ArticleThe Journal of comparative neurology2007
Effect of Rds abundance on cone outer segment morphogenesis, photoreceptor gene expression, and outer limiting membrane integrity.
Article in The Journal of comparative neurology, 2007. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 26 papers.
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Who cites it
26 citing papers in PubMed, 35 citations in OpenAlex.
- Gene therapy rescues cone function inMolecular therapy. Advances · 2026Article
- Comparative study of PRPH2 D2 loop mutants reveals divergent disease mechanism in rods and cones.Cellular and molecular life sciences : CMLS · 2023Article
- Delineating the Clinical Phenotype of Patients With the c.629C>G, p.Pro210Arg Mutation in Peripherin-2.Investigative ophthalmology & visual science · 2022Article
- InVision: An optimized tissue clearing approach for three-dimensional imaging and analysis of intact rodent eyes.iScience · 2021Article
- ROM1 contributes to phenotypic heterogeneity in PRPH2-associated retinal disease.Human molecular genetics · 2020Article
- Rom1 converts Y141C-Prph2-associated pattern dystrophy to retinitis pigmentosa.Human molecular genetics · 2017Article
- The K153Del PRPH2 mutation differentially impacts photoreceptor structure and function.Human molecular genetics · 2016Article
- PRPH2/RDS and ROM-1: Historical context, current views and future considerations.Progress in retinal and eye research · 2016Review
- Loss of human disease protein retinitis pigmentosa GTPase regulator (RPGR) differentially affects rod or cone-enriched retina.Human molecular genetics · 2016Article
- Gene therapy for PRPH2-associated ocular disease: challenges and prospects.Cold Spring Harbor perspectives in medicine · 2014Review
- Insights into the mechanisms of macular degeneration associated with the R172W mutation in RDS.Human molecular genetics · 2014Article
- Initiation of rod outer segment disc formation requires RDS.PloS one · 2014Article
- Overexpression of retinal degeneration slow (RDS) protein adversely affects rods in the rd7 model of enhanced S-cone syndrome.PloS one · 2013Article
- Increased cone sensitivity to ABCA4 deficiency provides insight into macular vision loss in Stargardt's dystrophy.Biochimica et biophysica acta · 2012Article
- Defects in the outer limiting membrane are associated with rosette development in the Nrl-/- retina.PloS one · 2012Article
- Degeneration of the mouse retina upon dysregulated activity of serum response factor.Molecular vision · 2011Article
- Cone photoreceptors are the main targets for gene therapy of NPHP5 (IQCB1) or NPHP6 (CEP290) blindness: generation of an all-cone Nphp6 hypomorph mouse that mimics the human retinal ciliopathy.Human molecular genetics · 2011Article
- Differences in RDS trafficking, assembly and function in cones versus rods: insights from studies of C150S-RDS.Human molecular genetics · 2010Article
- Impact of acute inflammation on spinal motoneuron synaptic plasticity following ventral root avulsion.Journal of neuroinflammation · 2010Article
- Gene delivery to mitotic and postmitotic photoreceptors via compacted DNA nanoparticles results in improved phenotype in a mouse model of retinitis pigmentosa.FASEB journal : official publication of the Federation of American Societies for Experimental Biology · 2010Article
Corrections and comments
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Authors and funding
3 authors at 2 institutions in 1 country.
Funding
Abstract
We examined the molecular, structural, and functional consequences on cone photoreceptors of the neural retinal leucine zipper knockout (Nrl(-/-)) mice when only one allele of retinal degeneration slow (Rds) is present (Rds(+/-)/Nrl(-/-)). Quantitative RT-PCR and immunoblot analysis were used to assess the expression levels of several phototransduction genes; electroretinography was used to assess quantitatively the retinal responsiveness to light; and immunohistochemistry and ultrastructural analysis were used to examine retinal protein distribution and morphology, respectively. In Rds/Nrl double-null mice, S-cones form dysmorphic outer segments that lack lamellae and fail to associate properly with the cone matrix sheath and the outer limiting membrane. In Rds(+/-)/Nrl(-/-) mice, cones form oversized and disorganized outer segment lamellae; although outer limiting membrane associations are maintained, normal interactions with cone matrix sheaths are not, and photoreceptor rosette formation is observed. These retinas produce significantly higher photopic a-wave and b-wave amplitudes than do those of Rds(-/-)/Nrl(-/-) mice, and the levels of several cone phototransduction genes are significantly increased coincidently with the presence of Rds and partial lamellae formation. Thus, as in rod photoreceptors, expression of only one Rds allele is unable to support normal outer segment morphogenesis in cones. However, cone lamellae assembly, albeit disorganized, concomitantly permits outer limiting membrane association, and this appears to be linked to photoreceptor rosette formation in the rodless (cone-only) Nrl(-/-) retina. In addition, photoreceptor gene expression alterations occur in parallel with changes in Rds levels.
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