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Swyer-James syndrome is a radiologic entity characterized by hyperlucency of one or several lobes, or even of one lung, scarce hilar shadow on the same side as the transradiency, and barely visible arterial network on the abnormal side of the thorax. Generally, it is acquired subsequent to viral bronchopulmonary infections in child-hood. A seven year old boy was admitted to our hospital with complaining of a cough. He had been hospitalised for bronchopulmonary infections when he was one year old. During follow up, hyperlucency of the left lung was seen on his chest x-ray. However, sweat testing and bronchoscopy examinations were found normal. Physical examination showed markedly decreased breathing sounds in the left hemitorax with fine inspiratory crackles and expiratory wheezes. The chest x-ray revealed hyperlucency of the left lung. A diagnosis of Swyer James syndrome was confirmed by computerized tomographic scans of chest and perfusion and ventilation lung scintigraphy. Computerized tomography findings were hyperlucency and decreased volume of left lung and thin pulmonary artery branches. Small left lung parenchyma and decreased perfusion were found on radionuclide scans. The scan showed markedly diminished ventilation, diminished vascular flow and diminished perfusion unilaterally on the left lung. We suggest that this condition should be considered during the differential diagnosis of a patient with unilaterally hyperlucent lung.