The greatest advance in medical care of congenital cytomegalovirus (CMV) infection of recent date should be the finding that ganciclovir/valganciclovir therapy is able to improve the prognosis of symptomatic congenital CMV disease. Accordingly, the importance of early definitive diagnosis is reconfirmed, since antiviral therapy started within 30 days of life proved to be effective in those studies. Recently, an in vitro diagnostics that amplifies and measures CMV DNA in urine specimens collected within 21 days of life was developed and approved for coverage by medical insurance. Unfortunately, however, many patients with congenital CMV infection still remain undiagnosed. CMV screening targeting neonates who failed neonatal hearing screening will be useful to find those with congenital CMV infection who may benefit from early diagnosis and therapeutic intervention.