Restless legs syndrome, which recently was coined as Willis-Ekbom disease (WED) is a sleep related movement disorder characterized by an indefinable sense in legs. Rarely, genital restless symptoms may be seen as isolated or accompanied by leg symptoms. Herein, we share a WED case mild leg symptoms and with severe genital symptoms affecting her social life. Genital symptoms have showed improvement by orgasm and L-dopa treatment. Therefore, WED should be considered in differential diagnosis of patients with genital symptoms fulfilling diagnostic criteria of WED defined for restless legs.