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We demonstrate a potentially novel application of adaptive optics fluorescence microscopy to visualize and track the in situ mosaicism of retinal pigment epithelial (RPE) cells directly in the human eye. After a short, dynamic period during which RPE cells take up i.v.-administered indocyanine green (ICG) dye, we observed a remarkably stable heterogeneity in the fluorescent pattern that gradually disappeared over a period of days. This pattern could be robustly reproduced with a new injection and follow-up imaging in the same eye out to at least 12 months, which enabled longitudinal tracking of RPE cells. Investigation of ICG uptake in primary human RPE cells and in a mouse model of ICG uptake alongside human imaging corroborated our findings that the observed mosaicism is an intrinsic property of the RPE tissue. We demonstrate a potentially novel application of fluorescence microscopy to detect subclinical changes to the RPE, a technical advance that has direct implications for improving our understanding of diseases such as oculocutaneous albinism, late-onset retinal degeneration, and Bietti crystalline dystrophy.<\/jats:p>","DOI":"10.1172\/jci.insight.124904","type":"journal-article","created":{"date-parts":[[2019,3,23]],"date-time":"2019-03-23T09:59:51Z","timestamp":1553335191000},"source":"Crossref","is-referenced-by-count":35,"title":["Longitudinal adaptive optics fluorescence microscopy reveals cellular mosaicism in patients"],"prefix":"10.1172","volume":"4","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-4621-427X","authenticated-orcid":true,"given":"HaeWon","family":"Jung","sequence":"first","affiliation":[{"name":"National Eye Institute, NIH, Bethesda, Maryland, 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