Since May 2022, the monkeypox (MPX) virus has represented an emerging issue due to outbreaks in non-endemic areas. This report presents the first case of paraphimosis caused by an MPX infection during the outbreak. The patient accessed the emergency department for a sudden onset of swelling of the penis and paraphimosis caused by MPX lesions that brought about stenosis of the foreskin. He therefore underwent a dorsal slit procedure with resolution. No antiviral therapy was required. A multidisciplinary approach should be preferred for the management of MPX, due to the possibility of uncommon and disseminated presentations.