Hemangiolymphangioma of the buccal mucosa in a 44-year-old patient, a case-report and literature review of the condition

OBJECTIVES Hemangiolymphangiomas are rare vascular malformations or de-velopmental anomalies with a propensity to invade underlying tissues and to recur locally. The aim of the present article is to report and discuss a case of hemangiolymphangioma of the oral cavity. MATERIALS AND METHODS A 44-year-old man was referred to the Department of Dentistry, University of Eastern Piedmont, Italy, with the chief complaint of a painless swelling in the right buc-cal mucosa, that had been present for years. Intra-oral examination revealed a painless violaceous nodular lesion in correspondence of the right buccal mucosa. Histo-pathological examination revealed numerous dilated lym-phatics and large, dilated ves-sels lined by normal-appearing endothelial cells and containing inside erythrocytes and protein-aceous eosinophilic material containing lymph and channels with blood cells. A definitive diagnosis of hemangio-lymphangioma was made. DISCUSSION Hemangiolymphangioma occur-ring in the oral cavity is extremely rare as the most frequent sites are the neck’s anterior and posterior cervical triangle. Microscopically, hemangiolymph-angiomas do not show active cel-lular proliferation, but appear as subepithelial enlarged channels. Hemangiolymphangiomas are mixed lesions, and they may present positive staining for some lymphatic markers (such as CD31 and D2-40), that supports their lymphatic derivation. CONCLUSIONS Hemangiolymphangioma is a rare lesion in the oral cavity. All sus-pected cases should be thorough-ly and carefully assessed to rule out bony, intracranial and system-ic involvements. CLINICAL SIGNIFICANCE Complete surgical excision is the treatment of choice for these lesions whenever possi-ble, although several factors in-cluding the age of the patient and the size and extent of the lesions should be taken in con-sideration.