Cytomegalovirus infection is symptomatic in only 10% of cases. The most frequent findings are cholestasis and hepatosplenomegaly. Ten patients who presented neonatal cholestasis associated with cytomegalovirus infection were studied. The majority had elevation of serum aminotransferases and mild abnormality of hepatic function. The histopathologic findings were: normal, giant cell hepatitis, bile duct proliferation (confused with extrahepatic biliary atresia) and ductopenia. The clinical course of the patients varied from disappearance of the symptoms (2 cases) to death (2 cases). Because of the possibility of confusing the histologic findings with extrahepatic biliary atresia, the etiology of neonatal cholestasis, including cytomegalovirus infection, should be determined as soon as possible.