Huntingtin suppression restores cognitive function in a mouse model of Huntington’s disease
Open accessAbstract
Mutant huntingtin suppression with antisense oligonucleotides reverses cognitive impairments in a mouse model of Huntington’s disease.
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ASO delivery to deep brain structures is limited, with no significant HTT silencing observed in NHP caudate after multiple injections.
“However, ASO delivery to deep brain structures is limited4, with no significant HTT silencing observed in NHP caudate after multiple injections42.”
From A divalent siRNA chemical scaffold for potent and sustained modulation of gene expression throughout the central nervous system (2019) · cites this paper as [42]
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References 46 · Cited by 126
- Authors
- Southwell, A. L., Kordasiewicz, H. B., Langbehn, D., Skotte, N. H., Parsons, M. P., Villanueva, E. B., Caron, N. S., Østergaard, M. E., Anderson, L. M., Xie, Y., Cengio, L. D., Findlay-Black, H., Doty, C. N., Fitsimmons, B., Swayze, E. E., Seth, P. P., Raymond, L. A., Frank Bennett, C., & Hayden, M. R.
- Affiliations
- University of British Columbia (CA)
- Ionis Pharmaceuticals (United States) (US)
- University of Iowa (US)
- Centre for Molecular Medicine and Therapeutics, Child and Family Research Institute, University of British Columbia, Vancouver, British Columbia V5Z 4H4, Canada.
- Ionis Pharmaceuticals, Carlsbad, CA 92008, USA.
- Corresponding author
- Hayden, Michael R.
- ORCID
- Venue
- Science Translational Medicine
- Published
- 2018-10-03
- Volume
- 10
- Issue
- 461
- ISSN
- 1946-6234 (ISSN-L)
- 1946-6242
- OpenAlex
- W2895512177
- Cited by
- 126
- References
- 46
- Citations by year
- 2026: 6 · 2025: 11 · 2024: 21 · 2023: 12 · 2022: 17 · 2021: 13 · 2020: 22 · 2019: 20 · 2018: 4
- FWCI
- 6.74
- Type
- journal article
- Language
- en
- Open access
- Locations
- Science Translational Medicine (publishedVersion)
- PubMed (publishedVersion)
- Journal of International Crisis and Risk Communication Research (submittedVersion)
- Funders
- Huntington Society of Canada
- Canadian Institutes of Health Research (MOP-84438)
- IONIS Pharmaceuticals
- Topics
- Life Sciences › Neuroscience › Cellular and Molecular Neuroscience › Genetic Neurodegenerative Diseases
- Health Sciences › Medicine › Neurology › Amyotrophic Lateral Sclerosis Research
- Health Sciences › Medicine › Physiology › Lysosomal Storage Disorders Research
- Keywords
- Huntington's disease, mutant huntingtin, antisense oligonucleotides, gene silencing, cognitive impairment, mouse model
- MeSH terms
- Huntingtin Protein, Animals, Anxiety, Atrophy, Behavior, Animal, Brain, Cognition, Disease Models, Animal, Female, Humans, Huntington Disease, Limbic System, Male, Primates, Oligonucleotides, Antisense, Mutant Proteins, Dopamine and cAMP-Regulated Phosphoprotein 32
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