Aberrant Development Corrected in Adult-Onset Huntington's Disease iPSC-Derived Neuronal Cultures via WNT Signaling Modulation.

Adult Age of Onset Cell Cycle / genetics Cell Differentiation / genetics Cells, Cultured Epigenesis, Genetic Humans Huntington Disease / genetics Induced Pluripotent Stem Cells / pathology Mitosis Neostriatum / pathology Neural Stem Cells / metabolism Neurons / pathology Transcription Factors / metabolism Transcriptome / genetics Up-Regulation / genetics Wnt Signaling Pathway
Huntington's disease WNT signaling adult-onset HD cell cycle development induced pluripotent stem cells medium spiny neurons neural stem cells single-cell RNA-seq

Journal

Stem cell reports
ISSN: 2213-6711
Titre abrégé: Stem Cell Reports
Pays: United States
ID NLM: 101611300

Informations de publication

Date de publication:
10 03 2020
Historique:
received: 19 08 2019
revised: 30 01 2020
accepted: 31 01 2020
pubmed: 29 2 2020
medline: 5 2 2021
entrez: 29 2 2020
Statut: ppublish

Résumé

Aberrant neuronal development and the persistence of mitotic cellular populations have been implicated in a multitude of neurological disorders, including Huntington's disease (HD). However, the mechanism underlying this potential pathology remains unclear. We used a modified protocol to differentiate induced pluripotent stem cells (iPSCs) from HD patients and unaffected controls into neuronal cultures enriched for medium spiny neurons, the cell type most affected in HD. We performed single-cell and bulk transcriptomic and epigenomic analyses and demonstrated that a persistent cyclin D1

Identifiants

DOI: 10.1016/j.stemcr.2020.01.015 PMID: 32109367 PMC: PMC7066322
pubmed: 32109367
pii: S2213-6711(20)30036-9
doi: 10.1016/j.stemcr.2020.01.015
pmc: PMC7066322
pii:
doi:

Substances chimiques

Transcription Factors 0

Types de publication

Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

406-419

Subventions

Organisme : NINDS NIH HHS
ID : U54 NS091046
Pays : United States
Organisme : NIGMS NIH HHS
ID : T32 GM087237
Pays : United States
Organisme : Medical Research Council
ID : MR/L023784/1
Pays : United Kingdom
Organisme : NIEHS NIH HHS
ID : P30 ES002109
Pays : United States
Organisme : Medical Research Council
ID : MR/L023784/2
Pays : United Kingdom
Organisme : NINDS NIH HHS
ID : R01 NS089076
Pays : United States

Informations de copyright

Copyright © 2020 The Authors. Published by Elsevier Inc. All rights reserved.

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Auteurs

Charlene Smith-Geater (C)

Department of Psychiatry and Human Behavior, University of California Irvine, Irvine, CA 92697, USA.

Sarah J Hernandez (SJ)

Department of Neurobiology and Behavior, University of California Irvine, Irvine, CA 96267, USA.

Ryan G Lim (RG)

Department of Memory Impairment and Neurological Disorders, University of California Irvine, Irvine, CA 92697, USA.

Miriam Adam (M)

Department of Biological Engineering, Massachusetts Institute of Technology, Cambridge, MA 02142, USA.

Jie Wu (J)

Department of Biological Chemistry, University of California Irvine, Irvine, CA 92617, USA.

Jennifer T Stocksdale (JT)

Department of Neurobiology and Behavior, University of California Irvine, Irvine, CA 96267, USA.

Brook T Wassie (BT)

LeadCrunch, San Diego, CA 92101, USA.

Maxwell Philip Gold (MP)

Department of Biological Engineering, Massachusetts Institute of Technology, Cambridge, MA 02142, USA.

Keona Q Wang (KQ)

Department of Neurobiology and Behavior, University of California Irvine, Irvine, CA 96267, USA.

Ricardo Miramontes (R)

Department of Memory Impairment and Neurological Disorders, University of California Irvine, Irvine, CA 92697, USA.

Lexi Kopan (L)

Department of Neurobiology and Behavior, University of California Irvine, Irvine, CA 96267, USA.

Iliana Orellana (I)

Department of Memory Impairment and Neurological Disorders, University of California Irvine, Irvine, CA 92697, USA.

Shona Joy (S)

Neural Stem Cell Institute, Rensselaer, NY 12144, USA.

Paul J Kemp (PJ)

School of Biosciences, Cardiff University, Cardiff CF10 3AX, UK.

Nicholas D Allen (ND)

School of Biosciences, Cardiff University, Cardiff CF10 3AX, UK.

Ernest Fraenkel (E)

Department of Biological Engineering, Massachusetts Institute of Technology, Cambridge, MA 02142, USA.

Leslie M Thompson (LM)

Department of Psychiatry and Human Behavior, University of California Irvine, Irvine, CA 92697, USA; Department of Neurobiology and Behavior, University of California Irvine, Irvine, CA 96267, USA; Department of Memory Impairment and Neurological Disorders, University of California Irvine, Irvine, CA 92697, USA; Department of Biological Engineering, Massachusetts Institute of Technology, Cambridge, MA 02142, USA; Department of Biological Chemistry, University of California Irvine, Irvine, CA 92617, USA. Electronic address: lmthomps@uci.edu.

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Classifications MeSH

questionsmedicales.fr › Personnes › Tranches d'âge › Adulte › Aberrant Development Corrected in Adult-Onset...
questionsmedicales.fr › Environnement et santé publique › Santé publique › Facteurs épidémiologiques › Facteurs âges › Âge de début › Aberrant Development Corrected in Adult-Onset...
questionsmedicales.fr › Phénomènes physiologiques cellulaires › Cycle cellulaire › Aberrant Development Corrected in Adult-Onset...
questionsmedicales.fr › Phénomènes physiologiques cellulaires › Différenciation cellulaire › Aberrant Development Corrected in Adult-Onset...
questionsmedicales.fr › Cellules › Cellules cultivées › Aberrant Development Corrected in Adult-Onset...
questionsmedicales.fr › Phénomènes génétiques › Régulation de l'expression des gènes › Épigenèse génétique › Aberrant Development Corrected in Adult-Onset...
questionsmedicales.fr › Eucaryotes › Animaux › Chordés › Vertébrés › Mammifères › Eutheria › Primates › Haplorhini › Catarrhini › Hominidae › Humains › Aberrant Development Corrected in Adult-Onset...
questionsmedicales.fr › Troubles mentaux › Troubles neurocognitifs › Démence › Maladie de Huntington › Aberrant Development Corrected in Adult-Onset...
questionsmedicales.fr › Cellules › Cellules souches › Cellules souches pluripotentes › Cellules souches pluripotentes induites › Aberrant Development Corrected in Adult-Onset...
questionsmedicales.fr › Phénomènes génétiques › Division du noyau cellulaire › Mitose › Aberrant Development Corrected in Adult-Onset...
questionsmedicales.fr › Système nerveux › Système nerveux central › Encéphale › Prosencéphale › Télencéphale › Cerveau › Noyaux gris centraux › Corps strié › Néostriatum › Aberrant Development Corrected in Adult-Onset...
questionsmedicales.fr › Cellules › Cellules souches › Cellules souches neurales › Aberrant Development Corrected in Adult-Onset...
questionsmedicales.fr › Cellules › Neurones › Aberrant Development Corrected in Adult-Onset...
questionsmedicales.fr › Acides aminés, peptides et protéines › Protéines › Facteurs de transcription › Aberrant Development Corrected in Adult-Onset...
questionsmedicales.fr › Phénomènes génétiques › Structures génétiques › Transcriptome › Aberrant Development Corrected in Adult-Onset...
questionsmedicales.fr › Phénomènes physiologiques › Phénomènes pharmacologiques et toxicologiques › Phénomènes pharmacologiques › Régulation positive › Aberrant Development Corrected in Adult-Onset...
questionsmedicales.fr › Phénomènes physiologiques cellulaires › Transduction du signal › Voie de signalisation Wnt › Aberrant Development Corrected in Adult-Onset...