Intravitreal anti-vascular endothelial growth factor therapy with bevacizumab for tuberous sclerosis with macular oedema

概要

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Purpose: To describe two patients with macular oedema secondary to tuberous sclerosis complex (TSC), who were treated with intravitreal bevacizumab injection. Methods: Interventional case reports. Bevacizumab 1.25 mg was injected into the vitreous of two patients with TSC-associated macular oedema / exudative retinal detachment. VEGF concentration in the vitreous fluid was measured by ELISA in one of these patients. Results: Case 1. 22-year-old woman with TSC was diagnosed as having multiple retinal hamartomas in both eyes. Eleven years later, the patient developed macular oedema with epiretinal membrane formation in the right eye. The patient underwent pars plana vitrectomy with retinal photocoagulation for retinal tumours. VEGF concentration in the vitreous fluid was high compared to that in patients without retinal vascular diseases. Recurrent macular oedema was resolved by intravitreal injection of bevacizumab. Case 2. 32-year-old woman with TSC-associated retinal hamartoma, temporally showing macular exudative retinal detachment, developed the neovascularization originated from the tumour. By intravitreal bevacizumab injection, the tumour size markedly reduced with regression of neovascularization. Conclusions: These results suggest that VEGF derived from retinal hamartomas causes macular oedema associated with TSC. Intravitreal injections of bevacizumab may be a useful therapeutic option for macular oedema secondary to TSC.