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Modeling Retinal Dystrophies Using Patient-Derived Induced Pluripotent Stem Cells

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Modeling Retinal Dystrophies Using Patient-Derived Induced Pluripotent Stem Cells

Auteurs : Karl J. Wahlin ; Julien Maruotti ; Donald J. Zack

Source :

Advances in experimental medicine and biology [ 0065-2598 ] ; 2014.

RBID : PMC:4117190

Abstract

Retinal degenerative disease involving photoreceptor (PR) cell loss results in permanent vision loss and often blindness. Generation of induced pluripotent stem cell (iPSC)-derived retinal cells and tissues from individuals with retinal dystrophies is a relatively new and promising method for studying retinal degeneration mechanisms in vitro. Recent advancements in strategies to differentiate human iPSCs (hiPSCs) into 3D retinal eyecups with a strong resemblance to the mature retina raise the possibility that this system could offer a reliable model for translational drug studies. However, despite the potential benefits, there are challenges that remain to be overcome before stem-cell-derived retinal eyecups can be routinely used to model human retinal diseases. This chapter will discuss both the potential of these 3D eyecup approaches and the nature of some of the challenges that remain.

Url:

http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4117190

DOI: 10.1007/978-1-4614-3209-8_20
PubMed: 24664693
PubMed Central: 4117190

Affiliations:

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PMC:4117190

Le document en format XML

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<author><name sortKey="Maruotti, Julien" sort="Maruotti, Julien" uniqKey="Maruotti J" first="Julien" last="Maruotti">Julien Maruotti</name>
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<front><div type="abstract" xml:lang="en"><p id="P1"><italic>Retinal</italic>
 degenerative disease involving photoreceptor (PR) cell loss results in permanent vision loss and often blindness. Generation of induced pluripotent stem cell (iPSC)-derived retinal cells and tissues from individuals with retinal dystrophies is a relatively new and promising method for studying retinal degeneration mechanisms in vitro. Recent advancements in strategies to differentiate human iPSCs (hiPSCs) into 3D retinal eyecups with a strong resemblance to the mature retina raise the possibility that this system could offer a reliable model for translational drug studies. However, despite the potential benefits, there are challenges that remain to be overcome before stem-cell-derived retinal eyecups can be routinely used to model human retinal diseases. This chapter will discuss both the potential of these 3D eyecup approaches and the nature of some of the challenges that remain.</p>
</div>
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  <front><journal-meta><journal-id journal-id-type="nlm-journal-id">0121103</journal-id>
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<journal-id journal-id-type="nlm-ta">Adv Exp Med Biol</journal-id>
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<contrib-group><contrib contrib-type="author" corresp="yes"><name><surname>Wahlin</surname>
<given-names>Karl J.</given-names>
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<aff id="A1">Department of Ophthalmology, The Johns Hopkins University School of Medicine, Smith Building Rm 3029, 400 N Broadway, Baltimore, MD, USA</aff>
<email>kwahlin1@jhmi.edu</email>
</contrib>
<contrib contrib-type="author"><name><surname>Maruotti</surname>
<given-names>Julien</given-names>
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<aff id="A2">Department of Ophthalmology, The Johns Hopkins University School of Medicine, Smith Building Rm 3029, 400 N Broadway, Baltimore, MD, USA</aff>
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<contrib contrib-type="author"><name><surname>Zack</surname>
<given-names>Donald J.</given-names>
</name>
<aff id="A3">Department of Ophthalmology, The Johns Hopkins University School of Medicine, Smith Building Rm 3029, 400 N Broadway, Baltimore, MD, USA. Department of Neuroscience, The Johns Hopkins University School of Medicine, Baltimore, MD, USA. Department of Molecular Biology and Genetics, The Johns Hopkins University School of Medicine, Baltimore, MD, USA. Institute of Genetic Medicine, The Johns Hopkins University School of Medicine, Baltimore, MD, USA. Institut de la Vision, Paris 75012, Paris, France</aff>
<email>dzack@jhmi.edu</email>
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<pub-date pub-type="nihms-submitted"><day>4</day>
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<year>2014</year>
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<pub-date pub-type="pmc-release"><day>31</day>
<month>7</month>
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<volume>801</volume>
<fpage>157</fpage>
<lpage>164</lpage>
<pmc-comment>elocation-id from pubmed: 10.1007/978-1-4614-3209-8_20</pmc-comment>
      <permissions><copyright-statement>© Springer Science+Business Media, LLC 2014</copyright-statement>
<copyright-year>2014</copyright-year>
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<abstract><p id="P1"><italic>Retinal</italic>
 degenerative disease involving photoreceptor (PR) cell loss results in permanent vision loss and often blindness. Generation of induced pluripotent stem cell (iPSC)-derived retinal cells and tissues from individuals with retinal dystrophies is a relatively new and promising method for studying retinal degeneration mechanisms in vitro. Recent advancements in strategies to differentiate human iPSCs (hiPSCs) into 3D retinal eyecups with a strong resemblance to the mature retina raise the possibility that this system could offer a reliable model for translational drug studies. However, despite the potential benefits, there are challenges that remain to be overcome before stem-cell-derived retinal eyecups can be routinely used to model human retinal diseases. This chapter will discuss both the potential of these 3D eyecup approaches and the nature of some of the challenges that remain.</p>
</abstract>
<kwd-group><kwd>Retinal degeneration</kwd>
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<kwd>Dystrophy</kwd>
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<kwd>hiPSC</kwd>
<kwd>Pluripotent</kwd>
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<name sortKey="Zack, Donald J" sort="Zack, Donald J" uniqKey="Zack D" first="Donald J." last="Zack">Donald J. Zack</name>
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Modeling Retinal Dystrophies Using Patient-Derived Induced Pluripotent Stem Cells

Modeling Retinal Dystrophies Using Patient-Derived Induced Pluripotent Stem Cells

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Abstract

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