Late-onset retinal macular degeneration: clinical insights into an inherited retinal degeneration

Br J Ophthalmol. 2009 Mar;93(3):284-9. doi: 10.1136/bjo.2008.150151. Epub 2008 Dec 19.

Abstract

Aim: This study describes, in detail, the phenotype of late-onset retinal macular degeneration (L-ORMD) an inherited condition affecting both the retina and anterior segment. A staging based on clinical characteristics is proposed, and the relevance of this condition to current understanding of age-related macular degeneration is discussed.

Methods: A systematic review of the literature regarding this condition supports a detailed description of the natural history. Clinical experiences in identifying, monitoring and managing patients are also presented.

Results: L-ORMD is a rare fully penetrant autosomal dominant condition resulting from a mutation in the C1QTNF5 gene on chromosome 11. Affected individuals develop bilateral loss of vision, dark-adaptation abnormalities, fundus drusen-like yellow spots, midperipheral pigmentation, choroidal neovascularisation, chorioretinal atrophy and long anteriorly inserted lens zonules. Patients may benefit from treatment with high-dose vitamin A.

Conclusions: Raised awareness of L-ORMD should lead to earlier diagnosis and improved care for patients. New antivascular endothelial growth factor treatment may provide a new possibility for management. A deeper insight into molecular and genetic mechanisms of L-ORMD may suggest avenues to explore new treatments of this disorder.

Publication types

  • Research Support, Non-U.S. Gov't
  • Review
  • Systematic Review

MeSH terms

  • Adult
  • Aged
  • Aged, 80 and over
  • Collagen / genetics
  • Female
  • Fundus Oculi
  • Humans
  • Macular Degeneration / drug therapy
  • Macular Degeneration / genetics
  • Macular Degeneration / pathology*
  • Male
  • Middle Aged
  • Mutation
  • Retina / pathology*
  • Vitamin A / therapeutic use
  • Vitamins / therapeutic use

Substances

  • Vitamins
  • Vitamin A
  • Collagen