Liposarcoma is one of the most common soft tissue sarcomas in adults. Atypical lipomatous tumour (ALT) rarely occurs in the head and neck region. Histopathologic grade of these tumours affect prognosis of this disease. The mainstay of treatment for ALT is surgical excision. In this article, a case of a huge ALT arising from the head and neck region and invading nearly entire left hemi-facial region is presented. Clinical and histopathologic features and therapeutic approaches related to this tumour are discussed reviewing the literature.

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