Rationale: Primary intracranial leiomyosarcoma (LMS) is an extremely rare tumor in the central nervous system (CNS), and usually seen in immunocompromised individuals. Only a few cases of primary intracranial LMS have been documented in the literature and no study focused on their MRI findings. We reported a case of primary intracranial leiomyosarcoma in a immunocompetent patient and review its imaging features. Patient concerns: A 20-year-old female was admitted to our hospital, complaining with nausea, weight loss and progressive headache in recent 2 years. Diagnosis: The magnetic resonance imaging scan of the brain revealed a large well-defined extra-cerebral mass adherent to left temporal meninges. The mass was iso-intense on T1-weighted images (T1WI), lightly iso to hypointense on T2-weighted images (T2WI) and enhanced uniformly with contrast medium. The preoperative diagnosis is meningioma. Interventions: A left craniotomy was performed for the complete resection of the mass. Outcomes: The lesion was diagnosed via surgical histopathology and immunochemistry as leiomyosarcoma. No evidence of recurrence and complications were found in the following 13 months. Lessions: Primary intracranial leiomyosarcoma which has some special MR imaging features should be considered in the differential diagnosis of intracranial tumor.