Isolated scrotal hair development in infancy is a rare disorder that has not been well-characterized. The article describes a case of transient isolated scrotal hairs in a five-month-old boy. Except for scrotal hair development, physical examination was normal. There were no other signs of virilization or secondary sexual development. Growth pattern, laboratory evaluation and bone-age radiographs were unremarkable. Spontaneous disappearance of scrotal hairs observed at the end of the first year of life finds confirmation in the literature that considers the transient isolated scrotal hairs as a rare self-limiting benign condition.