Rationale:Liposarcoma is a cancerous mesenchymal tumor and the most common soft-tissue sarcoma that starts in the adipose tissue. Liposarcoma is commonly found in lower extremities and retroperitoneum, but rarely occurs in the mediastinum.Patient concerns:A 63-year-old male was referred to our clinic with a 6-month history of chronic cough and dyspnea.Diagnose:Chest x-rays demonstrated a large mass occupying the left hemithorax. Contrast-enhanced computed tomography (CT) revealed a large mass in the anterior mediastinum, which caused the extrinsic compression of the main and left pulmonary artery and the right shift of mediastinum. Diagnosis of liposarcoma was confirmed by microscopic examination and immunohistochemistry analysis.Interventions:The patient underwent a thoracotomy for resection of the mediastinal lesion via left thoracic approach.Outcomes:The patient discharged without any complications and has been continuing to follow up in clinic without any complaints.Lessons:The primary mediastinal liposarcoma is rare, and we recommend that the liposarcoma should be considered in the differential diagnosis of a patient presenting with a mediastinal mass.

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