Challenging Cytologic Diagnosis of Intrahepatic Cholangiocarcinoma Arising in Ductal Plate Malformation

This case describes a rare intrahepatic cholangiocarcinoma arising in a background of ductal plate malformation/Von Meyenburg complex. Predominant sampling of benign ductal proliferations obscured a focal malignant component, highlighting the importance of recognizing subtle cytomorphologic atypia, correlating pathologic findings with imaging, and utilizing ancillary studies to establish the diagnosis.