Cystic echinococcosis (CE) caused by Echinococcus granulosus has been endemic in many parts of the world and involvement of the liver is the most frequently seen form. However, primary musculoskeletal involvement is rare. The radical treatment is surgical resection followed by anthelmintic medical therapy. In this study, a case of a 38-year-old woman with rare localization of CE in the psoas muscle at the proximal thigh is presented.