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@ARTICLE{Gasparini:164106,
author = {Gasparini, Sylvia J and Tessmer, Karen and Reh, Miriam and
Wieneke, Stephanie and Carido, Madalena and Völkner,
Manuela and Borsch, Oliver and Swiersy, Anka and Zuzic,
Marta and Goureau, Olivier and Kurth, Thomas and Busskamp,
Volker and Zeck, Günther and Karl, Mike Oliver and Ader,
Marius},
title = {{T}ransplanted human cones incorporate and function in a
murine cone degeneration model.},
journal = {The journal of clinical investigation},
volume = {132},
number = {12},
issn = {0021-9738},
address = {Ann Arbor, Mich.},
publisher = {ASCJ},
reportid = {DZNE-2022-00769},
pages = {e154619},
year = {2022},
note = {CC BY},
abstract = {Once human photoreceptors die, they do not regenerate, thus
photoreceptor transplantation has emerged as a potential
treatment approach for blinding diseases. Improvements in
transplant organization, donor cell maturation and synaptic
connectivity to the host will be critical in advancing this
technology to clinical practice. Unlike the unstructured
grafts of prior cell suspension transplantations into
end-stage degeneration models, we describe extensive
incorporation of iPSC retinal organoid-derived human
photoreceptors into mice with cone dysfunction. This
incorporative phenotype was validated in both cone-only as
well as pan-photoreceptor transplantations. Rather than
forming a glial barrier, Müller cells extended throughout
the graft, even forming a series of adherens junctions
between mouse and human cells, reminiscent of an outer
limiting membrane. Donor-host interaction appeared to
promote polarisation as well as development of morphological
features critical for light detection, namely formation of
inner and well stacked outer segments oriented towards the
retinal pigment epithelium. Putative synapse formation and
graft function was evident both at a structural and
electrophysiological level. Overall, these results show that
human photoreceptors interact readily with a partially
degenerated retina. Moreover, incorporation into the host
retina appears to be beneficial to graft maturation,
polarisation and function.},
keywords = {Animals / Ependymoglial Cells / Humans / Induced
Pluripotent Stem Cells: transplantation / Mice /
Photoreceptor Cells, Vertebrate: metabolism / Retina:
metabolism / Retinal Cone Photoreceptor Cells / Retinal
Degeneration: metabolism / Retinal Degeneration: therapy /
Human stem cells (Other) / Retinopathy (Other) / Stem cell
transplantation (Other) / Stem cells (Other) /
Transplantation (Other)},
cin = {Cell Culture Platform / AG Karl},
ddc = {610},
cid = {I:(DE-2719)1740003 / I:(DE-2719)1710004},
pnm = {352 - Disease Mechanisms (POF4-352)},
pid = {G:(DE-HGF)POF4-352},
typ = {PUB:(DE-HGF)16},
pmc = {pmc:PMC9197520},
pubmed = {pmid:35482419},
doi = {10.1172/JCI154619},
url = {https://pub.dzne.de/record/164106},
}