Molecular Therapies for Inherited Retinal Diseases

Following the implementation of next-generation sequencing technologies (e.g., exome and genome sequencing) in molecular diagnostics, the majority of genetic defects underlying inherited retinal disease (IRD) can readily be identified. In parallel, opportunities to counteract the molecular consequen...

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Publicado em: MDPI - Multidisciplinary Digital Publishing Institute 2021
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description Following the implementation of next-generation sequencing technologies (e.g., exome and genome sequencing) in molecular diagnostics, the majority of genetic defects underlying inherited retinal disease (IRD) can readily be identified. In parallel, opportunities to counteract the molecular consequences of these defects are rapidly emerging, providing hope for personalized medicine. ‘Classical’ gene augmentation therapy has been under study for several genetic subtypes of IRD and can be considered a safe and sometimes effective therapeutic strategy. The recent market approval of the first retinal gene augmentation therapy product (LuxturnaTM, for individuals with bi-allelic RPE65 mutations) by the FDA has not only demonstrated the potential of this specific approach, but also opened avenues for the development of other strategies. However, every gene—or even every mutation—may need a tailor-made therapeutic approach, in order to obtain the most efficacious strategy with minimal risks associated. In addition to gene augmentation therapy, other subtypes of molecular therapy are currently being designed and/or implemented, including splice modulation, DNA or RNA editing, optogenetics and pharmacological modulation. In addition, the development of proper delivery vectors has gained strong attention, and should not be overlooked when designing and testing a novel therapeutic approach. In this Special Issue, we aim to describe the current state of the art of molecular therapeutics for IRD, and discuss existing and novel therapeutic strategies, from idea to implementation, and from bench to bedside.
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spelling doab-20.500.12854ir-691742024-03-28T03:30:37Z Molecular Therapies for Inherited Retinal Diseases Collin, Rob W.J. Garanto, Alejandro induced pluripotent stem cell (iPSC) clustered regularly interspaced short palindromic repeats (CRISPR) homology-directed repair (HDR) Enhanced S-Cone Syndrome (ESCS) NR2E3 AAV retina gene therapy dual AAV gold nanoparticles DNA-wrapped gold nanoparticles ARPE-19 cells retinal pigment epithelium clathrin-coated vesicles endosomal trafficking retinitis pigmentosa autosomal dominant G56R putative dominant negative effect gapmer antisense oligonucleotides allele-specific knockdown Leber congenital amaurosis and allied retinal ciliopathies CEP290 Flanders founder c.4723A &gt T nonsense mutation Cilia elongation spontaneous nonsense correction AON-mediated exon skipping microRNA photoreceptors rods cones bipolar cells Müller glia retinal inherited disorders retinal degeneration antisense oligonucleotides Stargardt disease inherited retinal diseases splicing modulation RNA therapy ABCA4 iPSC-derived photoreceptor precursor cells cyclic GMP apoptosis necrosis drug delivery systems translational medicine Usher syndrome Leber congenital amaurosis RPE65 nonprofit patient registry translational protein trafficking protein folding protein degradation chaperones chaperonins heat shock response unfolded protein response autophagy therapy IRD DNA therapies RNA therapies compound therapies clinical trials Retinitis Pigmentosa GTPase Regulator adeno-associated viral Retinitis Pigmentosa (RP) choroideremia REP1 inherited retinal disease treatment apical polarity crumbs complex fetal retina PAR complex retinal organoids retinogenesis gene augmentation adeno-associated virus (AAV) n/a thema EDItEUR::G Reference, Information and Interdisciplinary subjects::GP Research and information: general thema EDItEUR::P Mathematics and Science::PS Biology, life sciences Following the implementation of next-generation sequencing technologies (e.g., exome and genome sequencing) in molecular diagnostics, the majority of genetic defects underlying inherited retinal disease (IRD) can readily be identified. In parallel, opportunities to counteract the molecular consequences of these defects are rapidly emerging, providing hope for personalized medicine. ‘Classical’ gene augmentation therapy has been under study for several genetic subtypes of IRD and can be considered a safe and sometimes effective therapeutic strategy. The recent market approval of the first retinal gene augmentation therapy product (LuxturnaTM, for individuals with bi-allelic RPE65 mutations) by the FDA has not only demonstrated the potential of this specific approach, but also opened avenues for the development of other strategies. However, every gene—or even every mutation—may need a tailor-made therapeutic approach, in order to obtain the most efficacious strategy with minimal risks associated. In addition to gene augmentation therapy, other subtypes of molecular therapy are currently being designed and/or implemented, including splice modulation, DNA or RNA editing, optogenetics and pharmacological modulation. In addition, the development of proper delivery vectors has gained strong attention, and should not be overlooked when designing and testing a novel therapeutic approach. In this Special Issue, we aim to describe the current state of the art of molecular therapeutics for IRD, and discuss existing and novel therapeutic strategies, from idea to implementation, and from bench to bedside. 2021-05-01T15:43:01Z 2021-05-01T15:43:01Z 2020 book ONIX_20210501_9783039431762_920 9783039431762 9783039431779 https://directory.doabooks.org/handle/20.500.12854/69174 eng application/octet-stream Attribution 4.0 International https://mdpi.com/books/pdfview/book/2946 https://mdpi.com/books/pdfview/book/2946 MDPI - Multidisciplinary Digital Publishing Institute 10.3390/books978-3-03943-177-9 10.3390/books978-3-03943-177-9 46cabcaa-dd94-4bfe-87b4-55023c1b36d0 9783039431762 9783039431779 262 Basel, Switzerland open access
spellingShingle induced pluripotent stem cell (iPSC)
clustered regularly interspaced short palindromic repeats (CRISPR)
homology-directed repair (HDR)
Enhanced S-Cone Syndrome (ESCS)
NR2E3
AAV
retina
gene therapy
dual AAV
gold nanoparticles
DNA-wrapped gold nanoparticles
ARPE-19 cells
retinal pigment epithelium
clathrin-coated vesicles
endosomal trafficking
retinitis pigmentosa
autosomal dominant
G56R
putative dominant negative effect
gapmer antisense oligonucleotides
allele-specific knockdown
Leber congenital amaurosis and allied retinal ciliopathies
CEP290
Flanders founder c.4723A &gt
T nonsense mutation
Cilia elongation
spontaneous nonsense correction
AON-mediated exon skipping
microRNA
photoreceptors
rods
cones
bipolar cells
Müller glia
retinal inherited disorders
retinal degeneration
antisense oligonucleotides
Stargardt disease
inherited retinal diseases
splicing modulation
RNA therapy
ABCA4
iPSC-derived photoreceptor precursor cells
cyclic GMP
apoptosis
necrosis
drug delivery systems
translational medicine
Usher syndrome
Leber congenital amaurosis
RPE65
nonprofit
patient registry
translational
protein trafficking
protein folding
protein degradation
chaperones
chaperonins
heat shock response
unfolded protein response
autophagy
therapy
IRD
DNA therapies
RNA therapies
compound therapies
clinical trials
Retinitis Pigmentosa GTPase Regulator
adeno-associated viral
Retinitis Pigmentosa (RP)
choroideremia
REP1
inherited retinal disease
treatment
apical polarity
crumbs complex
fetal retina
PAR complex
retinal organoids
retinogenesis
gene augmentation
adeno-associated virus (AAV)
n/a
thema EDItEUR::G Reference, Information and Interdisciplinary subjects::GP Research and information: general
thema EDItEUR::P Mathematics and Science::PS Biology, life sciences
Molecular Therapies for Inherited Retinal Diseases
title Molecular Therapies for Inherited Retinal Diseases
title_full Molecular Therapies for Inherited Retinal Diseases
title_fullStr Molecular Therapies for Inherited Retinal Diseases
title_full_unstemmed Molecular Therapies for Inherited Retinal Diseases
title_short Molecular Therapies for Inherited Retinal Diseases
title_sort molecular therapies for inherited retinal diseases
topic induced pluripotent stem cell (iPSC)
clustered regularly interspaced short palindromic repeats (CRISPR)
homology-directed repair (HDR)
Enhanced S-Cone Syndrome (ESCS)
NR2E3
AAV
retina
gene therapy
dual AAV
gold nanoparticles
DNA-wrapped gold nanoparticles
ARPE-19 cells
retinal pigment epithelium
clathrin-coated vesicles
endosomal trafficking
retinitis pigmentosa
autosomal dominant
G56R
putative dominant negative effect
gapmer antisense oligonucleotides
allele-specific knockdown
Leber congenital amaurosis and allied retinal ciliopathies
CEP290
Flanders founder c.4723A &gt
T nonsense mutation
Cilia elongation
spontaneous nonsense correction
AON-mediated exon skipping
microRNA
photoreceptors
rods
cones
bipolar cells
Müller glia
retinal inherited disorders
retinal degeneration
antisense oligonucleotides
Stargardt disease
inherited retinal diseases
splicing modulation
RNA therapy
ABCA4
iPSC-derived photoreceptor precursor cells
cyclic GMP
apoptosis
necrosis
drug delivery systems
translational medicine
Usher syndrome
Leber congenital amaurosis
RPE65
nonprofit
patient registry
translational
protein trafficking
protein folding
protein degradation
chaperones
chaperonins
heat shock response
unfolded protein response
autophagy
therapy
IRD
DNA therapies
RNA therapies
compound therapies
clinical trials
Retinitis Pigmentosa GTPase Regulator
adeno-associated viral
Retinitis Pigmentosa (RP)
choroideremia
REP1
inherited retinal disease
treatment
apical polarity
crumbs complex
fetal retina
PAR complex
retinal organoids
retinogenesis
gene augmentation
adeno-associated virus (AAV)
n/a
thema EDItEUR::G Reference, Information and Interdisciplinary subjects::GP Research and information: general
thema EDItEUR::P Mathematics and Science::PS Biology, life sciences
topic_facet induced pluripotent stem cell (iPSC)
clustered regularly interspaced short palindromic repeats (CRISPR)
homology-directed repair (HDR)
Enhanced S-Cone Syndrome (ESCS)
NR2E3
AAV
retina
gene therapy
dual AAV
gold nanoparticles
DNA-wrapped gold nanoparticles
ARPE-19 cells
retinal pigment epithelium
clathrin-coated vesicles
endosomal trafficking
retinitis pigmentosa
autosomal dominant
G56R
putative dominant negative effect
gapmer antisense oligonucleotides
allele-specific knockdown
Leber congenital amaurosis and allied retinal ciliopathies
CEP290
Flanders founder c.4723A &gt
T nonsense mutation
Cilia elongation
spontaneous nonsense correction
AON-mediated exon skipping
microRNA
photoreceptors
rods
cones
bipolar cells
Müller glia
retinal inherited disorders
retinal degeneration
antisense oligonucleotides
Stargardt disease
inherited retinal diseases
splicing modulation
RNA therapy
ABCA4
iPSC-derived photoreceptor precursor cells
cyclic GMP
apoptosis
necrosis
drug delivery systems
translational medicine
Usher syndrome
Leber congenital amaurosis
RPE65
nonprofit
patient registry
translational
protein trafficking
protein folding
protein degradation
chaperones
chaperonins
heat shock response
unfolded protein response
autophagy
therapy
IRD
DNA therapies
RNA therapies
compound therapies
clinical trials
Retinitis Pigmentosa GTPase Regulator
adeno-associated viral
Retinitis Pigmentosa (RP)
choroideremia
REP1
inherited retinal disease
treatment
apical polarity
crumbs complex
fetal retina
PAR complex
retinal organoids
retinogenesis
gene augmentation
adeno-associated virus (AAV)
n/a
thema EDItEUR::G Reference, Information and Interdisciplinary subjects::GP Research and information: general
thema EDItEUR::P Mathematics and Science::PS Biology, life sciences
url ONIX_20210501_9783039431762_920