@article{172b62737de148a0b2498203b46fc6bc,
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 = "Funding Information: This work was supported by R01MH107182 to P.P. and SFARI Explorer Grant 527556 to M.P., S.W., and L.J. We thank NU Nikon Cell Imaging Facility for use of the N-SIM. Protein expression and purification was performed in the Recombinant Protein Production Core Facility at Northwestern University. Special thanks go to Sergii Pshenychnyi for assistance throughout. In vitro analyses were performed in the Analytical BioNanoTechnology Core Facility of the Simpson Querrey Institute at Northwestern University. Northwestern University provided funding to develop this facility and ongoing support is being received from the Soft and Hybrid Nanotechnology Experimental (SHyNE) Resource ( NSF NNCI-1542205 ). Molecular modeling was performed by the Medicinal and Synthetic Chemistry Core at Northwestern University. Funding Information: This work was supported by R01MH107182 to P.P. and SFARI Explorer Grant 527556 to M.P. S.W. and L.J. We thank NU Nikon Cell Imaging Facility for use of the N-SIM. Protein expression and purification was performed in the Recombinant Protein Production Core Facility at Northwestern University. Special thanks go to Sergii Pshenychnyi for assistance throughout. In vitro analyses were performed in the Analytical BioNanoTechnology Core Facility of the Simpson Querrey Institute at Northwestern University. Northwestern University provided funding to develop this facility and ongoing support is being received from the Soft and Hybrid Nanotechnology Experimental (SHyNE) Resource (NSF NNCI-1542205). Molecular modeling was performed by the Medicinal and Synthetic Chemistry Core at Northwestern University. S.Y. initiated the project and performed all experiments, data analysis, and manuscript preparation unless otherwise stated. E.P. performed and analyzed homology modeling and DUB assay. M.P.F. analyzed the bioinformatics data. K.M. designed the yeast-2-hybrid experiments. S.P. and S.A.W. prepared the fixed brains from transgenic mice. M.P. and T.H.J.B. supervised and analyzed all behavioral tests in transgenic mice; M.K. performed the open-field, elevated plus maze, light/dark box tests, and metabolic measurements, and M.S. performed the forced-swim test. L.A.J. identified missense variants and analyzed neurological features. P.P. supervised the project and interpreted data. The authors declare no competing interests. Publisher Copyright: {\textcopyright} 2019 Elsevier Inc.",
year = "2020",
month = feb,
day = "5",
doi = "10.1016/j.neuron.2019.11.003",
language = "English (US)",
volume = "105",
pages = "506--521.e7",
journal = "Neuron",
issn = "0896-6273",
publisher = "Cell Press",
number = "3",
}