Epidermoid cyst of the presacral space is a rare congenital lesion. Cases of malignance arising from the lesion are extremely rare. After searching the literature, there are only 1 image report in English literature (PubMed) and 4 cases reports in Japanese literature of the disease. Here we report a 62-year-old female who was diagnosed with presacral cyst before surgery. Wide excision of the cystic tumor by Kraske's operation was performed. The pathology showed squamous cell carcinoma. Because of tumor rapture during operation, adjuvant radiotherapy 50 Gray/ 25 fractions was performed. After 1 year follow up, there is no evidence of disease.
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