Ocular von Hippel-Lindau disease: clinical update and emerging treatments

Curr Opin Ophthalmol. 2008 May;19(3):213-7. doi: 10.1097/ICU.0b013e3282fb7c04.

Abstract

Purpose of review: von Hippel-Lindau disease is an inherited, multisystemic cancer syndrome often involving the retina. This review will provide an update for the clinical characterization and treatment of ocular von Hippel-Lindau disease.

Recent findings: A comprehensive and quantitative clinical characterization of ocular von Hippel-Lindau disease has been limited by small patient numbers and nonrepresentative sampling. Recently, a large population of patients with clinically and genetically defined von Hippel-Lindau disease was systemically characterized in a single center, enabling a quantitative evaluation of the ocular involvement of this syndrome. Correlations between the nature of von Hippel-Lindau gene mutations and the ocular phenotype were also examined, providing clues as to how disruptions in von Hippel-Lindau protein function may result in eye disease. This understanding may be relevant to the development of new therapies targeting the molecular biology of von Hippel-Lindau disease, some of which are presently being investigated.

Summary: Quantitative studies enable a full characterization of the impact of von Hippel-Lindau disease on eye health and visual function. Establishing correlations between the genotype of the von Hippel-Lindau mutation and the phenotype of eye disease may inform us as to how ocular von Hippel-Lindau disease arises, and help guide molecular interventions in ocular von Hippel-Lindau disease.

Publication types

  • Review

MeSH terms

  • Angiogenesis Inhibitors / therapeutic use*
  • Genotype
  • Hemangioma, Capillary* / diagnosis
  • Hemangioma, Capillary* / drug therapy
  • Hemangioma, Capillary* / genetics
  • Humans
  • Mutation
  • Photochemotherapy / methods*
  • Prognosis
  • Retinal Neoplasms* / diagnosis
  • Retinal Neoplasms* / drug therapy
  • Retinal Neoplasms* / genetics
  • Von Hippel-Lindau Tumor Suppressor Protein / genetics
  • von Hippel-Lindau Disease* / diagnosis
  • von Hippel-Lindau Disease* / drug therapy
  • von Hippel-Lindau Disease* / genetics

Substances

  • Angiogenesis Inhibitors
  • Von Hippel-Lindau Tumor Suppressor Protein
  • VHL protein, human