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We describe here a rare case of giant intramuscular lipoma of the tongue. A 67-year-old male patient presented to our department with a large tumefaction on the tip of the tongue that he refers has started to grow about 20 years before. Clinical examination revealed a yellowish lesion, measuring no more than 5 cm in diameter, protruding from the lingual surface. Histological examination showed a non-encapsulated lipomatous tumor composed of mature adipocytes, uniform in size and shape, diffusely infiltrating striated muscle fibers of the tongue. The patient is fine with no local recurrence after 15 months follow-up period.