Human papillomaviruses (HPVs) of the alpha genus induce a wide spectrum of epithelial lesions ranging from benign warts to anogenital dysplasia or invasive cancer. Penile intraepithelial neoplasia is a potential penile cancer precursor, and 50% to 90% of cases are induced by high-risk HPV types such as HPV16.1 We herein describe a patient with an unusual HPV-induced pigmented high-grade dysplasia of the scrotum that does not fit into the established clinical classification of anogenital HPV-associated lesions.