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Purpose: to present a case of successful Gamma-knife radiosurgery (GKRS) of the single eye with diffuse choroidal hemangioma (DCH) in a child with Sturge-Weber syndrome. Methods: A 4.5-year old girl was referred to our center with port-wine stains of the face, leptomeningeal angiomas and bilateral DCH. The right eye was presented with DCH of 6.6 mm in thickness with no signs of exudation. The left eye was diagnosed with DCH of 6.8 mm in thickness, total retinal detachment and vision loss. Ruthenium brachytherapy was performed but retinal detachment increased. Within 5 months follow-up the right eye developed retinal detachment up to 6.6 mm in height. GKRS was used as the last opportunity to save the eye and improve vision. Dosimetric plan included double PTVs of 18 Gy @ 57% and 18 Gy @ 50% excluding central retina, and will be presented. Results: During 8 months of follow up choroidal tumor thickness decreased from 6.6mm to 3.0mm with retinal detachment resolution that provided child a good vision in the only seeing eye. Irradiation “tracks” according to the planning were seen on the eye fundus examination without radiation damage of central retina. No complications were revealed within the follow-up. Conclusion: This exceptional case presents the first to our knowledge experience of GKRS of DCH in a child with Sturge-Weber syndrome. GKRS allowed to irradiate intraocular vascular tumor precisely and save the eye with vision improvement. Further experience is essential.