Although its significance remains unknown, it has recently been reported that epidermal papillomatous hyperplasia could be frequently found in extramammary Paget’s disease (EMPD). The simultaneous occurrence of EMPD and various human-papillomavirus (HPV)-associated neoplasms has also been reported. We report a case of a perianal EMPD with multiple verrucous papules on its surface. Histologically, the verrucous papules consisted of two distinct histological patterns; one showed numerous Paget’s cells within the hyperplastic epidermis with papillomatosis, and the other showed the features compatible with condyloma acuminatum (CA). HPV-6 DNA was demonstrated in koilocytic keratinocytes of the CA by in situ hybridization. No positive signal was obtained for targeted HPV type 6/11/16/18/31/33 DNA in either the verrucous or plaque EMPD. Since the verrucous papules were localized on the plaque of EMPD, our case suggests that some undetermined EMPD-related factors may contribute to the development of epithelial hyperplasia, including HPV-associated neoplasms.

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