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To describe the treatment and natural history of a patient with complicated congenital retinoschisis.A retrospective case report. A 10-month-old boy with congenital retinoschisis presented with tractional retinal detachments and foveal schisis in both eyes.On presentation, vision was decreased in both eyes with presumed amblyopia of the left eye. Funduscopic examination revealed bilateral foveal schisis and tractional retinal detachment involving the macula. Follow-up examination revealed superior retinal dragging and peripheral ischemia on fluorescein angiogram in both eyes. Nine months after presentation, combined rhegmatogenous and tractional retinal detachment developed in the right eye and was treated by scleral buckle. After vitrectomy for nonclearing vitreous hemorrhage in the left eye, a combined rhegmatogenous and tractional retinal detachment developed. Vitrectomy and lensectomy with silicone oil was performed. At 6 years of follow-up, both retinas were attached and foveal schisis had resolved.Sight threatening complications of congenital retinoschisis include retinal detachment and vitreous hemorrhage. Vitrectomy and/or scleral buckling may prevent progression of vision loss and promote resolution of schisis.