Due to acute renal failure, abdominal computed tomography scan (CT scan) was done without contrast injection. Results were no conclusive concerning a suspicious intra hepatic lesion (7.9 × 6.4 cm) along with signs of chronic liver injury. The patient underwent an endoscopic retrograde cholangiopancreatography (ERCP) that showed left and right hepatic duct stenosis. Thick purulent material was evacuated from narrowing site and bile was collected before and after BDB and sent for cytological examination. Bile cytopathologic findings Bile aspiration specimen was performed as previously described by our team. Bile smear was hypercellular, showing both highly isolated malignant cells and tridimensional clusters. The tumor cells were large, polygonal with an abundant granular cytoplasm, an enlarged nucleus and prominent nucleoli along with multiple naked nuclei. The presence of cytoplasmic bile pigments was observed. The background was rich in neutrophils with some benign appearing bile duct epithelium. Hematoxylin-eosin-safron staining on cell block (CB) showed histologically sheets malignant hepatocytes with few intracytoplasmic bile pigments and Mallory-Denk bodies. Immunohistochemical study performed on CB confirmed the presence of hepatocellular differenciation with Hepatocyte and Arginase antibodies positivity and the malignant nature with Glypican 3 positivity.