The genital localized myiasis is a rare zooparasitic dermatosis. This case reported to a penile myiasis by Dermatobia hominis larvae in a 62 year-old man, who is a retired military official from São Gonçalo city, Rio de Janeiro state, Brazil. This patient was guided to the sexually transmitted diseases (STD) clinic from Universidade Federal Fluminense (UFF) by professionals from a urology outpatient clinic of a public hospital from Rio de Janeiro state. The exam showed a 2 cm diameter nodular lesion with a hyperemic area in the glands penis that had arisen 20 days before the exam. It evolved in a favorable way after larvae removal, through previous local anesthesia and slight pressure in lesion's base with the extraction of the larvae. Penile myiasis is not a very frequent situation, although it must be considered as an option in differential diagnosis of genital infectious diseases, even among people of low economical level, as in the case presented.