Intratumoral ligation is used to treat venous malformations; however, its clinical course and efficacy remain largely unknown. We report the case of a patient with a large venous malformation of the tongue who underwent successful intratumoral ligation. A 26-year-old woman presented to our clinic with a chief complaint of tongue swelling. Based on the results of imaging examinations and her medical history, a lingual venous malformation was diagnosed. The lesion was too large for surgical resection and the patient refused sclerosing therapy. We therefore carried out intratumoral ligation. The patient's postoperative course was uneventful, the lesion disappeared almost completely, and her tongue regained its normal shape and function. In conclusion, intratumoral ligation could be a useful technique for treating large orofacial venous malformations.