A case of a 45 year-old woman who presented with a perianal fistula was reported. Histologically, the excised lesion showed features of apocrine fibroadenoma. In addition, the lesion had both glandular and stromal growth patterns and active chronic inflammation in the background. Malignant or benign counterparts of these types of lesions are widely described in literature. However fistula does not usually accompany and is very rare. Therefore, it is crucial for the pathologists and clinicians to be aware of such lesions. The relationship between apocrine fibroadenoma and perianal region was also discussed.
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