ArticleDocumenta ophthalmologica. Advances in ophthalmology2014
Flicker cone function in normal and day blind sheep: a large animal model for human achromatopsia caused by CNGA3 mutation.
Article in Documenta ophthalmologica. Advances in ophthalmology, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 9 papers.
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9 citing papers in PubMed, 21 citations in OpenAlex.
- Outer retinal transduction by AAV2-7m8 following intravitreal injection in a sheep model of CNGA3 achromatopsia.Gene therapy · 2022Article
- Naturally-occurring myopia and loss of cone function in a sheep model of achromatopsia.Scientific reports · 2020Article
- Six Years and Counting: Restoration of Photopic Retinal Function and Visual Behavior Following Gene Augmentation Therapy in a Sheep Model ofHuman gene therapy · 2018Article
- Consecutive unilateral recording of the two eyes affects dark-adapted ERG responses, when compared to simultaneous bilateral recording.Documenta ophthalmologica. Advances in ophthalmology · 2018Article
- Valence and Intensity of Video Stimuli of Dogs and Conspecifics in Sheep: Approach-Avoidance, Operant Response, and Attention.Animals : an open access journal from MDPI · 2018Article
- Gene Augmentation Therapy for a Missense Substitution in the cGMP-Binding Domain of Ovine CNGA3 Gene Restores Vision in Day-Blind Sheep.Investigative ophthalmology & visual science · 2017Article
- Residual Foveal Cone Structure in CNGB3-Associated Achromatopsia.Investigative ophthalmology & visual science · 2016Article
- Gene Augmentation Therapy Restores Retinal Function and Visual Behavior in a Sheep Model of CNGA3 Achromatopsia.Molecular therapy : the journal of the American Society of Gene Therapy · 2015Article
- Retinal Gene Therapy: Current Progress and Future Prospects.Expert review of ophthalmology · 2015Article
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Authors and funding
9 authors at 4 institutions in 2 countries.
Funding
Abstract
purposeRecently we reported on day blindness in sheep caused by a mutation in the CNGA3 gene, thus making affected sheep a naturally occurring large animal model for therapeutic intervention in CNGA3 achromatopsia patients. The purpose of this study was to characterize flicker cone function in normal and day blind sheep, with the aim of generating a normative data base for ongoing gene therapy studies.
methodsElectoretinographic (ERG) cone responses were evoked with full-field conditions in 10 normal, 6 heterozygous carriers and 36 day blind sheep. Following light adaptation (10 min, 30 cd/m(2)), responses were recorded at four increasing light intensities (1, 2.5, 5 and 10 cd s/m(2)). At each of these intensities, a single photopic flash response followed by 8 cone flicker responses (10-80 Hz) was recorded. Results were used to generate a normative data base for the three groups. Differences between day blind and normal control animals were tested in two age-matched groups (n = 10 per group).
resultsThe normal sheep cone ERG wave is bipartite in nature, with critical flicker fusion frequency (CFF) >80 Hz. In all four flash intensities, the single photopic flash a-wave and b-wave amplitudes were significantly lower (p < 0.005), and implicit times significantly delayed (p < 0.0001), in day blind animals. In all four flash intensities, CFF values were significantly lower (p < 0.0001) in day blind sheep.
conclusionsCone function is severely depressed in day blind sheep. Our results will provide a normative data base for ongoing gene therapy studies.
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