Introduction The preferred localization of human echinococcosis is the liver and lungs, which account for 85% of cases. Primary musculoskeletal hydatidosis are seldom reported in literature and their incidence is unknown. Primary solitary intramuscular hydatid disease is rare, even in countries in which echinococcosis is endemic and accounts for 1% of all human echinococcosis sites. Case presentation 37-year-old male farmer presented with primary hydatid cyst of the upper medial side of right thigh (Inside adductor magnus muscle) who was managed successfully by preoperative and postoperative dual treatment of albendazole together with surgery. Discussion This site of localization has not been reported previously. It is essential to establish definitive preoperative diagnosis of skeletal muscle hydatid cysts. This contraindicates certain treatment options like marginal excision or incisional biopsy due to the likelihood of dissemination and anaphylactic shock on spillage. Pericystectomy remains treatment of choice in musculoskeletal hydatid cysts. Conclusion Hydatid disease should be included in the differential diagnosis of muscular masses, regardless of its location, especially in endemic areas.
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