BackgroundLipomas are benign soft tissue neoplasms of mature adipose tissue commonly occurring in the trunk or extremities. But, intraoral lipomas are rare entities which may be only noticed during routine dental examinations. Especially intramuscular lipomas on the tongue have been reported very rarely. In this study, we report a case of intramuscular lipoma on tongue, with a review of the literature from 1978 to 2019, providing data on age, gender, location, presenting symptoms, size, surgical methods, and recurrence.Case presentationA case of intramuscular lipoma occurring in tongue region in a 65-year-old male is reported. Surgical excision is the mainstay of treatment for the lesion. In order to decrease the deformity and discomfort after the excision, we tried to modify surgical technique using enveloped mucosal flap. This technique provided more comfortable healing procedure on the operative site without recurrence.ConclusionThis is a rare case of large intramuscular lipoma on tongue. Surgical excision with enveloped mucosal flap design was performed to diminish postoperative raw surface and discomfort and a 24-month follow-up showed excellent healing without any recurrence. A case of intramuscular lipoma on tongue and relevant literature reviews are presented in this study.