May 2004
Volume 45, Issue 13
Free
ARVO Annual Meeting Abstract  |   May 2004
Intravitreal Anti–VEGF Therapy for Advanced Ocular Disease of Von Hippel–Lindau Disease
Author Affiliations & Notes
  • M. Cusick
    Division of Epidemiology and Clinical Research, National Eye Institute / NIH, Bethesda, MD
  • S.S. Dahr
    Division of Epidemiology and Clinical Research, National Eye Institute / NIH, Bethesda, MD
  • S. Srivastava
    Division of Epidemiology and Clinical Research, National Eye Institute / NIH, Bethesda, MD
  • H.R. Coleman
    Division of Epidemiology and Clinical Research, National Eye Institute / NIH, Bethesda, MD
  • D. Thompson
    EMMES Corporation, Potomac, MD
  • E.Y. Chew
    Division of Epidemiology and Clinical Research, National Eye Institute / NIH, Bethesda, MD
  • Footnotes
    Commercial Relationships  M. Cusick, None; S.S. Dahr, None; S. Srivastava, None; H.R. Coleman, None; D. Thompson, None; E.Y. Chew, None.
  • Footnotes
    Support  none
Investigative Ophthalmology & Visual Science May 2004, Vol.45, 1184. doi:
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      M. Cusick, S.S. Dahr, S. Srivastava, H.R. Coleman, D. Thompson, E.Y. Chew; Intravitreal Anti–VEGF Therapy for Advanced Ocular Disease of Von Hippel–Lindau Disease . Invest. Ophthalmol. Vis. Sci. 2004;45(13):1184.

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      © ARVO (1962-2015); The Authors (2016-present)

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Abstract

Abstract: : Purpose:To investigate the safety and efficacy of pegylated anti–vascular endothelial growth factor (VEGF) therapy, EYE001 (Macugen), for the treatment for patients with severe ocular disease associated with von Hippel–Lindau (VHL) disease. The VHL germline mutation in chromosome 3 results in an increase of VEGF in the eye lesions as well as multiple organs. Methods: This study was an open label, non–randomized, prospective, pilot study of intravitreal injections of EYE001 (Macugen, 3mg/100uL) in 5 patients with severe ocular VHL lesions with decreased vision secondary to macular edema.. Three patients had juxtapapillary angiomas and the other two had peripheral angiomas. Participants underwent 6 injections over a 30–week period with follow–up through 1 year after initiating treatment. The primary outcome of this study was changes of 15 letters (3 lines) or more in best corrected visual acuity (ETDRS) by 1 year. Secondary outcomes included reduction of macular thickening at 1 year as determined by optical coherence tomography (OCT) and reduction of fluorescein leakage in intensity and area. Results: Of the five participants with VHL enrolled in the study, 4 were female. At baseline, age ranged from 23 to 51, and visual acuity ranged from 20/40 to 20/200. Preliminary results revealed no significant change in visual acuity following injections. There was no decrease in tumor size. Fluorescein angiography revealed minimal decrease in fluorescein leakage while fundus photography showed a decrease in retinal hard exudate in 2 patients. OCT revealed a decrease in retinal thickness of 200 microns in 2 patients. Reported adverse events included a post–injection decrease in intraocular pressure. One eye experienced retinal detachment with increased fibrosis of peripheral angiomas. Another patient had increased fibrovascular proliferation around her lens implant. Conclusions: Intravitreal injections of anti–VEGF therapy, EYE001, may inhibit the vasopermeability with a decrease in retinal hard exudate and retinal thickening in 3 patients with advanced ocular disease of VHL. The size of the lesions was not affected. Further investigation in patients with retinal angiomas may be warranted.

Keywords: retinal neovascularization • neovascularization • drug toxicity/drug effects 
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