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This review focuses on the initial steps leading to the seminal discoveries of proteoglycogen and glycogenin at the beginning of the 1980s, which paved the way for subsequent foundational breakthroughs that propelled forward this new research field. We also explore the current, as well as potential, impact this research field is having on human health and disease from the perspective of glycogen storage diseases. Important new questions arising from recent studies, their links to basic mechanisms involved in the de novo glycogen biogenesis, and the pervading presence of glycogenin across the evolutionary scale, fueled by high throughput -omics technologies, are also addressed.<\/jats:p>","DOI":"10.1042\/bcj20190441","type":"journal-article","created":{"date-parts":[[2019,11,5]],"date-time":"2019-11-05T17:34:08Z","timestamp":1572975248000},"page":"3109-3124","update-policy":"https:\/\/doi.org\/10.1042\/crossmark_policy","source":"Crossref","is-referenced-by-count":11,"title":["From the seminal discovery of proteoglycogen and glycogenin to emerging knowledge and research on glycogen biology"],"prefix":"10.1042","volume":"476","author":[{"given":"Juan A.","family":"Curtino","sequence":"first","affiliation":[{"name":"CIQUIBIC (CONICET), Departamento de Qu\u00edmica Biol\u00f3gica Ranwel Caputto, Facultad de Ciencias Qu\u00edmicas, Universidad Nacional de C\u00f3rdoba, C\u00f3rdoba, Argentina"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4355-5431","authenticated-orcid":false,"given":"Miguel A.","family":"Aon","sequence":"additional","affiliation":[{"name":"Laboratory of Cardiovascular Science, National Institute on Aging, National Institutes of Health, Baltimore, MD, U.S.A."},{"name":"Translational Gerontology Branch, National Institute on Aging, National Institutes of Health, Baltimore, MD, U.S.A."}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"288","published-online":{"date-parts":[[2019,11,5]]},"reference":[{"key":"2021112218030893300_BCJ-2019-0441C1","first-page":"103","article-title":"Protein binding of physiologically important substances. 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