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      Differentiation of human ESCs to retinal ganglion cells using a CRISPR engineered reporter cell line

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          Abstract

          Retinal ganglion cell (RGC) injury and cell death from glaucoma and other forms of optic nerve disease is a major cause of irreversible vision loss and blindness. Human pluripotent stem cell (hPSC)-derived RGCs could provide a source of cells for the development of novel therapeutic molecules as well as for potential cell-based therapies. In addition, such cells could provide insights into human RGC development, gene regulation, and neuronal biology. Here, we report a simple, adherent cell culture protocol for differentiation of hPSCs to RGCs using a CRISPR-engineered RGC fluorescent reporter stem cell line. Fluorescence-activated cell sorting of the differentiated cultures yields a highly purified population of cells that express a range of RGC-enriched markers and exhibit morphological and physiological properties typical of RGCs. Additionally, we demonstrate that aligned nanofiber matrices can be used to guide the axonal outgrowth of hPSC-derived RGCs for in vitro optic nerve-like modeling. Lastly, using this protocol we identified forskolin as a potent promoter of RGC differentiation.

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          Transcellular degradation of axonal mitochondria.

          It is generally accepted that healthy cells degrade their own mitochondria. Here, we report that retinal ganglion cell axons of WT mice shed mitochondria at the optic nerve head (ONH), and that these mitochondria are internalized and degraded by adjacent astrocytes. EM demonstrates that mitochondria are shed through formation of large protrusions that originate from otherwise healthy axons. A virally introduced tandem fluorophore protein reporter of acidified mitochondria reveals that acidified axonal mitochondria originating from the retinal ganglion cell are associated with lysosomes within columns of astrocytes in the ONH. According to this reporter, a greater proportion of retinal ganglion cell mitochondria are degraded at the ONH than in the ganglion cell soma. Consistently, analyses of degrading DNA reveal extensive mtDNA degradation within the optic nerve astrocytes, some of which comes from retinal ganglion cell axons. Together, these results demonstrate that surprisingly large proportions of retinal ganglion cell axonal mitochondria are normally degraded by the astrocytes of the ONH. This transcellular degradation of mitochondria, or transmitophagy, likely occurs elsewhere in the CNS, because structurally similar accumulations of degrading mitochondria are also found along neurites in superficial layers of the cerebral cortex. Thus, the general assumption that neurons or other cells necessarily degrade their own mitochondria should be reconsidered.
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            Modeling early retinal development with human embryonic and induced pluripotent stem cells.

            Human pluripotent stem cells have the potential to provide comprehensive model systems for the earliest stages of human ontogenesis. To serve in this capacity, these cells must undergo a targeted, stepwise differentiation process that follows a normal developmental timeline. Here we demonstrate the ability of both human embryonic stem cells (hESCs) and induced pluripotent stem (iPS) cells to meet these requirements for human retinogenesis. Upon differentiation, hESCs initially yielded a highly enriched population of early eye field cells. Thereafter, a subset of cells acquired features of advancing retinal differentiation in a sequence and time course that mimicked in vivo human retinal development. Application of this culture method to a human iPS cell line also generated retina-specific cell types at comparable times in vitro. Lastly, altering endogenous signaling during differentiation affected lineage-specific gene expression in a manner consistent with established mechanisms of early neural and retinal cell fate determination. These findings should aid in the investigation of the molecular events governing retinal specification from human pluripotent stem cells.
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              Glaucoma is second leading cause of blindness globally

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                Author and article information

                Journal
                Sci Rep
                Sci Rep
                Scientific Reports
                Nature Publishing Group
                2045-2322
                13 November 2015
                2015
                : 5
                : 16595
                Affiliations
                [1 ]Department of Molecular Biology and Genetics, Johns Hopkins University School of Medicine Baltimore, MD 21287
                [2 ]Hugo W. Moser Research Institute, Kennedy Krieger Institute , Baltimore, MD, 21205
                [3 ]Department of Ophthalmology, Wilmer Eye Institute, Johns Hopkins University School of Medicine , Baltimore, MD 21287
                [4 ]Synaptic Physiology Section, National Institute of Neurological Disorders and Stroke, National Institutes of Health , Bethesda, MD 20892
                [5 ]Department of Biomedical Engineering, Johns Hopkins University School of Medicine , Baltimore, MD 21205
                [6 ]Translational Tissue Engineering Center, Johns Hopkins University School of Medicine , Baltimore, MD 21287
                [7 ]Department of Materials Science and Engineering, Whiting School of Engineering, and Institute for NanoBioTechnology, Johns Hopkins University , Baltimore, MD 21218
                [8 ]The Solomon H. Snyder Department of Neuroscience, Johns Hopkins University School of Medicine , Baltimore, MD, 21205
                [9 ]Institute of Genetic Medicine, Johns Hopkins University School of Medicine , Baltimore, MD 21287.
                Author notes
                Article
                srep16595
                10.1038/srep16595
                4643248
                26563826
                b109e590-f063-4367-913b-accf39b08481
                Copyright © 2015, Macmillan Publishers Limited

                This work is licensed under a Creative Commons Attribution 4.0 International License. The images or other third party material in this article are included in the article’s Creative Commons license, unless indicated otherwise in the credit line; if the material is not included under the Creative Commons license, users will need to obtain permission from the license holder to reproduce the material. To view a copy of this license, visit http://creativecommons.org/licenses/by/4.0/

                History
                : 27 May 2015
                : 13 October 2015
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