A 16-year-old boy with X-linked retinoschisis was examined for the visual decline on his left eye. Upon examination, a vasopermeable elevated mass lesion was determined at the lower temporal retinal periphery associated with surrounding arc-like hard exudates which is consistent with a vasoactive proliferative tumor of the retina. After discussing the therapeutic options with the patient and his parents, 3 successive dexamethasone implants 6 months apart were administered. As no satisfactory regression was noticed, CyberKnife stereotactic radiosurgery (SRS) was performed. Regression of the exudative tumor was obtained in follow-up examinations at 12 and 24 months after the SRS session and the thickness of the lesion was markedly reduced.