Cytomegalovirus (CMV) is currently the most common cause of congenital infection and the leading infectious cause of brain damage and hearing loss in children. Perinatal CMV infection rarely causes clinical manifestations in normal individuals and usually follows a benign course in immunocompetent infants. However, ~15-25% of infected preterm infants may develop pneumonia, hepatitis or sepsis-like illness, bradycardia, hepatosplenomegaly, distended bowel, anemia, or thrombocytopenia. Bronchiolitis obliterans (BO) is a rare, fibrosing form of chronic obstructive lung disease that follows severe insults to the lower respiratory tract and results in narrowing and/or complete obliteration of the small airways. In non-transplant children, the most common form of BO is a severe lower respiratory tract infection, especially of adenovirus. We experienced a case of a 37-day-old male who was diagnosed as BO on chest computed tomography (CT) after CMV pneumonia. To our best knowledge, this is the first case of BO caused by CMV pneumonia in a healthy infant.