{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,4]],"date-time":"2026-08-04T14:57:18Z","timestamp":1785855438602,"version":"3.56.0"},"reference-count":145,"publisher":"MDPI AG","issue":"19","license":[{"start":{"date-parts":[[2024,10,7]],"date-time":"2024-10-07T00:00:00Z","timestamp":1728259200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"State Government of Victoria\u2019s Operational Infrastructure Support Program","award":["2018-16"],"award-info":[{"award-number":["2018-16"]}]},{"name":"State Government of Victoria\u2019s Operational Infrastructure Support Program","award":["MDBR-20-106-CDKL5"],"award-info":[{"award-number":["MDBR-20-106-CDKL5"]}]},{"name":"State Government of Victoria\u2019s Operational Infrastructure Support Program","award":["MRF2007465"],"award-info":[{"award-number":["MRF2007465"]}]},{"name":"Foundation for Children Project","award":["2018-16"],"award-info":[{"award-number":["2018-16"]}]},{"name":"Foundation for Children Project","award":["MDBR-20-106-CDKL5"],"award-info":[{"award-number":["MDBR-20-106-CDKL5"]}]},{"name":"Foundation for Children Project","award":["MRF2007465"],"award-info":[{"award-number":["MRF2007465"]}]},{"name":"Murdoch Children\u2019s Research Institute Strategic Pilot Project in Stem Cell and Genomic Medicine Research","award":["2018-16"],"award-info":[{"award-number":["2018-16"]}]},{"name":"Murdoch Children\u2019s Research Institute Strategic Pilot Project in Stem Cell and Genomic Medicine Research","award":["MDBR-20-106-CDKL5"],"award-info":[{"award-number":["MDBR-20-106-CDKL5"]}]},{"name":"Murdoch Children\u2019s Research Institute Strategic Pilot Project in Stem Cell and Genomic Medicine Research","award":["MRF2007465"],"award-info":[{"award-number":["MRF2007465"]}]},{"name":"Orphan Disease Center of the University of Pennsylvania","award":["2018-16"],"award-info":[{"award-number":["2018-16"]}]},{"name":"Orphan Disease Center of the University of Pennsylvania","award":["MDBR-20-106-CDKL5"],"award-info":[{"award-number":["MDBR-20-106-CDKL5"]}]},{"name":"Orphan Disease Center of the University of Pennsylvania","award":["MRF2007465"],"award-info":[{"award-number":["MRF2007465"]}]},{"name":"CDKL5 Forum Junior Fellowship from the Loulou Foundation","award":["2018-16"],"award-info":[{"award-number":["2018-16"]}]},{"name":"CDKL5 Forum Junior Fellowship from the Loulou Foundation","award":["MDBR-20-106-CDKL5"],"award-info":[{"award-number":["MDBR-20-106-CDKL5"]}]},{"name":"CDKL5 Forum Junior Fellowship from the Loulou Foundation","award":["MRF2007465"],"award-info":[{"award-number":["MRF2007465"]}]},{"name":"Medical Research Future Funds (MRFF) Stem Cell Therapies Mission","award":["2018-16"],"award-info":[{"award-number":["2018-16"]}]},{"name":"Medical Research Future Funds (MRFF) Stem Cell Therapies Mission","award":["MDBR-20-106-CDKL5"],"award-info":[{"award-number":["MDBR-20-106-CDKL5"]}]},{"name":"Medical Research Future Funds (MRFF) Stem Cell Therapies Mission","award":["MRF2007465"],"award-info":[{"award-number":["MRF2007465"]}]},{"name":"MCRI Near Miss","award":["2018-16"],"award-info":[{"award-number":["2018-16"]}]},{"name":"MCRI Near Miss","award":["MDBR-20-106-CDKL5"],"award-info":[{"award-number":["MDBR-20-106-CDKL5"]}]},{"name":"MCRI Near Miss","award":["MRF2007465"],"award-info":[{"award-number":["MRF2007465"]}]},{"name":"MCRI Strategic Pilot Project in Stem Cell and Genomics Medicine and the VESKI Near Miss Pilot Award","award":["2018-16"],"award-info":[{"award-number":["2018-16"]}]},{"name":"MCRI Strategic Pilot Project in Stem Cell and Genomics Medicine and the VESKI Near Miss Pilot Award","award":["MDBR-20-106-CDKL5"],"award-info":[{"award-number":["MDBR-20-106-CDKL5"]}]},{"name":"MCRI Strategic Pilot Project in Stem Cell and Genomics Medicine and the VESKI Near Miss Pilot Award","award":["MRF2007465"],"award-info":[{"award-number":["MRF2007465"]}]},{"name":"The Royal Children\u2019s Hospital Foundation","award":["2018-16"],"award-info":[{"award-number":["2018-16"]}]},{"name":"The Royal Children\u2019s Hospital Foundation","award":["MDBR-20-106-CDKL5"],"award-info":[{"award-number":["MDBR-20-106-CDKL5"]}]},{"name":"The Royal Children\u2019s Hospital Foundation","award":["MRF2007465"],"award-info":[{"award-number":["MRF2007465"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["IJMS"],"abstract":"<jats:p>Pediatric genetic epilepsies, such as CDKL5 Deficiency Disorder (CDD), are severely debilitating, with early-onset seizures occurring more than ten times daily in extreme cases. Existing antiseizure drugs frequently prove ineffective, which significantly impacts child development and diminishes the quality of life for patients and caregivers. The relaxation of cannabis legislation has increased research into potential therapeutic properties of phytocannabinoids such as cannabidiol (CBD) and \u03949-tetrahydrocannabinol (THC). CBD\u2019s antiseizure properties have shown promise, particularly in treating drug-resistant genetic epilepsies associated with Lennox\u2013Gastaut syndrome (LGS), Dravet syndrome (DS), and Tuberous Sclerosis Complex (TSC). However, specific research on CDD remains limited. Much of the current evidence relies on anecdotal reports of artisanal products lacking accurate data on cannabinoid composition. Utilizing model systems like patient-derived iPSC neurons and brain organoids allows precise dosing and comprehensive exploration of cannabinoids\u2019 pharmacodynamics. This review explores the potential of CBD, THC, and other trace cannabinoids in treating CDD and focusing on clinical trials and preclinical models to elucidate the cannabinoid\u2019s potential mechanisms of action in disrupted CDD pathways and strengthen the case for further research into their potential as anti-epileptic drugs for CDD. This review offers an updated perspective on cannabinoid\u2019s therapeutic potential for CDD.<\/jats:p>","DOI":"10.3390\/ijms251910768","type":"journal-article","created":{"date-parts":[[2024,10,7]],"date-time":"2024-10-07T06:23:18Z","timestamp":1728282198000},"page":"10768","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":10,"title":["Cannabinoids and Genetic Epilepsy Models: A Review with Focus on CDKL5 Deficiency Disorder"],"prefix":"10.3390","volume":"25","author":[{"given":"Sean","family":"Massey","sequence":"first","affiliation":[{"name":"Brain and Mitochondrial Research Group, Murdoch Children\u2019s Research Institute, Royal Children\u2019s Hospital, Melbourne, VIC 3052, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anita","family":"Quigley","sequence":"additional","affiliation":[{"name":"Electrical and Biomedical Engineering, School of Engineering, RMIT University, Melbourne, VIC 3000, Australia"},{"name":"Aikenhead Centre for Medical Discovery, St. Vincent\u2019s Hospital, Melbourne, VIC 3065, Australia"},{"name":"Centre for Clinical Neuroscience and Neurological Research, St. Vincent\u2019s Hospital, Melbourne, VIC 3065, Australia"},{"name":"Department of Medicine, University of Melbourne, Melbourne, VIC 3065, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8442-6081","authenticated-orcid":false,"given":"Simone","family":"Rochfort","sequence":"additional","affiliation":[{"name":"School of Applied Systems Biology, La Trobe University, Bundoora, VIC 3083, Australia"},{"name":"Agriculture Victoria Research, AgriBio Centre, AgriBio, Melbourne, VIC 3083, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8431-0641","authenticated-orcid":false,"given":"John","family":"Christodoulou","sequence":"additional","affiliation":[{"name":"Brain and Mitochondrial Research Group, Murdoch Children\u2019s Research Institute, Royal Children\u2019s Hospital, Melbourne, VIC 3052, Australia"},{"name":"Department of Paediatrics, University of Melbourne, Melbourne, VIC 3052, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6768-3665","authenticated-orcid":false,"given":"Nicole J.","family":"Van Bergen","sequence":"additional","affiliation":[{"name":"Brain and Mitochondrial Research Group, Murdoch Children\u2019s Research Institute, Royal Children\u2019s Hospital, Melbourne, VIC 3052, Australia"},{"name":"Department of Paediatrics, University of Melbourne, Melbourne, VIC 3052, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2024,10,7]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"1377","DOI":"10.1016\/j.bmc.2015.01.059","article-title":"Cannabidiol (CBD) and its analogs: A review of their effects on inflammation","volume":"23","author":"Burstein","year":"2015","journal-title":"Bioorg. 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