A patient with culturally confirmed congenital cytomegalic inclusion disease was treated at 12 days of age with Floxuridine (FUDR). FUDR was intravenously administered in a dose of 0.5 mg/kg/day as a single continuous 12-hour infusion each day for 5 consecutive days. Positive cultures for cytomegalovirus were obtained from urine and saliva throughout the course of therapy as well as from cerebrospinal fluid and a hip joint effusion at the conclusion of therapy. No therapeutic effect of FUDR was demonstrable clinically and no toxic drug effects were noted.