Usp9X Controls Ankyrin-Repeat Domain Protein Homeostasis during Dendritic Spine Development

Sehyoun Yoon; Euan Parnell; Maria Kasherman; Marc P. Forrest; Kristoffer Myczek; Susitha Premarathne; Michelle C. Sanchez Vega; Michael Piper; Thomas H.J. Burne; Lachlan A. Jolly; Stephen A. Wood; Peter Penzes

doi:10.1016/j.neuron.2019.11.003

Usp9X Controls Ankyrin-Repeat Domain Protein Homeostasis during Dendritic Spine Development

Sehyoun Yoon, Euan Parnell, Maria Kasherman, Marc P. Forrest, Kristoffer Myczek, Susitha Premarathne, Michelle C. Sanchez Vega, Michael Piper, Thomas H.J. Burne, Lachlan A. Jolly, Stephen A. Wood, Peter Penzes^*

^*Corresponding author for this work

Neuroscience

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

34 Scopus citations

Abstract

Yoon et al. show that deubiquitination of proteins containing ankyrin-repeat domains is essential for the correct developmental trajectory of cortical synapses, with disruption of the deubiquitinase Usp9X resulting in deficient synaptic structural plasticity as well as behavioral and clinical abnormalities.

Original language	English (US)
Pages (from-to)	506-521.e7
Journal	Neuron
Volume	105
Issue number	3
DOIs	https://doi.org/10.1016/j.neuron.2019.11.003
State	Published - Feb 5 2020

Keywords

ANK
SHANK
ankyrin-G
deubiquitinase
intellectual disability
proximity ligation assay
structured illumination microscopy

ASJC Scopus subject areas

General Neuroscience

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10.1016/j.neuron.2019.11.003

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title = "Usp9X Controls Ankyrin-Repeat Domain Protein Homeostasis during Dendritic Spine Development",

abstract = "Yoon et al. show that deubiquitination of proteins containing ankyrin-repeat domains is essential for the correct developmental trajectory of cortical synapses, with disruption of the deubiquitinase Usp9X resulting in deficient synaptic structural plasticity as well as behavioral and clinical abnormalities.",

keywords = "ANK, SHANK, ankyrin-G, deubiquitinase, intellectual disability, proximity ligation assay, structured illumination microscopy",

author = "Sehyoun Yoon and Euan Parnell and Maria Kasherman and Forrest, {Marc P.} and Kristoffer Myczek and Susitha Premarathne and {Sanchez Vega}, {Michelle C.} and Michael Piper and Burne, {Thomas H.J.} and Jolly, {Lachlan A.} and Wood, {Stephen A.} and Peter Penzes",

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

year = "2020",

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language = "English (US)",

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T1 - Usp9X Controls Ankyrin-Repeat Domain Protein Homeostasis during Dendritic Spine Development

AU - Yoon, Sehyoun

AU - Parnell, Euan

AU - Kasherman, Maria

AU - Forrest, Marc P.

AU - Myczek, Kristoffer

AU - Premarathne, Susitha

AU - Sanchez Vega, Michelle C.

AU - Piper, Michael

AU - Burne, Thomas H.J.

AU - Jolly, Lachlan A.

AU - Wood, Stephen A.

AU - Penzes, Peter

PY - 2020/2/5

Y1 - 2020/2/5

N2 - Yoon et al. show that deubiquitination of proteins containing ankyrin-repeat domains is essential for the correct developmental trajectory of cortical synapses, with disruption of the deubiquitinase Usp9X resulting in deficient synaptic structural plasticity as well as behavioral and clinical abnormalities.

AB - Yoon et al. show that deubiquitination of proteins containing ankyrin-repeat domains is essential for the correct developmental trajectory of cortical synapses, with disruption of the deubiquitinase Usp9X resulting in deficient synaptic structural plasticity as well as behavioral and clinical abnormalities.

KW - ANK

KW - SHANK

KW - ankyrin-G

KW - deubiquitinase

KW - intellectual disability

KW - proximity ligation assay

KW - structured illumination microscopy

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VL - 105

SP - 506-521.e7

JO - Neuron

JF - Neuron

IS - 3

ER -

Usp9X Controls Ankyrin-Repeat Domain Protein Homeostasis during Dendritic Spine Development

Abstract

Keywords

ASJC Scopus subject areas

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