頬部に発生した primary adenoid cystic carcinoma of the skin の1例

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A 65-year-old Japanese man presented with an asymptomatic tumor in his left cheek, apart from the parotid area. The tumor had slowly increased in size over fifteen years. In physical examination, a firm, 2 cm sized in diameter, subcutaneous nodule was observed. Dark red erythema without erosion was seen on the overlying skin (Fig. 1) . Regional lymph nodes and superficial lymph nodes on the body were not palpable. CT and ultrasonography of the face and neck revealed that the tumor was so clearly separated from the surrounding tissues not to suggest any invasion to muscles and bone and that there was no clear lymph node swelling in the face and neck. Further examinations including gastrointestinal fiberscopy, CT, and garium scintigraphy ruled out a metastatic tumor.Wide local excision was performed 5 cm apart from the tumor margin and on the periosteum of the zygoma and the fascias of the masseter and expression muscles. Because the zygomatic and buccal branches of the facial nerve were invaded, they were also excised with enough free margin. Pathological examination could not completely deny any residual tumor on the bottom of the excised specimen. Accordingly 2 weeks later, additional excision was done more 5 cm apart from the previous margin and involving the periosteum of the zygoma and a portion of the masseter and expression muscles and the raw surface was covered by split thickness skin (Fig. 2) . Pathological examination demonstrated no residual tumors.Histologically, tumor cells were arranged in a cribriform pattern from the upper dermis to subcutaneous fatty tissue (Fig. 3) . Some of the tumor cells were found continuous to the hair follicles and sebaceous glands (Fig. 4) . Perinural extension was noted (Fig. 5) .Immunohistochemically, keratin and epithelial membrane antibody were immunoreactive with the tumor cells, whereas S-100 protein, vimentin, and carcino-embryonic antigen were not.On electron microscopy, the tumor cells were arranged in a luminal structure. Most of them, however, were pseudolumina containing fine stellate granules, basal lamina, and collagen fibers (Fig. 6) although some were true lumina with numerous microvilli and junctional complexes nearby where the tumor cells were connected to the hair follicles and subcutaneous glands (Fig. 7), which suggests that the tumor originates from a sweat gland. On the above-mentioned, the tumor was diagnosed as a primary cutaneous adenoid cystic carcinoma.After 14-month-follow, the tissue defect of the cheek was reconstructed by the transfer as a free flap of the secondary vascularized musculocutaneous flap composed of the anterior seratus muscle, of which the volume is almost equal to that of the excised muscles of the cheek, and the anterior chest skin, of which the color and texture are matched to that of the cheek skin (Fig. 8) . The use of expander promoted the secondary formation of vascular network between the anterior seratus muscle and the anterior chest skin and made the primary closure of the flap donor without free skin graft possible.No recurrence or metastasis of the tumor has been observed in the course of 27 months' follow and the patient is very satisfied with the restored cheek.

頬部に発生した primary adenoid cystic carcinoma of the skin の1例

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