Echinococcosis is endemic in Mediterranean regions and is found primarily in the liver. Biliary fistula is a common complication, but major biliary duct involvement is very rare, and occurs in 0.47% of patients with hepatic hydatid disease. Cyst rupture causing secondary peritoneal hydatidosis is a rare but serious complication. We report the case of a 27-year-old man with multiple peritoneal and hepatic hydatid cysts. The patient came to our attention with cholestatic jaundice. Imaging exams showed numerous peritoneal cysts and massive hydatid disease of the liver, which involved the hepatic confluence, with destruction of the right hepatic duct and fistula formation to the left hepatic duct. The patient was treated with pre-operative albendazole therapy and radical surgery, which consisted of resection of all peritoneal cysts and extended right hepatectomy with biliary reconstruction. No recurrence was seen on CT investigations on the 12th month following surgery. Radical surgical approach remains the treatment of choice.
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