ArticleStem cell reports2014
Reversal of cellular phenotypes in neural cells derived from Huntington's disease monkey-induced pluripotent stem cells.
Article in Stem cell reports, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 18 papers.
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Who cites it
18 citing papers in PubMed, 26 citations in OpenAlex.
- Modeling genetic diseases in nonhuman primates through embryonic and germline modification: Considerations and challenges.Science translational medicine · 2022Review
- Divergent FUS phosphorylation in primate and mouse cells following double-strand DNA damage.Neurobiology of disease · 2020Article
- Recent Overview of the Use of iPSCs Huntington's Disease Modeling and Therapy.International journal of molecular sciences · 2020Review
- On the Right Track to Treat Movement Disorders: Promising Therapeutic Approaches for Parkinson's and Huntington's Disease.Frontiers in aging neuroscience · 2020Review
- Chromatin accessibility and transcription dynamics during in vitro astrocyte differentiation of Huntington's Disease Monkey pluripotent stem cells.Epigenetics & chromatin · 2019Article
- FGF2 and dual agonist of NCAM and FGF receptor 1, Enreptin, rescue neurite outgrowth loss in hippocampal neurons expressing mutated huntingtin proteins.Journal of neural transmission (Vienna, Austria : 1996) · 2019Article
- Generation of genetically engineered non-human primate models of brain function and neurological disorders.American journal of primatology · 2019Review
- Combination of stem cell and gene therapy ameliorates symptoms in Huntington's disease mice.NPJ Regenerative medicine · 2019Article
- Article
- Huntington Disease as a Neurodevelopmental Disorder and Early Signs of the Disease in Stem Cells.Molecular neurobiology · 2018Review
- Progress in developing transgenic monkey model for Huntington's disease.Journal of neural transmission (Vienna, Austria : 1996) · 2018Review
- FOXOs modulate proteasome activity in human-induced pluripotent stem cells of Huntington's disease and their derived neural cells.Human molecular genetics · 2017Article
- Induced Pluripotent HD Monkey Stem Cells Derived Neural Cells for Drug Discovery.SLAS discovery : advancing life sciences R & D · 2017Article
- iPSC-based drug screening for Huntington's disease.Brain research · 2016Review
- Novel targets for Huntington's disease: future prospects.Degenerative neurological and neuromuscular disease · 2016Review
- miR-196a Ameliorates Cytotoxicity and Cellular Phenotype in Transgenic Huntington's Disease Monkey Neural Cells.PloS one · 2016Article
- Germline transmission in transgenic Huntington's disease monkeys.Theriogenology · 2015Article
- Induced pluripotent stem cells and their use in cardiac and neural regenerative medicine.International journal of molecular sciences · 2015Review
Corrections and comments
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Authors and funding
10 authors at 2 institutions in 2 countries.
Funding
Abstract
Huntington's disease (HD) is a dominant neurodegenerative disorder caused by the expansion of glutamine residues in the N-terminal region of the huntingtin (HTT) protein. The disease results in progressive neuronal loss, leading to motor, cognitive, and psychiatric impairment. Here, we report the establishment of neural progenitor cell (NPC) lines derived from induced pluripotent stem cells (iPSCs) of transgenic HD monkeys. Upon differentiation to neurons, HD neural cells develop cellular features of HD, including the formation of nuclear inclusions and oligomeric mutant HTT (mHTT) aggregates, as well as increased apoptosis. These phenotypes are rescued by genetic suppression of HTT and pharmacological treatment, demonstrating the ability of our HD cell model to respond to therapeutic treatment. The development and reversal of HD-associated phenotypes in neural cells from HD monkeys provides a unique nonhuman primate (NHP) model for exploring HD pathogenesis and evaluating therapeutics that could be assessed further in HD monkeys.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.