An unusual case of Echinococcus oligarthrus infestation of the submandibular salivary gland is reported. Echinococcus oligarthrus is a rare variant of the Echinococcus species affecting humans. To the best of our knowledge only one case of submandibular hydatid cyst caused by Echinococcus oligarthrus has been reported. A 28-year-old female patient was admitted with a progressively increasing swelling in the left submandibular region of four years’ duration. There was no pulmonary or hepatic involvement. The present case of submandibular hydatid cyst caused by Echinococcus oligarthrus is of interest because of the unusual site of the disease.