{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,14]],"date-time":"2026-10-14T09:51:07Z","timestamp":1791971467289,"version":"4.3.4"},"reference-count":139,"publisher":"The Company of Biologists","issue":"5","license":[{"start":{"date-parts":[[2024,6,4]],"date-time":"2024-06-04T00:00:00Z","timestamp":1717459200000},"content-version":"vor","delay-in-days":34,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"crossref","award":["EY017400 and EY022954"],"award-info":[{"award-number":["EY017400 and EY022954"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]},{"name":"Medical Research Council","award":["MR\/V012177\/1"],"award-info":[{"award-number":["MR\/V012177\/1"]}]},{"name":"Medical Research Council","award":["EP\/Y01720X\/1"],"award-info":[{"award-number":["EP\/Y01720X\/1"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2024,5,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:p>Recent progress in human disease genetics is leading to rapid advances in understanding pathobiological mechanisms. However, the sheer number of risk-conveying genetic variants being identified demands in vivo model systems that are amenable to functional analyses at scale. Here we provide a practical guide for using the diploid frog species Xenopus tropicalis to study many genes and variants to uncover conserved mechanisms of pathobiology relevant to human disease. We discuss key considerations in modelling human genetic disorders: genetic architecture, conservation, phenotyping strategy and rigour, as well as more complex topics, such as penetrance, expressivity, sex differences and current challenges in the field. As the patient-driven gene discovery field expands significantly, the cost-effective, rapid and higher throughput nature of Xenopus make it an essential member of the model organism armamentarium for understanding gene function in development and in relation to disease.<\/jats:p>","DOI":"10.1242\/dmm.050754","type":"journal-article","created":{"date-parts":[[2024,6,4]],"date-time":"2024-06-04T08:20:36Z","timestamp":1717489236000},"source":"Crossref","is-referenced-by-count":13,"title":["Modelling human genetic disorders in <i>Xenopus tropicalis<\/i>"],"prefix":"10.1242","volume":"17","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-8404-3291","authenticated-orcid":false,"given":"Helen Rankin","family":"Willsey","sequence":"first","affiliation":[{"name":"Weill Institute for Neurosciences, University of California San Francisco 1 Department of Psychiatry and Behavioral Sciences , , San Francisco, CA 94158 , USA"},{"name":"Chan Zuckerberg Biohub - San Francisco 2 , San Francisco, CA 94518 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6814-8648","authenticated-orcid":false,"given":"Eleanor G.","family":"Seaby","sequence":"additional","affiliation":[{"name":"University of Southampton 3 Genomic Informatics Group, Faculty of Medicine , , Southampton SO16 6YD , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5264-7824","authenticated-orcid":false,"given":"Annie","family":"Godwin","sequence":"additional","affiliation":[{"name":"European Xenopus Resource Centre (EXRC), School of Biological Sciences, University of Portsmouth 4 , Portsmouth PO1 2DY , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2648-0869","authenticated-orcid":false,"given":"Sarah","family":"Ennis","sequence":"additional","affiliation":[{"name":"University of Southampton 3 Genomic Informatics Group, Faculty of Medicine , , Southampton SO16 6YD , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6865-4519","authenticated-orcid":false,"given":"Matthew","family":"Guille","sequence":"additional","affiliation":[{"name":"European Xenopus Resource Centre (EXRC), School of Biological Sciences, University of Portsmouth 4 , Portsmouth PO1 2DY , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4539-5280","authenticated-orcid":false,"given":"Robert M.","family":"Grainger","sequence":"additional","affiliation":[{"name":"University of Virginia 5 Department of Biology , , Charlottesville, VA 22904 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2024,6,4]]},"reference":[{"key":"2024060408104402800_DMM050754C1","doi-asserted-by":"publisher","first-page":"111","DOI":"10.1007\/978-1-0716-3004-4_10","article-title":"CRISPR\/Cas9 gene disruption studies in F0 Xenopus tadpoles: understanding development and disease in the frog","volume":"2633","author":"Abu-Daya","year":"2023","journal-title":"Methods Mol. 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