ArticleStem cells and development2013
Pathogenic cellular phenotypes are germline transmissible in a transgenic primate model of Huntington's disease.
Article in Stem cells and development, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 24 papers.
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Who cites it
24 citing papers in PubMed, 31 citations in OpenAlex.
- Article
- Genetically modified non-human primate models for research on neurodegenerative diseases.Zoological research · 2024Review
- A controlled ovarian stimulation procedure suitable for cynomolgus macaques.Experimental animals · 2022Article
- Suppression of trinucleotide repeat expansion in spermatogenic cells in Huntington's disease.Journal of assisted reproduction and genetics · 2022Article
- Modeling genetic diseases in nonhuman primates through embryonic and germline modification: Considerations and challenges.Science translational medicine · 2022Review
- CAG repeat instability in embryonic stem cells and derivative spermatogenic cells of transgenic Huntington's disease monkey.Journal of assisted reproduction and genetics · 2021Article
- Local transgene expression and whole-body transgenesis to model brain diseases in nonhuman primate.Animal models and experimental medicine · 2019Review
- Generation of genetically engineered non-human primate models of brain function and neurological disorders.American journal of primatology · 2019Review
- 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
- Close encounters: Moving along bumps, breaks, and bubbles on expanded trinucleotide tracts.DNA repair · 2017Review
- Induced Pluripotent HD Monkey Stem Cells Derived Neural Cells for Drug Discovery.SLAS discovery : advancing life sciences R & D · 2017Article
- Increased irritability, anxiety, and immune reactivity in transgenic Huntington's disease monkeys.Brain, behavior, and immunity · 2016Article
- Generation of transgenic marmosets expressing genetically encoded calcium indicators.Scientific reports · 2016Article
- Cryotolerance of Sperm from Transgenic Rhesus Macaques (Macaca mulatta).Journal of the American Association for Laboratory Animal Science : JAALAS · 2016Article
- 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
- Article
- Article
- Reversal of cellular phenotypes in neural cells derived from Huntington's disease monkey-induced pluripotent stem cells.Stem cell reports · 2014Article
Corrections and comments
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Authors and funding
6 authors at 3 institutions in 2 countries.
Funding
Abstract
A transgenic primate model for Huntington's Disease (HD) first reported by our group that (HD monkeys) carry the mutant Huntingtin (HTT) gene with expanded polyglutamine (CAG) repeats and, develop chorea, dystonia, and other involuntary motor deficiencies similar to HD [ 1 ]. More recently, we have found that longitudinal magnetic resonance imaging of the HD monkey brain revealed significant atrophy in regions associated with cognitive deficits symptomatic in HD patients, providing the first animal model which replicates clinical phenotypes of diagnosed humans. Here we report germline transmission of the pathogenic mutant HTT in HD monkey by the production of embryos and subsequent derivation of HD monkey embryonic stem cells (rHD-ESCs) using HD monkey sperm. rHD-ESCs inherit mutant HTT and green fluorescent protein (GFP) genes through the gametes of HD monkey. rHD-ESCs express mutant HTT and form intranuclear inclusion, a classical cellular feature of HD. Notably, mosaicism of the pathogenic polyQ region in the sperm as well as derived ESCs were also observed, consistent with intraindividual and intergenerational reports of mosaic CAG repeats [ 2 , 3 ]and CAG expansion in HD patients [ 4-7 ]. The confirmation of transgene inheritability and development of pathogenic HD phenotype in derived rHD-ESCs reported in this study is a milestone in the pursuit of a transgenic primate model with inherited mutant HTT for development of novel disease biomarkers and therapeutics.
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