BACKGROUND: Gilles de la Tourette syndrome is a neurological disorder characterized by motor and vocal tics. Extreme forms of this disease manifest themselves with self-mutilating behavior and foul language. HISTORY AND SIGNS: A 59-year-old man, treated for Gilles de la Tourette syndrome, presented with visual loss after an attack during which he punched his right eye with his fist. On examination visual acuity was decreased to finger counting but could be improved to 1.0 with an S + 13D lens. There was vitreous prolaps into the anterior chamber and pigment was noted in the vitreous cavity. The complete dislocated lens could be seen ophthalmoscopically in the inferior fundus of the eye. THERAPY AND OUTCOME: The patient was treated with vitrectomy and phacoemulsification through the pars plana. An additional encircling band was placed due to peripheral retinal tears in the inferior quadrants. Visual rehabilitation was achieved with a secondary implant of a retropupillary iris-claw lens nine months later. DISCUSSION: Self-inflicted injuries are common in Gilles de la Tourette syndrome. Ocular lesions described in the literature comprise isolated cases of orbital hemorrhage, retinal detachment and permanent severe visual loss. Lens luxation has not been described before, and is probably due to weakened zonulae induced by repeated trauma. Treatment with a retropupillary iris-claw lens is the preferred therapy as it reduces the risk of endothelial touch during repeated trauma on the eye.