Gene augmentation for X-linked retinitis pigmentosa caused by mutations in RPGR

Cold Spring Harb Perspect Med. 2014 Oct 9;5(2):a017392. doi: 10.1101/cshperspect.a017392.

Abstract

X-linked retinitis pigmentosa (XLRP) caused by mutations in the RPGR gene is a severe and early onset form of retinal degeneration, and no treatment is currently available. Recent evidence in two clinically relevant canine models shows that adeno-associated viral (AAV)-mediated RPGR gene transfer to rods and cones can prevent disease onset and rescue photoreceptors at early- and mid-stages of degeneration. There is thus a strong incentive for conducting long-term, preclinical efficacy and safety studies, while concomitantly pursuing the detailed phenotypic characterization of XLRP disease in patients that may benefit from such corrective therapy.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't
  • Review

MeSH terms

  • Animals
  • Carrier Proteins / genetics*
  • Disease Models, Animal
  • Dogs
  • Eye Proteins / genetics*
  • Female
  • Genetic Diseases, X-Linked / genetics*
  • Genetic Diseases, X-Linked / therapy
  • Genetic Linkage
  • Genetic Therapy
  • Humans
  • Male
  • Mice
  • Mutation
  • Phenotype
  • Retinitis Pigmentosa / genetics*
  • Retinitis Pigmentosa / therapy
  • Treatment Outcome

Substances

  • Carrier Proteins
  • Eye Proteins
  • RPGR protein, human
  • RPGR protein, mouse