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

Abstract

. 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. Vascular endothelial growth factor (VEGF) concentration in the vitreous fluid was measured by enzyme‐linked immunosorbent assay (ELISA) in one of these patients. Results: Patient 1: a 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 disappeared by intravitreal injection of bevacizumab. Patient 2: a 32‐year‐old woman with TSC‐associated retinal hamartoma, temporally showing macular exudative retinal detachment, developed neovascularization originated from the tumour. By intravitreal bevacizumab injection, the tumour size reduced markedly with regression of neovascularization. Conclusion: 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.

Vasoproliferative retinal tumours in a Swedish population

“…With regard to the clinical effect on exudations and the shrinkage of tumour size, it is possible that antivascular endothelial growth factor (VEGF) could be a complementary or alternative treatment when cryotherapy or brachytherapy is insufficient or inappropriate for certain reasons. The positive effect of both anti-VEGF (Kenawy et al 2007) and photodynamic treatment (PDT) with verteporfin (Barbezetto & Smith 2003;Blasi et al 2006;Osman et al 2007;Saldanha & Edrich 2008) has been described in earlier reports on VPRTs and similar tumorous lesions (Eskelin et al 2008;Saito et al 2008). However, further evaluation is needed before any of these treatments are accepted as established therapies for VPRTs.…”

Expression of Vascular Endothelial Growth Factor and Intravitreal Anti-Vegf Therapy With Bevacizumab in Vasoproliferative Retinal Tumors

“…8,[11][12][13] However, there are only a few reports regarding the use of anti-VEGF drugs for VPRTs. 5,14 Kenawy 14 described the usefulness of IVB for a patient with VPRT with visual acuity loss of 6/19 and 5.0 mm in diameter and 1.7 mm thick in tumor size.…”

“…We previously reported that tumor-derived vascular endothelial growth factor (VEGF) caused macular edema/exudative retinal detachment in these patients, identified from the results of high intravitreal VEGF levels and favorable efficacy of these changes by intravitreal bevacizumab injection (IVB) in these patients. 8 Also, VPRT is a highly vascularized tumor, consisting of spindle-shaped glial cells and a dense capillary network with large hyalinized blood vessels. 3,4,9,10 To the best of our knowledge, however, no reports have examined the expression of VEGF in VPRTs.…”

Exudative retinal astrocytic hamartoma diagnosed and treated with pars plana vitrectomy and intravitreal bevacizumab

“…A recent report suggests that intravitreal bevacizumab may be effective for macula edema secondary to tuberous sclerosis. 4 Although retinal astrocytoma in tuberous sclerosis is usually a stationary lesion, vitrectomy may be useful for the diagnosis and management of a progressive retinal tumor in such patients. Furthermore, intravitreal bevacizumab provides a useful adjunctive therapy.…”