Rationale:Focal myositis (FM) is a very rare myopathy of unknown etiology characterized by focal enlargement within one single skeletal muscle. In particular, it occurs only involving the Sartorius muscle has never been reported.Patient concerns:A 25-year-old man was admitted to the hospital with progressive restricted left hip joint extension, left thigh discomfort and gait disturbance for 6 years.Diagnoses:Combining clinical manifestations with results of radiological and pathological examinations, it was consistent with the diagnosis of FMInterventions:The patient received a surgery under general anesthesia to release the contracted Sartorius tendon.Outcomes:The range of motion of the patient's left hip and ipsilateral knee has significantly improved as well as the discomfort of his left thigh relieved obviously after the surgery.Lessons:This case report is the first to report FM presenting as sartorius muscle contracture and the surgery is an alternative therapy for these patients.