Squamous Cell Carcinoma Arising in a Port-Wine Stain with a Remote History of Cryosurgery

Abstract

The authors have indicated no significant interest with commercial supporters. P ort-wine stain (PWS) is a benign vascular malformation consisting of ectatic blood vessels situated predominantly in the superficial dermis, with a reported incidence of 0.3% of the population. 1 Currently developed laser treatment achieves observable lightening of PWS by reducing the number and size of the abnormal vessels without notable scar formation. 2 Previously applied methods, however, including surgical excision and grafting, radiation, dermabrasion, and cryotherapy, had produced unfavorable outcomes.The development of malignancy within a PWS lesion is a rare condition, and the mechanism for malignant changes in PWSs is not fully understood. MaganaGarcia and Magana-Lozano 3 proposed that tumor development might involve an oncogenic factor produced by the abnormal vessels that makes the overlying epidermis more susceptible to ultraviolet light. In contrast, some authors have suggested that prior treatments such as radiation are involved in the development of malignant skin lesions. 4Although more than 20 cases of basal cell carcinoma associated with PWSs have been previously reported, 3-6 association of squamous cell carcinoma (SCC) is extremely rare, with only 3 cases 7-9 reported to date in the English literature. We present herein a case of SCC developing within a PWS with a remote history of cryosurgery and discuss the possible etiology. Case ReportA 69-year-old male patient visited our clinic in November 2004 with a chronic ulceration within the boundary of a PWS involving the V1 to V2 area of the right trigeminal nerve. The history of the case was carefully taken from not only himself but also his older sister. His history revealed that he had undergone cryosurgery several times when he was 5 to 7 years old. He did not receive any other therapy including radiotherapy, laser therapy, and surgical excision. The ulcer (3 cm in diameter), of 7 months duration, was associated with a surrounding atrophic scar (Figure 1). A biopsy specimen taken from the margin of the ulcer showed histologic features of well-differentiated SCC. No distinct metastases were detected by systemic evaluation.With these findings, we performed local wide excision involving all scar tissue and the surrounding PWS in his right cheek on December 20, 2004. After affirmation of tumor-free margins on the basis of the pathologic report of frozen sections, a free deltopectoral flap sized 15 Â 10 cm was raised based on the second and third anterior perforating branches of the internal mammary vessel. The second costal cartilage was removed at a point 1.5 to 2 cm from the sternal border to facilitate isolation of a 2-cm segment of the internal mammary vessels, including the perforating branches. The internal mammary artery and vein were anastomosed to the superior thyroid artery and external jugular vein, respectively.