{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,29]],"date-time":"2026-09-29T19:17:57Z","timestamp":1790709477406,"version":"4.1.0"},"reference-count":111,"publisher":"Portland Press Ltd.","issue":"2","license":[{"start":{"date-parts":[[2024,3,25]],"date-time":"2024-03-25T00:00:00Z","timestamp":1711324800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["portlandpress.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2024,4,24]]},"abstract":"<jats:p>The genetic landscape of neurodegenerative diseases encompasses genes affecting multiple cellular pathways which exert effects in an array of neuronal and glial cell-types. Deconvolution of the roles of genes implicated in disease and the effects of disease-associated variants remains a vital step in the understanding of neurodegeneration and the development of therapeutics. Disease modelling using patient induced pluripotent stem cells (iPSCs) has enabled the generation of key cell-types associated with disease whilst maintaining the genomic variants that predispose to neurodegeneration. The use of CRISPR interference (CRISPRi), alongside other CRISPR-perturbations, allows the modelling of the effects of these disease-associated variants or identifying genes which modify disease phenotypes. This review summarises the current applications of CRISPRi in iPSC-derived neuronal models, such as fluorescence-activated cell sorting (FACS)-based screens, and discusses the future opportunities for disease modelling, identification of disease risk modifiers and target\/drug discovery in neurodegeneration.<\/jats:p>","DOI":"10.1042\/bst20230190","type":"journal-article","created":{"date-parts":[[2024,3,25]],"date-time":"2024-03-25T13:49:56Z","timestamp":1711374596000},"page":"539-551","update-policy":"https:\/\/doi.org\/10.1042\/crossmark_policy","source":"Crossref","is-referenced-by-count":11,"title":["CRISPRi: a way to integrate iPSC-derived neuronal models"],"prefix":"10.1042","volume":"52","author":[{"ORCID":"https:\/\/orcid.org\/0009-0007-8611-0161","authenticated-orcid":false,"given":"Sarah N.J.","family":"Franks","sequence":"first","affiliation":[{"name":"1Oxford Parkinson's Disease Centre and Department of Physiology, Anatomy and Genetics, University of Oxford, Oxford OX1 3QU, UK"},{"name":"2Kavli Institute for Nanoscience Discovery, Dorothy Crowfoot Hodgkin Building, University of Oxford, Oxford OX1 3QU, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9723-2227","authenticated-orcid":false,"given":"Rachel","family":"Heon-Roberts","sequence":"additional","affiliation":[{"name":"1Oxford Parkinson's Disease Centre and Department of Physiology, Anatomy and Genetics, University of Oxford, Oxford OX1 3QU, UK"},{"name":"2Kavli Institute for Nanoscience Discovery, Dorothy Crowfoot Hodgkin Building, University of Oxford, Oxford OX1 3QU, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6703-3718","authenticated-orcid":false,"given":"Brent J.","family":"Ryan","sequence":"additional","affiliation":[{"name":"1Oxford Parkinson's Disease Centre and Department of Physiology, Anatomy and Genetics, University of Oxford, Oxford OX1 3QU, UK"},{"name":"2Kavli Institute for Nanoscience Discovery, Dorothy Crowfoot Hodgkin Building, University of Oxford, Oxford OX1 3QU, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"288","published-online":{"date-parts":[[2024,3,25]]},"reference":[{"key":"2024042414455833000_BST-2023-0190CC1","doi-asserted-by":"publisher","first-page":"331","DOI":"10.1038\/nature10886","article-title":"RNA-guided genetic silencing systems in bacteria and archaea","volume":"482","year":"2012","journal-title":"Nature"},{"key":"2024042414455833000_BST-2023-0190CC2","doi-asserted-by":"publisher","first-page":"823","DOI":"10.1126\/science.1232033","article-title":"RNA-guided human genome engineering via Cas9","volume":"339","year":"2013","journal-title":"Science"},{"key":"2024042414455833000_BST-2023-0190CC3","doi-asserted-by":"publisher","first-page":"230","DOI":"10.1038\/nbt.2507","article-title":"Targeted genome engineering in human cells with the Cas9 RNA-guided endonuclease","volume":"31","year":"2013","journal-title":"Nat. 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