Antisense oligonucleotide-mediated ataxin-1 reduction prolongs survival in SCA1 mice and reveals disease-associated transcriptome profiles.

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作者： Jillian,Friedrich ^[1] ; Holly B,Kordasiewicz ^[2] ; Brennon,O'Callaghan ^[3] ; Hillary P,Handler ^[1] ; Carmen,Wagener ^[2] ; Lisa,Duvick ^[1] ; Eric E,Swayze ^[4] ; Orion,Rainwater ^[1] ; Bente,Hofstra ^[2] ; Michael,Benneyworth ^[1] ; Tessa,Nichols-Meade ^[2] ; Praseuth,Yang ^[3] ; Zhao,Chen ^[1] ; Judit Perez,Ortiz ^[2] ; H Brent,Clark ^[1] ; Gülin,Öz ^[2] ; Sarah,Larson ^[5] ; Huda Y,Zoghbi ^[5] ; Christine,Henzler ^[1] ; Harry T,Orr ^[2]

作者单位： Institute for Translational Neuroscience and. ^[1] Department of Laboratory Medicine and Pathology, University of Minnesota, Minneapolis, Minnesota, USA. ^[2] Ionis Pharmaceuticals, Carlsbad, California, USA. ^[3] Graduate Program in Neuroscience. ^[4] Department of Neuroscience, and. ^[5] Center for Magnetic Resonance Research, Department of Radiology, University of Minnesota, Minneapolis, Minnesota, USA. ^[6] Jan and Dan Duncan Neurological Research Institute at Texas Children's Hospital and Department of Molecular and Human Genetics, Howard Hughes Medical Institute, Baylor College of Medicine, Houston, Texas, USA. ^[7] Research Informatics Support Systems Bioinformatics, Minnesota Supercomputing Institute, University of Minnesota, Minneapolis, Minnesota, USA. ^[8]

关键词 Drug therapy Neurodegeneration Neuroscience

主题词动物(Animals);女(雌)性(Female);磁共振波谱学(Magnetic Resonance Spectroscopy);男(雄)性(Male);小鼠(Mice);神经组织蛋白质类(Nerve Tissue Proteins);神经变性疾病(Neurodegenerative Diseases);寡核苷酸类, 反义(Oligonucleotides, Antisense);表型(Phenotype);序列分析, RNA(Sequence Analysis, RNA);脊髓小脑共济失调(Spinocerebellar Ataxias);存活率分析(Survival Analysis)

DOI 10.1172/jci.insight.123193

PMID 30385727

发布时间 2023-04-13

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Antisense oligonucleotide-mediated ataxin-1 reduction prolongs survival in SCA1 mice and reveals disease-associated transcriptome profiles.

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Antisense oligonucleotide-mediated ataxin-1 reduction prolongs survival in SCA1 mice and reveals disease-associated transcriptome profiles.

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