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Intravitreal Ranibizumab Therapy for Retinal Capillary Hemangioblastoma Related to von Hippel-Lindau Disease

2008; Elsevier BV; Volume: 115; Issue: 11 Linguagem: Inglês

10.1016/j.ophtha.2008.04.033

1549-4713

Wai T. Wong, Katharine J. Liang, Keri Hammel, Hanna R. Coleman, Emily Y. Chew,

Vascular Tumors and Angiosarcomas

Purpose To evaluate the effect of intravitreal ranibizumab on retinal capillary hemangioblastomas (RCHs) associated with von Hippel-Lindau (VHL) disease that are not amenable or responsive to standard therapy. Design Prospective, noncomparative, interventional case series. Participants Five patients with VHL-associated RCH with exudative changes and visual loss. Methods Monthly intravitreal injections of ranibizumab (0.5 mg) were given over a course of 6 months for a total of 7 injections, with additional injections considered until week 52. The final study visit was designated as 8 weeks after the final study injection. Main Outcome Measures The primary outcome was the change in best-corrected visual acuity (BCVA) of ≥15 letters at the final visit compared with baseline. Secondary outcomes included change in lesion size, exudation as assessed clinically and by fluorescein angiography, change in retinal thickness as evaluated by optical coherence tomography, and adverse event assessments. Results Patients received an average of 10.0±3.1 injections over an average period of 47±14 weeks, including follow-up. Mean change in BCVA was a decrease of 9±20 letters, with 1 patient gaining ≥15 letters, and 2 patients losing ≥15 letters. Changes in both lesion size and exudation were variable. Conclusions Intravitreal ranibizumab, delivered as monotherapy every 4 weeks, had minimal beneficial effects on most VHL-related RCHs. Possible treatment efficacy was demonstrated in the patient with the smallest lesion with less exudation. Future prospective studies are needed to determine the potential role of an antiangiogenic agent, possibly in combination with other therapies for the treatment of such advanced ocular tumors associated with VHL. Financial Disclosure(s) The authors have no proprietary or commercial interest in any materials discussed in this article. To evaluate the effect of intravitreal ranibizumab on retinal capillary hemangioblastomas (RCHs) associated with von Hippel-Lindau (VHL) disease that are not amenable or responsive to standard therapy. Prospective, noncomparative, interventional case series. Five patients with VHL-associated RCH with exudative changes and visual loss. Monthly intravitreal injections of ranibizumab (0.5 mg) were given over a course of 6 months for a total of 7 injections, with additional injections considered until week 52. The final study visit was designated as 8 weeks after the final study injection. The primary outcome was the change in best-corrected visual acuity (BCVA) of ≥15 letters at the final visit compared with baseline. Secondary outcomes included change in lesion size, exudation as assessed clinically and by fluorescein angiography, change in retinal thickness as evaluated by optical coherence tomography, and adverse event assessments. Patients received an average of 10.0±3.1 injections over an average period of 47±14 weeks, including follow-up. Mean change in BCVA was a decrease of 9±20 letters, with 1 patient gaining ≥15 letters, and 2 patients losing ≥15 letters. Changes in both lesion size and exudation were variable. Intravitreal ranibizumab, delivered as monotherapy every 4 weeks, had minimal beneficial effects on most VHL-related RCHs. Possible treatment efficacy was demonstrated in the patient with the smallest lesion with less exudation. Future prospective studies are needed to determine the potential role of an antiangiogenic agent, possibly in combination with other therapies for the treatment of such advanced ocular tumors associated with VHL.