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Telomerase plays key roles in maintaining genome stability; and its dysfunction and misregulation have been linked to different types of cancers and a spectrum of human genetic disorders. Over the years, a wealth of genetic and biochemical studies of human telomerase have illuminated its numerous fascinating features. Yet, structural studies of human telomerase have lagged behind due to various challenges. Recent technical developments in cryo-electron microscopy have allowed for the first detailed visualization of the human telomerase holoenzyme, revealing unprecedented insights into its active site and assembly. This review summarizes the cumulative work leading to the recent structural advances, as well as highlights how the future structural work will further advance our understanding of this enzyme.<\/jats:p>","DOI":"10.1042\/bst20200042","type":"journal-article","created":{"date-parts":[[2021,10,10]],"date-time":"2021-10-10T22:29:15Z","timestamp":1633904955000},"page":"1927-1939","update-policy":"https:\/\/doi.org\/10.1042\/crossmark_policy","source":"Crossref","is-referenced-by-count":19,"title":["Structural biology of human telomerase: progress and prospects"],"prefix":"10.1042","volume":"49","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-2180-3430","authenticated-orcid":false,"given":"Thi Hoang Duong","family":"Nguyen","sequence":"first","affiliation":[{"name":"Structural Studies Division, Medical Research Council Laboratory of Molecular Biology, Cambridge, U.K."}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"288","published-online":{"date-parts":[[2021,10,8]]},"reference":[{"key":"2021111916145460100_BST-2020-0042CC1","doi-asserted-by":"publisher","first-page":"405","DOI":"10.1073\/pnas.25.8.405","article-title":"The behavior in successive nuclear divisions of a chromosome broken at meiosis","volume":"25","year":"1939","journal-title":"Proc. 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