Abstract
A rare case of occult cytomegalovirus (CMV) cholangitis along with an updated literature review is presented. The patient had not undergone solid organ–bone marrow transplantation and had no chronic autoimmune biliary tract disorder or obvious immunodeficiency. The patient had underwent a pyloric-sparing duodenocephalopancreasectomy, and the postoperative histopathologic examination revealed multiple ulcerative lesions and an occult CMV infection. The copy number of CMV DNA was high (38,000 copies/μg DNA) in common bile duct surgical specimens measured by real-time quantitative polymerase chain reaction. CMV was not detected in blood and urine samples from the patient after full doses of intravenous ganciclovir and full doses of oral valganciclovir antiviral therapy. This report recapitulates the role of CMV in causing severe cholangiopathy in the absence of obvious immunosuppression or autoimmune disorder of the biliary tract.