Hydatid cyst is a parasitic infection of the human caused by the larval stage of Echinococcus. Hydatid cyst of the head and neck region is uncommon and the involvement of salivary glands, especially the submandibular gland is very rare. We report a case of isolated hydatid cyst of left submandibular gland. An 11-year-old female presented with a slowly growing painless swelling of left submandibular region since 4 months. Fine needle aspiration cytology yielded fluid aspirate and smear showed fragment of laminated membrane, suggestive of HC. FNAC was suggestive of lymphatic cyst of left submandibular region .Ultrasonography of neck was suggestive of a cystic mass with internal debris. The cyst was excised as a whole along with submandibular gland and it was sent for histopathological examination. Pathological examination confirmed the swelling to be a hydatid cyst of the left submandibular gland.
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