Hydatid cyst (HC) in mediastinum is very rare. To the best of our knowledge, a case with multiple HCs in mediastinum is not reported already. We herein report a case of multiple HCs of the mediastinum and liver in a - 50 year-old woman presented with chest pain, cough and dyspnea for about two years. She had been treated for tuberculosis for 20 years. Chest CT scan showed multiple cysts in posterior mediastinum and one cyst in left lobe of liver. Via right posterolateral thoracotomy, multiple cysts were excised in mediastinum. And then, hepatic left lobe cyst was removed trans-diaphragmatically. Histopathologic examination confirmed HCs. Despite its rarity, primary HCs should be considered in the differential diagnosis of mediastinal multiple cystic lesions in endemic regions.
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