Document Detail

Improvement of visual performance with intravitreal administration of 9-cis-retinal in Rpe65-mutant dogs.
MedLine Citation:
PMID:  20837787     Owner:  NLM     Status:  MEDLINE    
OBJECTIVE: To determine the efficacy of intravitreal administration of 9-cis-retinal in restoring visual function in Rpe65-mutant dogs.
METHODS: Intravitreal injection of 9-cis-retinal was administered in 1 eye of 7 Rpe65-/- dogs at a range of ages. Electroretinogram analysis and testing of visual performance was used to evaluate outcomes after a single injection and in 2 dogs after a second injection in the same eye.
RESULTS: In 5 of 7 injected dogs, 9-cis-retinal injection resulted in increased rod electroretinogram responses and improved functional vision. Three injected dogs exhibited increased 33-Hz flicker amplitudes characteristic of cone-mediated responses. Electroretinogram improvement was no longer evident by week 10 postinjection in 1 dog monitored over time. A second injection of 9-cis-retinal was performed in the same eye of 2 of the 7 dogs and also resulted in rescue of visual function.
CONCLUSION: Our findings establish that 9-cis-retinoid therapy can restore visual function in a canine model of human disease resulting from RPE65 mutations.
CLINICAL RELEVANCE: These positive proof-of-principle results provide support for the development of intravitreal devices for sustained delivery of 9-cis-retinal as a therapy for conditions resulting from failure of the visual cycle.
Patricia M Gearhart; Chris Gearhart; Debra A Thompson; Simon M Petersen-Jones
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Publication Detail:
Type:  Journal Article; Research Support, Non-U.S. Gov't     Date:  2010-09-13
Journal Detail:
Title:  Archives of ophthalmology     Volume:  128     ISSN:  1538-3601     ISO Abbreviation:  Arch. Ophthalmol.     Publication Date:  2010 Nov 
Date Detail:
Created Date:  2010-11-09     Completed Date:  2010-12-10     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  7706534     Medline TA:  Arch Ophthalmol     Country:  United States    
Other Details:
Languages:  eng     Pagination:  1442-8     Citation Subset:  AIM; IM    
Department of Small Animal Clinical Sciences, Michigan State University, East Lansing, MI 48824, USA.
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MeSH Terms
Carrier Proteins / genetics*
Dark Adaptation
Disease Models, Animal*
Eye Proteins / genetics*
Intravitreal Injections
Leber Congenital Amaurosis / drug therapy*,  genetics,  physiopathology
Photoreceptor Cells, Vertebrate / physiology*
Retinaldehyde / administration & dosage*
Vision, Ocular / physiology*
Visual Acuity / physiology
Reg. No./Substance:
0/Carrier Proteins; 0/Eye Proteins; 116-31-4/Retinaldehyde; 514-85-2/9-cis-retinal
Comment In:
Arch Ophthalmol. 2010 Nov;128(11):1483-5   [PMID:  21060052 ]

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