Phlegmasia cerulea dolens (PCD) is one of the most critical disorders of acute deep vein thrombosis in that it can cause permanent disability secondary to the compartment syndrome. Although several etiological factors have been proposed, PCD after coronary artery bypass surgery is extremely rare and its definitive pathophysiology is still under debate. We herein present a case of PCD that resulted in the compartment syndrome after coronary artery bypass surgery. Early recognition and decompression of PCD are crucial for saving the affected limbs.