ArticleScientific reports2016
CRISPR/Cas9 mediated knockout of rb1 and rbl1 leads to rapid and penetrant retinoblastoma development in Xenopus tropicalis.
Article in Scientific reports, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 40 papers.
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Who cites it
40 citing papers in PubMed, 68 citations in OpenAlex.
- Advancements in CRISPR-based therapies for ocular pathologies: from disease mechanisms to intervention strategies.Theranostics · 2026Review
- Establishment and rescue of fibroblast cell lines carrying a nonsense mutation of RB1 by CRISPR-based base editing.Scientific reports · 2025Article
- CRISPR/Cas-mediated mRNA knockdown in the embryos of Xenopus tropicalis.Cell & bioscience · 2025Article
- Systematic review:Frontiers in medicine · 2025Review
- Evolutionary origin of Hoxc13-dependent skin appendages in amphibians.Nature communications · 2024Article
- Mutations in the histone methyltransferase Ezh2 drive context-dependent leukemia in Xenopus tropicalis.Leukemia · 2023Article
- CRISPR/Cas9 systems: Delivery technologies and biomedical applications.Asian journal of pharmaceutical sciences · 2023Review
- The application and progression of CRISPR/Cas9 technology in ophthalmological diseases.Eye (London, England) · 2023Review
- Automated multi-sample DNA extraction for genotyping live Xenopus embryos.Developmental dynamics : an official publication of the American Association of Anatomists · 2023Article
- Review
- Engraftment of Allotransplanted Tumor Cells in AdultCancers · 2022Article
- Identification and functional evaluation of GRIA1 missense and truncation variants in individuals with ID: An emerging neurodevelopmental syndrome.American journal of human genetics · 2022Article
- The Xenopus phenotype ontology: bridging model organism phenotype data to human health and development.BMC bioinformatics · 2022Article
- CRISPR-SID: Identifying EZH2 as a druggable target for desmoid tumors via in vivo dependency mapping.Proceedings of the National Academy of Sciences of the United States of America · 2021Article
- Deep learning is widely applicable to phenotyping embryonic development and disease.Development (Cambridge, England) · 2021Article
- Ttc30a affects tubulin modifications in a model for ciliary chondrodysplasia with polycystic kidney disease.Proceedings of the National Academy of Sciences of the United States of America · 2021Article
- Retinoblastoma tumor cell proliferation is negatively associated with an immune gene expression signature and increased immune cells.Laboratory investigation; a journal of technical methods and pathology · 2021Article
- Genome-editing approaches and applications: a brief review on CRISPR technology and its role in cancer.3 Biotech · 2021Review
- Biallelic variants in COPB1 cause a novel, severe intellectual disability syndrome with cataracts and variable microcephaly.Genome medicine · 2021Article
- Maximizing CRISPR/Cas9 phenotype penetrance applying predictive modeling of editing outcomes in Xenopus and zebrafish embryos.Scientific reports · 2020Article
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Authors and funding
13 authors at 3 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Retinoblastoma is a pediatric eye tumor in which bi-allelic inactivation of the Retinoblastoma 1 (RB1) gene is the initiating genetic lesion. Although recently curative rates of retinoblastoma have increased, there are at this time no molecular targeted therapies available. This is, in part, due to the lack of highly penetrant and rapid retinoblastoma animal models that facilitate rapid identification of targets that allow therapeutic intervention. Different mouse models are available, all based on genetic deactivation of both Rb1 and Retinoblastoma-like 1 (Rbl1), and each showing different kinetics of retinoblastoma development. Here, we show by CRISPR/Cas9 techniques that similar to the mouse, neither rb1 nor rbl1 single mosaic mutant Xenopus tropicalis develop tumors, whereas rb1/rbl1 double mosaic mutant tadpoles rapidly develop retinoblastoma. Moreover, occasionally presence of pinealoblastoma (trilateral retinoblastoma) was detected. We thus present the first CRISPR/Cas9 mediated cancer model in Xenopus tropicalis and the first genuine genetic non-mammalian retinoblastoma model. The rapid kinetics of our model paves the way for use as a pre-clinical model. Additionally, this retinoblastoma model provides unique possibilities for fast elucidation of novel drug targets by triple multiplex CRISPR/Cas9 gRNA injections (rb1 + rbl1 + modifier gene) in order to address the clinically unmet need of targeted retinoblastoma therapy.
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