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Rapid and durable recovery of visual function in a patient with von hippel-lindau syndrome after systemic therapy with vascular endothelial growth factor receptor inhibitor su5416.

Publication ,  Journal Article
Aiello, LP; George, DJ; Cahill, MT; Wong, JS; Cavallerano, J; Hannah, AL; Kaelin, WG
Published in: Ophthalmology
September 2002

PURPOSE: To present a case of rapid and durable recovery of visual function in a patient with von Hippel-Lindau syndrome and optic nerve head hemangioblastoma after systemic treatment with the vascular endothelial growth factor (VEGF) receptor inhibitor SU5416. DESIGN: Interventional case report. METHODS: Visual function parameters, including visual acuity, automated visual field, and contrast sensitivity, were evaluated using standardized methods repeatedly over a 15-month period. Fundus photographs and fluorescein angiograms were also obtained. Central nervous system lesions were monitored by computed tomography (CT) and magnetic resonance imaging (MRI) scans. Treatment involved the systemic administration of the VEGF receptor inhibitor SU5416. MAIN OUTCOME MEASURES: Clinical presentation, visual acuity using Early Treatment Diabetic Retinopathy Study protocol, Humphrey automated perimetry, (Zeiss Humphrey Systems, Dublin, CA) Vistech contrast sensitivity (Vistech Consultants Inc., Dayton, OH) Farnsworth (Farnsworth-Munsell Color Services, New Windsor, NY) dichotomous panel D-15, retinal photography, fluorescein angiography, and CT and MRI scans. RESULTS: Within 4 weeks of therapy, visual acuity had improved from 20/32(-2) to 20/16(-1), the visual field had expanded from being circumferentially constricted to within 8 degrees of fixation to normal, and contrast sensitivity improved in all but the lowest spatial frequency (1.5 cycles/degree). No change was observed in lesion size by fundus photography. Improvement has been maintained over 18 months with intermittent SU5416 therapy. CONCLUSIONS: We report rapid, extensive, and durable recovery of visual function after systemic administration of the VEGF receptor inhibitor SU5416 to a patient with VHL syndrome and optic nerve head hemangioblastoma. These findings suggest that continued evaluation of VEGF inhibitors for ocular neovascular disorders is warranted.

Duke Scholars

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Published In

Ophthalmology

DOI

ISSN

0161-6420

Publication Date

September 2002

Volume

109

Issue

9

Start / End Page

1745 / 1751

Location

United States

Related Subject Headings

  • von Hippel-Lindau Disease
  • Visual Fields
  • Visual Acuity
  • Recovery of Function
  • Receptors, Vascular Endothelial Growth Factor
  • Receptors, Growth Factor
  • Receptor Protein-Tyrosine Kinases
  • Pyrroles
  • Optic Nerve Neoplasms
  • Optic Disk
 

Citation

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Aiello, L. P., George, D. J., Cahill, M. T., Wong, J. S., Cavallerano, J., Hannah, A. L., & Kaelin, W. G. (2002). Rapid and durable recovery of visual function in a patient with von hippel-lindau syndrome after systemic therapy with vascular endothelial growth factor receptor inhibitor su5416. Ophthalmology, 109(9), 1745–1751. https://doi.org/10.1016/s0161-6420(02)01159-4
Aiello, Lloyd Paul, Daniel J. George, Mark T. Cahill, Jun S. Wong, Jerry Cavallerano, Alison L. Hannah, and William G. Kaelin. “Rapid and durable recovery of visual function in a patient with von hippel-lindau syndrome after systemic therapy with vascular endothelial growth factor receptor inhibitor su5416.Ophthalmology 109, no. 9 (September 2002): 1745–51. https://doi.org/10.1016/s0161-6420(02)01159-4.
Aiello, Lloyd Paul, et al. “Rapid and durable recovery of visual function in a patient with von hippel-lindau syndrome after systemic therapy with vascular endothelial growth factor receptor inhibitor su5416.Ophthalmology, vol. 109, no. 9, Sept. 2002, pp. 1745–51. Pubmed, doi:10.1016/s0161-6420(02)01159-4.
Aiello LP, George DJ, Cahill MT, Wong JS, Cavallerano J, Hannah AL, Kaelin WG. Rapid and durable recovery of visual function in a patient with von hippel-lindau syndrome after systemic therapy with vascular endothelial growth factor receptor inhibitor su5416. Ophthalmology. 2002 Sep;109(9):1745–1751.
Journal cover image

Published In

Ophthalmology

DOI

ISSN

0161-6420

Publication Date

September 2002

Volume

109

Issue

9

Start / End Page

1745 / 1751

Location

United States

Related Subject Headings

  • von Hippel-Lindau Disease
  • Visual Fields
  • Visual Acuity
  • Recovery of Function
  • Receptors, Vascular Endothelial Growth Factor
  • Receptors, Growth Factor
  • Receptor Protein-Tyrosine Kinases
  • Pyrroles
  • Optic Nerve Neoplasms
  • Optic Disk