A 37-year-old man presented with jaundice, upper right quadrant pain, and intermittent fever with chills. Laboratory assessment showed biliary stasis, with total bilirubin of 203 µmol/L (2-20), conjugated bilirubin of 105 µmol/L, and alkaline phosphatase of 556 U/L (30-120). Markers for hepatitis A-E viruses were negative. Serology assessment for rubeola, herpes simplex virus, Epstein-Barr virus, and Toxoplasma gondii showed negative IgM antibodies. HIV serology status was negative. For cytomegalovirus, both types of antibodies (IgM and IgG) were positive, with an IgM level >300 U/mL. pp65 antigen was also detected as well as CMV DNA. Diagnostic imaging of the abdomen except the dilated common bile duct showed a normal appearance of the gallbladder, liver, pancreas, spleen, and both kidneys. To our knowledge, cytomegalovirus cholangiopathy in the absence of any other underlying disease has not been reported. Therefore, the presence of cholangiopathy in our patient is interesting from an imaging, laboratory, and clinical point of view.