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@ARTICLE{Carido:265755,
author = {Carido, Madalena and Völkner, Manuela and Steinheuer, Lisa
Maria and Wagner, Felix and Kurth, Thomas and Dumler,
Natalie and Ulusoy, Selen and Wieneke, Stephanie and
Norniella, Anabel Villanueva and Golfieri, Cristina and
Khattak, Shahryar and Schoenfelder, Bruno and Scamozzi,
Maria and Zoschke, Katja and Canzler, Sebastian and
Hackermüller, Jörg and Ader, Marius and Karl, Mike O},
title = {{R}eliability of human retina organoid generation from
hi{PSC}-derived neuroepithelial cysts.},
journal = {Frontiers in cellular neuroscience},
volume = {17},
issn = {1662-5102},
address = {Lausanne},
publisher = {Frontiers Research Foundation},
reportid = {DZNE-2023-01030},
pages = {1166641},
year = {2023},
abstract = {The possible applications for human retinal organoids
(HROs) derived from human induced pluripotent stem cells
(hiPSC) rely on the robustness and transferability of the
methodology for their generation. Standardized strategies
and parameters to effectively assess, compare, and optimize
organoid protocols are starting to be established, but are
not yet complete. To advance this, we explored the
efficiency and reliability of a differentiation method,
called CYST protocol, that facilitates retina generation by
forming neuroepithelial cysts from hiPSC clusters. Here, we
tested seven different hiPSC lines which reproducibly
generated HROs. Histological and ultrastructural analyses
indicate that HRO differentiation and maturation are
regulated. The different hiPSC lines appeared to be a larger
source of variance than experimental rounds. Although
previous reports have shown that HROs in several other
protocols contain a rather low number of cones, HROs from
the CYST protocol are consistently richer in cones and with
a comparable ratio of cones, rods, and Müller glia. To
provide further insight into HRO cell composition, we
studied single cell RNA sequencing data and applied CaSTLe,
a transfer learning approach. Additionally, we devised a
potential strategy to systematically evaluate different
organoid protocols side-by-side through parallel
differentiation from the same hiPSC batches: In an
explorative study, the CYST protocol was compared to a
conceptually different protocol based on the formation of
cell aggregates from single hiPSCs. Comparing four hiPSC
lines showed that both protocols reproduced key
characteristics of retinal epithelial structure and cell
composition, but the CYST protocol provided a higher HRO
yield. So far, our data suggest that CYST-derived HROs
remained stable up to at least day 200, while single
hiPSC-derived HROs showed spontaneous pathologic changes by
day 200. Overall, our data provide insights into the
efficiency, reproducibility, and stability of the CYST
protocol for generating HROs, which will be useful for
further optimizing organoid systems, as well as for basic
and translational research applications.},
keywords = {gliosis (Other) / hiPSC (Other) / human (Other) /
neurodevelopment (Other) / organoid (Other) / pathology
(Other) / retina (Other) / stem cells (Other)},
cin = {AG Karl},
ddc = {610},
cid = {I:(DE-2719)1710004},
pnm = {352 - Disease Mechanisms (POF4-352)},
pid = {G:(DE-HGF)POF4-352},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:37868194},
pmc = {pmc:PMC10587494},
doi = {10.3389/fncel.2023.1166641},
url = {https://pub.dzne.de/record/265755},
}