Pulmonary vein thrombosis (PVT) is a known complication after lung transplantation but has rarely been reported after lower lobectomy or bilobectomy. We report a case of a left PVT extending into left atrium along with left hemiparesis following an uneventful left lower lobectomy in a case of adult congenital cystic adenomatoid malformation type 2 (CCAM). CCAM being rare in adults further its association with PVT following lower lobectomy makes this case an unusual interesting case.