Unilateral hyperlucent lung syndrome, initially described by Swyer-James-Macleod (SJMS) in 1953, is considered a post-infectious complication of bronchiolitis occurring in childhood and is characterized by airflow obstruction and a decrease in the number and diameter of ipsilateral peripheral pulmonary vessels. It is a relatively uncommon and complex disease. In the literature, small groups of patients with this syndrome have been described. This article refers to a male patient, 64 years old, smoker for 80 pack-years, admitted for investigation of pulmonary thromboembolism, after presenting with dyspnea, tachypnea and desaturation. After performing a chest tomography, cylindrical cystic bronchiectasis on the left was revealed, affecting segmental and subsegmental bronchi, notably in the lower lobe and lingula, associated with volumetric reduction of the left lung, with deviation of the ipsilateral mediastinum, as well as pulmonary reticulation. Transthoracic echocardiography and arterial tomography angiography of the chest were also performed, where pulmonary thromboembolism was ruled out and an abnormal image was visualized with a hypertransparent area in his left lung, drawing the attention of the team responsible for the case. SJMS can be confused with asthma or pulmonary embolism due to similar symptoms, and because of this, it can result in inappropriate therapy. This study aimed to examine the clinical and imaging spectrum of a patient diagnosed with Swyer-James-Macleod in adulthood. Therefore, a brief review of the relevant literature was carried out to better understand the subject.