EN
Echinococcal infection is a consequence of accidental ingestion of tapeworm eggs by humans. Liver scrutinises the initial haematogenous spread of portal blood and thus it is the most common organ involved. Isolated, primary involvement of other organs is a rarity. We describe a case of isolated orbital hydatid disease. To further add to the uniqueness of our case, two hydatid cysts were seen in our patient. The patient presented with unilateral proptosis with vision loss of the left eye since 6 months. Radiological imaging revealed two intraconal cystic lesions in the left eye. The chest radiography, abdominal sonogram and serology were negative for echinococcal disease. Surgical removal of the cysts was performed via lateral oribitotomy approach. Definitive diagnosis of Echinococcus was established on histopathology. The patient received post-operative albendazole therapy for 12 weeks. At 10 months’ follow-up there was no recurrence or evidence of hydatid disease elsewhere.