Cytomegalovirus infection is widely spread globally in the form of primary infection, reactivation of infection, or reinfection with a clinical spectrum that varies from asymptomatic to severe symptoms. The infection in pregnant women needs special attention since it can cause congenital infections in infants. Clinical manifestations that often appear in infants with congenital cytomegalovirus infection include biliary atresia. Diagnosis of Cytomegalovirus infection is established based on clinical findings, positive laboratory results of anti-CMV IgM and IgG, and hepatobiliary investigation. Definitive therapy in the form of the Kasai procedure, when performed at an early age, can increase the infants' healing rate and improve their growth and development. In this case report, a baby boy aged 4 months 29 days experienced yellowing of the skin since the age of 2 months accompanied by pale stools. The anti-CMV IgM and IgG showed positive results, and the abdominal ultrasound showed no biliary atresia. However, the hepatobiliary investigation results showed narrowing of the biliary tract lumen, and the liver biopsy results showed neonatal giant cell hepatitis.