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It is a complex condition which results in multiple lifelong health problems, including varying degrees of intellectual disability and delays in speech, memory, and learning. As both length and quality of life are improving for individuals with Down syndrome, attention is now being directed to understanding and potentially treating the associated cognitive difficulties and their underlying biological substrates. These have included imaging and postmortem studies which have identified decreased regional brain volumes and histological anomalies that accompany early onset dementia. In addition, advances in genome\u2010wide analysis and Down syndrome mouse models are providing valuable insight into potential targets for intervention that could improve neurogenesis and long\u2010term cognition. As little is known about early brain development in human Down syndrome, we review recent advances in magnetic resonance imaging that allow non\u2010invasive visualization of brain macro\u2010 and microstructure, even in utero. It is hoped that together these advances may enable Down syndrome to become one of the first genetic disorders to be targeted by antenatal treatments designed to \u2018normalize\u2019 brain development.<\/jats:p><\/jats:sec><jats:sec><jats:title>What this paper adds<\/jats:title><jats:p>\n<jats:list list-type=\"bullet\">\n\n<jats:list-item><jats:p>Magnetic resonance imaging can provide non\u2010invasive characterization of early brain development in Down syndrome.<\/jats:p><\/jats:list-item>\n\n<jats:list-item><jats:p>Down syndrome mouse models enable study of underlying pathology and potential intervention strategies.<\/jats:p><\/jats:list-item>\n\n<jats:list-item><jats:p>Potential therapies could modify brain structure and improve early cognitive levels.<\/jats:p><\/jats:list-item>\n\n<jats:list-item><jats:p>Down syndrome may be the first genetic disorder to have targeted therapies which alter antenatal brain development.<\/jats:p><\/jats:list-item>\n<\/jats:list>\n<\/jats:p><\/jats:sec>","DOI":"10.1111\/dmcn.14260","type":"journal-article","created":{"date-parts":[[2019,5,18]],"date-time":"2019-05-18T05:29:14Z","timestamp":1558157354000},"page":"867-879","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":65,"title":["New approaches to studying early brain development in\u00a0Down\u00a0syndrome"],"prefix":"10.1111","volume":"61","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-7537-6534","authenticated-orcid":false,"given":"Ana A","family":"Baburamani","sequence":"first","affiliation":[{"name":"Centre for the Developing Brain Department of Perinatal Imaging and Health School of Biomedical Engineering &amp; Imaging Sciences King's College London King's Health Partners St Thomas\u2019 Hospital London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Prachi 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