AbstractObjective:We report a case of lymphangioma circumscriptum of the tongue, a very rare site of occurrence, which was successfully managed with intralesional bleomycin therapy.Method:We present a case report and review of available literature regarding lymphangioma circumscriptum of the tongue and the role of bleomycin therapy.Results:The patient was a 19-year-old man with a long-standing lesion involving the tongue, who presented with spontaneous, episodic bleeding over the previous few months. A diagnosis of lymphangioma circumscriptum was established on biopsy. Intralesional bleomycin injection resulted in successful resolution, and the patient remained asymptomatic over more than one year's follow up.Conclusion:Lymphangioma circumscriptum is usually seen in the extremities and genitals. This case had a very rare site of occurrence, the tongue, and was successfully managed with conservative treatment, using intralesional bleomycin alone.