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Matilda Ziegler Foundation"},{"name":"Rebecca Meyer Brown Professorship"},{"name":"UW-ICTR NIH","award":["1UL1RR025011"],"award-info":[{"award-number":["1UL1RR025011"]}]},{"name":"UW Eye Research Institute\/RRF Murfee Chair","id":[{"id":"https:\/\/ror.org\/05783y657","id-type":"ROR","asserted-by":"crossref"}]},{"name":"NHMRC Overseas Biomedical Fellowship"},{"name":"NEI or NIH"},{"name":"Department of Biology, Indiana University-Purdue University Indianapolis, Indianapolis, IN","id":[{"id":"https:\/\/ror.org\/05gxnyn08","id-type":"ROR","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2011,8,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Differentiation methods for human induced pluripotent stem cells (hiPSCs) typically yield progeny from multiple tissue lineages, limiting their use for drug testing and autologous cell transplantation. In particular, early retina and forebrain derivatives often intermingle in pluripotent stem cell cultures, owing to their shared ancestry and tightly coupled development. Here, we demonstrate that three-dimensional populations of retinal progenitor cells (RPCs) can be isolated from early forebrain populations in both human embryonic stem cell and hiPSC cultures, providing a valuable tool for developmental, functional, and translational studies. Using our established protocol, we identified a transient population of optic vesicle (OV)-like structures that arose during a time period appropriate for normal human retinogenesis. These structures were independently cultured and analyzed to confirm their multipotent RPC status and capacity to produce physiologically responsive retinal cell types, including photoreceptors and retinal pigment epithelium (RPE). We then applied this method to hiPSCs derived from a patient with gyrate atrophy, a retinal degenerative disease affecting the RPE. RPE generated from these hiPSCs exhibited a disease-specific functional defect that could be corrected either by pharmacological means or following targeted gene repair. The production of OV-like populations from human pluripotent stem cells should facilitate the study of human retinal development and disease and advance the use of hiPSCs in personalized medicine.<\/jats:p>","DOI":"10.1002\/stem.674","type":"journal-article","created":{"date-parts":[[2011,6,15]],"date-time":"2011-06-15T09:11:46Z","timestamp":1308129106000},"page":"1206-1218","source":"Crossref","is-referenced-by-count":438,"title":["Optic Vesicle-like Structures Derived from Human Pluripotent Stem Cells Facilitate a Customized Approach to Retinal Disease Treatment"],"prefix":"10.1093","volume":"29","author":[{"given":"Jason S.","family":"Meyer","sequence":"first","affiliation":[{"name":"Waisman Center, University of Wisconsin, Madison, Wisconsin, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sara E.","family":"Howden","sequence":"additional","affiliation":[{"name":"Department of Cell and Regenerative Biology, University of Wisconsin, Madison, Wisconsin, 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USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bikash","family":"Pattnaik","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology and Visual Sciences, University of Wisconsin, Madison, Wisconsin, USA"},{"name":"Department of Pediatrics, and University of Wisconsin, Madison, Wisconsin, USA"},{"name":"Eye Research Institute, University of Wisconsin, Madison, Wisconsin, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"James A.","family":"Thomson","sequence":"additional","affiliation":[{"name":"Department of Cell and Regenerative Biology, University of Wisconsin, Madison, Wisconsin, USA"},{"name":"The Genome Center of Wisconsin, University of Wisconsin, Madison, Wisconsin, USA"},{"name":"Morgridge Institute for Research, Madison, Wisconsin, USA"},{"name":"Department of Molecular, Cellular, and Developmental Biology, University of California Santa Barbara, Santa Barbara, California, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David M.","family":"Gamm","sequence":"additional","affiliation":[{"name":"Waisman Center, University of Wisconsin, Madison, Wisconsin, USA"},{"name":"Department of Ophthalmology and Visual Sciences, University of Wisconsin, Madison, Wisconsin, USA"},{"name":"Eye Research Institute, University of Wisconsin, Madison, Wisconsin, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2011,7,26]]},"reference":[{"key":"2022011210470814100_bib1","doi-asserted-by":"crossref","first-page":"1129","DOI":"10.1101\/gad.1303605","article-title":"Embryonic stem cell differentiation: Emergence of a new era in biology and medicine","volume":"19","author":"Keller","year":"2005","journal-title":"Genes Dev"},{"key":"2022011210470814100_bib2","doi-asserted-by":"crossref","first-page":"5515","DOI":"10.1242\/dev.01451","article-title":"Human embryonic stem cells: Prospects for 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