Solitary intrapulmonary cystic lymphangioma in newborn or infant is an extremely rare disease. We describe a case of solitary intrapulmonary cystic lymphangioma in a 4-month-old boy with dyspnea and tachypnea. It was in the left lower lobe and type 1 congenital cystic adenomatoid malformation was suspected radiologically. The resected cyst was unilocular with a thin wall, and was 9.5cm in size. Histologically, the cyst was lined by flattened endothelial cells supported by a minimal fibrous stroma.