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We report herein the case of a 46-year-old woman investigated for recurrent acute cholangitis. Ultrasound, endoscopic retrograde cholangiopancreatography, and computed tomography scan revealed dilatation and multiple images suggestive of intrahepatic biliary stones in the ducts that drained segments V and VI of the liver. Endoscopic treatment was attempted unsuccessfully, and based on the severity of the last crisis of cholangitis a laparotomy was performed. A right hepatic lobectomy including segments V and VI was carried out without any complications, resulting in complete relief of symptoms. Pathological examination of the liver demonstrated the presence of worm nests in the liver parenchyma with chronic granulomatous lesions.