Huntington's Disease Pathogenesis Is Modified In Vivo by Alfy/Wdfy3 and Selective Macroautophagy

Leora M. Fox; Kiryung Kim; Christopher W. Johnson; Shawei Chen; Katherine R. Croce; Matheus B. Victor; Evelien Eenjes; Joan R. Bosco; Lisa K. Randolph; Ioannis Dragatsis; Joanna M. Dragich; Andrew S. Yoo; Ai Yamamoto

doi:10.1016/j.neuron.2019.12.003

Huntington's Disease Pathogenesis Is Modified In Vivo by Alfy/Wdfy3 and Selective Macroautophagy

Leora M. Fox
, Kiryung Kim
, Christopher W. Johnson
, Shawei Chen
, Katherine R. Croce
, Matheus B. Victor
, Evelien Eenjes
, Joan R. Bosco
, Lisa K. Randolph
, Ioannis Dragatsis
, Joanna M. Dragich
, Andrew S. Yoo
, Ai Yamamoto

Research output: Contribution to journal › Article › peer-review

63 Scopus citations

Abstract

Fox et al. demonstrate in a Huntington's disease (HD) mouse model and HD-patient-derived neurons that the autophagy adaptor protein Alfy/WDFY3 is required for removal of aggregated protein and that depletion of Alfy hastens the pathogenic onset of HD.

Original language	English
Pages (from-to)	813-821.e6
Journal	Neuron
Volume	105
Issue number	5
DOIs	https://doi.org/10.1016/j.neuron.2019.12.003
State	Published - 4 Mar 2020
Externally published	Yes

Keywords

Alfy
Huntington's disease
Wdfy3
autophagy
direct conversion
mice
neurodegeneration
patient fibroblasts
proteinopathy
selective autophagy

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10.1016/j.neuron.2019.12.003

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title = "Huntington's Disease Pathogenesis Is Modified In Vivo by Alfy/Wdfy3 and Selective Macroautophagy",

abstract = "Fox et al. demonstrate in a Huntington's disease (HD) mouse model and HD-patient-derived neurons that the autophagy adaptor protein Alfy/WDFY3 is required for removal of aggregated protein and that depletion of Alfy hastens the pathogenic onset of HD.",

keywords = "Alfy, Huntington's disease, Wdfy3, autophagy, direct conversion, mice, neurodegeneration, patient fibroblasts, proteinopathy, selective autophagy",

author = "Fox, \{Leora M.\} and Kiryung Kim and Johnson, \{Christopher W.\} and Shawei Chen and Croce, \{Katherine R.\} and Victor, \{Matheus B.\} and Evelien Eenjes and Bosco, \{Joan R.\} and Randolph, \{Lisa K.\} and Ioannis Dragatsis and Dragich, \{Joanna M.\} and Yoo, \{Andrew S.\} and Ai Yamamoto",

note = "Publisher Copyright: {\textcopyright} 2019 Elsevier Inc.",

year = "2020",

month = mar,

day = "4",

doi = "10.1016/j.neuron.2019.12.003",

language = "English",

volume = "105",

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journal = "Neuron",

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T1 - Huntington's Disease Pathogenesis Is Modified In Vivo by Alfy/Wdfy3 and Selective Macroautophagy

AU - Fox, Leora M.

AU - Kim, Kiryung

AU - Johnson, Christopher W.

AU - Chen, Shawei

AU - Croce, Katherine R.

AU - Victor, Matheus B.

AU - Eenjes, Evelien

AU - Bosco, Joan R.

AU - Randolph, Lisa K.

AU - Dragatsis, Ioannis

AU - Dragich, Joanna M.

AU - Yoo, Andrew S.

AU - Yamamoto, Ai

PY - 2020/3/4

Y1 - 2020/3/4

N2 - Fox et al. demonstrate in a Huntington's disease (HD) mouse model and HD-patient-derived neurons that the autophagy adaptor protein Alfy/WDFY3 is required for removal of aggregated protein and that depletion of Alfy hastens the pathogenic onset of HD.

AB - Fox et al. demonstrate in a Huntington's disease (HD) mouse model and HD-patient-derived neurons that the autophagy adaptor protein Alfy/WDFY3 is required for removal of aggregated protein and that depletion of Alfy hastens the pathogenic onset of HD.

KW - Alfy

KW - Huntington's disease

KW - Wdfy3

KW - autophagy

KW - direct conversion

KW - mice

KW - neurodegeneration

KW - patient fibroblasts

KW - proteinopathy

KW - selective autophagy

UR - https://www.scopus.com/pages/publications/85080068826

U2 - 10.1016/j.neuron.2019.12.003

DO - 10.1016/j.neuron.2019.12.003

M3 - Article

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AN - SCOPUS:85080068826

SN - 0896-6273

VL - 105

SP - 813-821.e6

JO - Neuron

JF - Neuron

IS - 5

ER -

Huntington's Disease Pathogenesis Is Modified In Vivo by Alfy/Wdfy3 and Selective Macroautophagy

Abstract

Keywords

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