radiological investigations showed a liver tumor with no other concomitant disease. Tumors were resected, and microscopic examination revealed in one case a hilar cholangiocarcinoma, in the other case a peripheric cholangiocarcinoma, both being well-differentiated. It revealed also numerous bile duct hamartomas scattered throughout the liver. In one case, some bile duct hamartomas showed a gradual morphologic transition from benign to dysplastic and neoplastic epithelium. Patients with multiple bile duct hamartomas may have an increased risk of developing cholangiocarcinomas.

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