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Dataset results
194 results for “Retinitis pigmentosa”
Study of SAR421869 in Participants With Retinitis Pigmentosa Associated With Usher Syndrome Type 1B
ClinicalTrials.gov study NCT01505062. IPD Sharing: YES. Countries: 2. Publications: 1.
Retinal proteome profiling of inherited retinal degeneration across three different mouse models suggests common drug targets in retinitis pigmentosa
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Supplementary data for "The mechanistic functional landscape of Retinitis Pigmentosa: an ML-driven approach to drug repurposing"
<p>Supplementary data for "The mechanistic functional landscape of Retinitis Pigmentosa: an ML-driven approach to drug repurposing"</p><p> </p><p>version: 10.5281/zenodo.10203479</p><ul><li>added missing file: "drug_actions_withSimplAction.csv"</li></ul>
Gene augmentation prevents retinal degeneration in a CRISPR/Cas9-based mouse model of PRPF31 retinitis pigmentosa
<p>Mutations in <em>PRPF31</em> cause autosomal dominant retinitis pigmentosa, an untreatable form of blindness. Gene therapy is a promising treatment for <em>PRPF31</em>-retinitis pigmentosa, however, there are currently no suitable animal models in which to develop AAV-mediated gene augmentation. Here we establish <em>Prpf31</em> mutant mouse models using AAV-mediated CRISPR/Cas9 knockout, and characterize the resulting retinal degeneration phenotype. Mouse models with early-onset morphological and functional impairments like those in patients were established, providing new platforms in which to investigate pathogenetic mechanisms and develop therapeutic methods. AAV-mediated <em>PRPF31</em> gene augmentation restored the retinal structure and function in a rapidly degenerating mouse model, demonstrating the first in vivo proof-of-concept for AAV-mediated gene therapy to treat <em>PRPF31</em>-retinitis pigmentosa. AAV-CRISPR/Cas9-PRPF31 knockout constructs also mediated efficient <em>PRPF31</em> knockout in human and non-human primate retinal explants, laying a foundation for establishing non-human primate models using the method developed here.</p>
Late gene therapy limits the restoration of retinal function in a mouse model of retinitis pigmentosa
<p><span>Retinitis pigmentosa is an inherited photoreceptor degeneration that begins with rod loss followed by cone loss. This cell loss greatly diminishes vision, with most patients becoming legally blind. Gene therapies are being developed, but it is unknown how retinal function depends on the time of intervention. To uncover this dependence, we utilize a mouse model of retinitis pigmentosa capable of artificial genetic rescue. This model enables a benchmark of best-case gene therapy by removing variables that complicate the ability to answer this vital question. Complete genetic rescue was performed at 25%, 50%, and 70% rod loss (early, mid, and late, respectively). Here we show early- and mid-treatment restores retinal function to near wild-type levels, specifically the sensitivity and signal fidelity of retinal ganglion cells, the output neurons of the retina. However, some anatomical defects persist. Late treatment retinas exhibit continued, albeit slowed, loss of sensitivity and signal fidelity among retinal ganglion cells, as well as persistent gliosis. We conclude that gene replacement therapies delivered after 50% rod loss are unlikely to restore visual function to normal. This is critical information for administering gene therapies to rescue vision.</span></p>
Safety of a Single, Intravitreal Injection of Human Retinal Progenitor Cells (jCell) in Retinitis Pigmentosa
ClinicalTrials.gov study NCT02320812. IPD Sharing: NO. Countries: 1. Publications: 1.
Study of Oral Minocycline in Treating Bilateral Cystoid Macular Edema Associated With Retinitis Pigmentosa
ClinicalTrials.gov study NCT02140164. IPD Sharing: NO. Countries: 1. Publications: 4.
Gene Therapy for X-linked Retinitis Pigmentosa (XLRP) - Retinitis Pigmentosa GTPase Regulator (RPGR)
ClinicalTrials.gov study NCT03252847. IPD Sharing: NO. Countries: 2. Publications: 1.
A Study of Encapsulated Cell Technology (ECT) Implant for Participants With Early Stage Retinitis Pigmentosa
ClinicalTrials.gov study NCT00447980. IPD Sharing: NO. Countries: 1. Publications: 3.
DHA and X-Linked Retinitis Pigmentosa
ClinicalTrials.gov study NCT00100230. IPD Sharing: Not stated. Countries: 1. Publications: 5.
AGN-151597 (Formerly RST-001) Phase I/II Trial for Advanced Retinitis Pigmentosa
ClinicalTrials.gov study NCT02556736. IPD Sharing: YES. Countries: 1. Publications: 1.
Feasibility Study of a Chronic Retinal Stimulator in Retinitis Pigmentosa
ClinicalTrials.gov study NCT00279500. IPD Sharing: NO. Countries: 1. Publications: 8.
A First-in-human, Proof of Concept Study of CPK850 in Patients With RLBP1 Retinitis Pigmentosa
ClinicalTrials.gov study NCT03374657. IPD Sharing: UNDECIDED. Countries: 1. Publications: 1.
Retinal Imaging in CNTF -Releasing Encapsulated Cell Implant Treated Patients for Early-stage Retinitis Pigmentosa
ClinicalTrials.gov study NCT01530659. IPD Sharing: Not stated. Countries: 1. Publications: 3.
Cone Rescue in Retinitis Pigmentosa by the Treatment of Lycium Barbarum
ClinicalTrials.gov study NCT02244996. IPD Sharing: Not stated. Countries: 1. Publications: 1.
Oral N-acetylcysteine for Retinitis Pigmentosa
ClinicalTrials.gov study NCT05537220. IPD Sharing: YES. Countries: 7. Publications: 80.
Trial of Oral Valproic Acid for Retinitis Pigmentosa
ClinicalTrials.gov study NCT01233609. IPD Sharing: Not stated. Countries: 1. Publications: 20.
Transcorneal Electrostimulation for Therapy of Retinitis Pigmentosa
ClinicalTrials.gov study NCT01837901. IPD Sharing: Not stated. Countries: 1. Publications: 6.
Aflibercept for Macular Oedema With Underlying Retinitis Pigmentosa (AMOUR) Study
ClinicalTrials.gov study NCT02661711. IPD Sharing: NO. Countries: 1. Publications: 1.
A Clinical Trial of Retinal Gene Therapy for X-linked Retinitis Pigmentosa Using BIIB112
ClinicalTrials.gov study NCT03116113. IPD Sharing: YES. Countries: 2. Publications: 4.
ScienceDex guides
Understand access before you commit
These curated guides explain access requirements, typical timelines, costs, and reuse considerations for widely used research datasets.
Allen Brain Atlas
Allen Brain Atlas is an Allen Institute collection of brain map atlases, datasets, APIs, and analysis tools covering mouse, human, and non-human primate brain resources.
Annotated Behaviour and Observability Dataset (ABODe)
ABODe is a University of Edinburgh DataShare dataset for behavior classification in group-housed mice using home-cage video, identities, bounding boxes, ground-plate positions, and annotator labels.
DANDI Archive for NWB datasets
DANDI is a BRAIN Initiative archive for publishing and sharing neurophysiology data, including electrophysiology, optophysiology, and behavioral data packaged as NWB and related standards.
International Brain Laboratory public data
The International Brain Laboratory public data releases expose standardized mouse decision-making experiments, including Neuropixels recordings, widefield calcium imaging, behavior, and session metadata accessed through the ONE API.
OpenNeuro
OpenNeuro is a free, open platform for sharing neuroimaging datasets, with public search, dataset pages, and download paths for web, S3, DataLad, and the OpenNeuro CLI.