Intrahepatic primary lithiasis is extremely rare in the Western world. The development of endoscopic technique permitted a conservative treatment for this disease. Because of the high recurrence rate after conservative treatment due to the remaining biliary stricture and the risk of incidental cholangiocarcinoma, we believe that hepatic resection is the treatment of choice of unilateral liver intrahepatic primary lithiasis. Herein we present a case affected with intrahepatic primary lithiasis localized into the left biliary system that successfully underwent left hepatic lobectomy.
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