ArticleInvestigative ophthalmology & visual science2014
A Pro23His mutation alters prenatal rod photoreceptor morphology in a transgenic swine model of retinitis pigmentosa.
Article in Investigative ophthalmology & visual science, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 25 papers.
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Who cites it
25 citing papers in PubMed.
- Subretinal Injection Volume Correlates to Persistent Outer Retinal Thinning in the Pig Eye.Investigative ophthalmology & visual science · 2025Article
- RHO1-2 meganuclease gene editing targets human P23H rhodopsin-induced retinitis pigmentosa to rejuvenate rods and maintain cones.bioRxiv : the preprint server for biology · 2025Article
- Gene therapy advances using canine and feline animal models of inherited retinal degeneration.Eye (London, England) · 2025Review
- Animal models for the evaluation of retinal stem cell therapies.Progress in retinal and eye research · 2025Review
- Optimizing Subretinal Bleb Formation for Visual Streak Involvement in a Porcine Model for Retinal Gene Therapy.Investigative ophthalmology & visual science · 2024Article
- Pig Models in Retinal Research and Retinal Disease.Cold Spring Harbor perspectives in medicine · 2024Review
- Gene Therapy for Rhodopsin Mutations.Cold Spring Harbor perspectives in medicine · 2022Article
- Gene Therapy for Rhodopsin-associated Autosomal Dominant Retinitis Pigmentosa.International ophthalmology clinics · 2021Article
- Yap1 is required for maintenance of adult RPE differentiation.FASEB journal : official publication of the Federation of American Societies for Experimental Biology · 2020Article
- Article
- Noninvasive Imaging and Correlative Histology of Cone Photoreceptor Structure in the Pig Retina.Translational vision science & technology · 2019Article
- Growth and Behavior of Congenitally Anophthalmic Lee-Sung Pigs.Comparative medicine · 2019Article
- Sector retinitis pigmentosa: Report of ten cases and a review of the literature.Molecular vision · 2019Review
- Correlation of spectral domain optical coherence tomography with histology and electron microscopy in the porcine retina.Experimental eye research · 2018Article
- Anatomic Studies of the Miniature Swine Cornea.Anatomical record (Hoboken, N.J. : 2007) · 2018Article
- Progression of Pro23His Retinopathy in a Miniature Swine Model of Retinitis Pigmentosa.Translational vision science & technology · 2017Article
- Two-Step Reactivation of Dormant Cones in Retinitis Pigmentosa.Cell reports · 2016Article
- Animal modelling for inherited central vision loss.The Journal of pathology · 2016Review
- Prenatal Exposure to Curcumin Protects Rod Photoreceptors in a Transgenic Pro23His Swine Model of Retinitis Pigmentosa.Translational vision science & technology · 2015Article
- Integration of Perforated Subretinal Prostheses With Retinal Tissue.Translational vision science & technology · 2015Article
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4 authors.
Funding
Abstract
purposeFunctional studies have detected deficits in retinal signaling in asymptomatic children from families with inherited autosomal dominant retinitis pigmentosa (RP). Whether retinal abnormalities are present earlier during gestation or shortly after birth in a subset of children with autosomal dominant RP is unknown and no appropriate animal RP model possessing visual function at birth has been available to examine this possibility. In a recently developed transgenic P23H (TgP23H) rhodopsin swine model of RP, we tracked changes in pre- and early postnatal retinal morphology, as well as early postnatal retinal function.
methodsDomestic swine inseminated with semen from a TgP23H miniswine founder produced TgP23H hybrid and wild type (Wt) littermates. Outer retinal morphology was assessed at light and electron microscopic levels between embryonic (E) and postnatal (P) day E85 to P3. Retinal function was evaluated using the full field electroretinogram at P3.
resultsEmbryonic TgP23H rod photoreceptors are malformed and their rhodopsin expression pattern is abnormal. Consistent with morphological abnormalities, rod-driven function is absent at P3. In contrast, TgP23H and Wt cone photoreceptor morphology (E85-P3) and cone-driven retinal function (P3) are similar.
conclusionsPrenatal expression of mutant rhodopsin alters the normal morphological and functional development of rod photoreceptors in TgP23H swine embryos. Despite this significant change, cone photoreceptors are unaffected. Human infants with similarly aggressive RP might never have rod vision, although cone vision would be unaffected. Such aggressive forms of RP in preverbal children would require early intervention to delay or prevent functional blindness.
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