Echinococcus granulosus with primary muscular involvement is difficult to diagnose and treat, as it is rarely observed. It may lead to an acute clinical presentation that is occasionally confused with peripheral circulatory disorder findings due to pressure impacts. A 48-year-old male patient has applied to our clinic with severe pain of the right cruris and circulatory disorder that is caused by the compressive effect of the extensive intermuscular cystic lesion surrounded by the septa between the gastrocnemius and soleus muscles. The cyst was carefully drained with needle aspiration, under the guidance of ultrasonography both for histopathological diagnosis and to reduce the compressive impact of the mass. Echinococcus granulosus was identified by histological and pathological inspection. Albendazole was administered daily 400 mg (2×1). After four weeks of treatment, the cyst, together with its capsule, was surgically removed and the patients recovered clinically. Prior to mass excision of the cyst, percutaneous needle aspiration increases the efficacy of medical and surgical treatment, and may be recommended in suitable cases in order to mitigate the risk of recurrence.