Introduction Pulmonary hydatid cysts are uncommon in non-endemic regions. Extrapulmonary intrapleural hydatid cysts are rarer still. They are unique in that they are outside the visceral pleura, but inside the parietal pleura. Their features and management needs to be discussed separately. Methods In a retrospective review conducted for intrathoracic hydatid cysts that had undergone surgical resection during last 5 years (2005 to 2009), we found four cases of extrapulmonary intrapleural hydatid cysts. Results There were two women and two men with a mean age of 35.25 years. Half of them had right sided and another half had left sided cysts. Cysts ranged from 8 to 17 cm at the longest diameter. Computed Tomography (CT) scan showed the cysts with smooth margin and calcified rims. Cysts were pleural based and showed secondary collapse of surrounding lung parenchyma. During surgery cysts were found in intrapleural space, inside the parietal pleura and outside the visceral pleura. Conclusions The extrapulmonary intrapleural hydatid cysts can grow up to any size and cause compression of lung parenchyma without invading the visceral pleura. They are usually suspected on CT scan, by lack of parenchyma around the cyst, but confirmed intra-operatively. They rarely complicate with intra-bronchial rupture or Bronchopleural fistula.
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