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Microcystic adnexal carcinoma is a rare cutaneous neoplasm characterized by slow but locally aggressive growth, which normally does not lead to systemic metastasis. Frequent local recurrences are reported, which are most likely due to insufficient operative technique. We present the fourth case of cervical ipsilateral metastatic microcystic adnexal carcinoma in an otherwise healthy woman. The patient presented with a previously diagnosed but not completely resected microcystic adnexal carcinoma in the area of the right posterior scalp and two palpable ipsilateral lymph nodes. The tumor was resected using intraoperative snap frozen histological evaluation of the resection borders. In the same procedure two lymph nodes were resected from the right neck. The lymph nodes were histologically assessed and showed infiltration by small strains of tumor cells. After exclusion of a second primary tumor, e.g., mammary carcinoma, as the cause for cervical lymph node metastases, we performed a modified radical neck dissection with resection of the sternocleidomastoid muscle and the accessory nerve, which was histologically proven to be perineurally infiltrated by tumor cells. In this second procedure the histological evaluation of the specimen showed no sign of remaining tumor infiltration. After exclusion of distant metastasis the patient was irradiated with 60 Gy. The patient is well 1 year after the initial treatment without signs of recurrence. [ABSTRACT FROM AUTHOR]