A 70-yr-old woman presented with nonspecific complaints and had findings of a cholestatic liver enzyme pattern and a dilated left intrahepatic ductal system by radiographic imaging. Endoscopic retrograde cholangiopancreatography revealed multiple irregular flat stones and a fixed filling defect of the left hepatic duct. At the time of surgery a stonelike lesion of this ductal orifice was found along with a miliary pattern of infection of the left liver lobe. Histologic and microbiologic studies confirmed an infection by Blastomyces dermatitidis. Therapy with amphotericin B resulted in eradication of this infection. Despite their rarity, fungal infections of the biliary tree need to be considered in the differential diagnosis of biliary tract disease in the elderly and immunocompromised patient.