Intramuscular lipoma (IML) is an uncommon tumor in the oral cavity, and characterized by infiltrating adipose tissue and muscle atrophy. Although oral IMLs are almost always solitary, we reported a rare case of IML of the tongue complicated with diffuse lipomatosis of the forearm. A 58-year-old woman was referred to our department with a painless swelling of the left tongue that had been present for 6 months. Clinical examination revealed a 35mm×30mm×20mm well-defined, elastic soft mass in the anterior left region of the tongue border. The lesion was removed under general anesthesia via the left margin of the tongue. No signs of recurrence have been observed 15 months after surgery.
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