Lichen sclerosus (LS) is a chronic cutaneous disorder of uncertain aetiology seen predominantly in females. It is characterized by white atrophic macules and plaques with a predilection for involvement of the genitalia and perianal skin. Gender-identity disorder (transsexualism) is a descriptive term for individuals having the genetic, somatic and hormonal basis of one sex but who identify sexually with the opposite sex. Surgical gender reassignment is a fundamental step in the management of these complex patients and is driven by the individual's belief of being the opposite sex 'trapped' in the wrong body. We report a case of genital lichen sclerosus in hair-bearing skin originally from the scrotum, in an individual who had undergone male-to-female gender reassignment. To our knowledge, no similar cases have been reported to date.