Hydatid disease is a zoonotic infection caused by Echinococcus granulosus. Most frequently cysts develop in liver (75%) and lungs (15%). However, it tends to appear in different and unusual body sites. Occurrence of hydatid cyst in the head and neck region is of extreme rarity (0.5-4.7 %) even in endemic areas; only a few such cases have been reported in the literature. Here we report an unusual presentation of hydatid cyst in the neck. A 22-years-old female presented with slowly growing painless swelling in the left side of the neck since 3 years, diagnosed by Fine Needle Aspiration Cytology. FNAC yielded fluid aspirate and diagnosis of hydatid cyst was made. Later ultrasound neck demonstrated a well-defined cystic lesion suggestive of hydatid cyst. Though the site is unusual and uncommon for hydatid cyst presentation, it should be considered in the differential diagnosis of head and neck swelling for proper management.

Keywords: Echinococcus, Fine Needle Aspiration Cytology, Hydatid cyst.