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

Research output: Contribution to journalArticle

3 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 languageEnglish (US)
Pages (from-to)506-521.e7
JournalNeuron
Volume105
Issue number3
DOIs
StatePublished - Feb 5 2020

Keywords

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

ASJC Scopus subject areas

  • Neuroscience(all)

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    Yoon, S., Parnell, E., Kasherman, M., Forrest, M. P., Myczek, K., Premarathne, S., Sanchez Vega, M. C., Piper, M., Burne, T. H. J., Jolly, L. A., Wood, S. A., & Penzes, P. (2020). Usp9X Controls Ankyrin-Repeat Domain Protein Homeostasis during Dendritic Spine Development. Neuron, 105(3), 506-521.e7. https://doi.org/10.1016/j.neuron.2019.11.003