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Unilateral pulmonary agenesis is a rare congenital abnormality that may be associated with other anomalies. Diagnosis is usually made in the neonatal period or early childhood, and it is very rare in adults. A 39-year-old female patient with increasing shortness of breath, cough, and sputum production was admitted to our hospital with complaints of coughing up blood. There were no breath sounds from the left lung in the physical examination. A postero- anterior chest X-ray revealed displacement of the mediastinum and diaphragm to the left, and the space normally occupied by the left lung was almost completely replaced with a homogenous opacity. A diagnosis of left pulmonary agenesis was supported by a computed tomography scan of the chest, pulmonary magnetic resonance (MR) angiography and bronchoscopy. This case is a rare example of pulmonary agenesis diagnosed in adulthood. [ABSTRACT FROM AUTHOR]