Lymphangioma circumscriptum on buccal mucosa: a case report

Yoo, Won Min; Oh, Seung Hoon; Rah, Dong Kyun; Park, Beyoung Yun

Abstract

Lymphangioma circumscriptum is a benign disorder of lymphatic channels. The pathologic findings consist of a collection of deep, sequestered subcutaneous lymphatic cisterns which are encircled by smooth muscle. The lymphatic cisterns are communicating with superficial clusters of vesicles through dilated lymphatic channels. The etiology of this lesion is unclear ; however, lymphatic obstruction has been suggested as a possible cause. It is characterized clinically by grouped vesicles, some of which may be filled with serosanguinous fluid. It is an unpleasant, but benign condition conventionally managed by wide local excision, which provides symptom control and often prevents recurrence. Neither long term complication nor association with squamous cell carcinoma has been previously documented. Some cases were reported following mastectomy and radiation therapy or radiation therapy for squamous cell carcinoma of the cervix. We present a 17-year-old patient with lymphangioma circumscriptum developed on buccal mucosa, which developed 3 months after innervated serratus anterior muscle free flap for functional reconstruction of cheek. We suppose that it was caused not only by lymphatic blockage resulted from wide operation but also by chronic masticating local irritation. In review of literatures, we didn't have found any report of lymphangioma circumscriptum developed on buccal mucosa.