Generation of a heterozygous and a homozygous CSF1R knockout line from iPSC using CRISPR/Cas9.


Journal

Stem cell research
ISSN: 1876-7753
Titre abrégé: Stem Cell Res
Pays: England
ID NLM: 101316957

Informations de publication

Date de publication:
06 2023
Historique:
received: 18 06 2022
revised: 05 03 2023
accepted: 08 03 2023
medline: 2 6 2023
pubmed: 23 3 2023
entrez: 22 3 2023
Statut: ppublish

Résumé

Mutations in Colony-stimulating factor 1 receptor (CSF1R) lead to CSF1R-related leukoencephalopathy, also known as Adult-onset leukoencephalopathy with axonal spheroids and pigmented glia (ALSP), a rapidly progressing neurodegenerative disease with severe cognitive and motor impairment. In this study, a homozygous and a heterozygous CSF1R knockout induced pluripotent stem cell (iPSC) line were generated by CRISPR/Cas9-based gene editing. These in vitro models will provide a helpful tool for investigating the still largely unknown pathophysiology of CSF1R-related leukoencephalopathy.

Identifiants

pubmed: 36947995
pii: S1873-5061(23)00052-1
doi: 10.1016/j.scr.2023.103066
pii:
doi:

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

103066

Informations de copyright

Copyright © 2023 The Author(s). Published by Elsevier B.V. All rights reserved.

Déclaration de conflit d'intérêts

Declaration of Competing Interest The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.

Auteurs

Anne S Schmitz (AS)

Hertie Institute for Clinical Brain Research, University of Tübingen, Tübingen, Germany; German Center for Neurodegenerative Diseases (DZNE), Tübingen, Germany.

Milena Korneck (M)

Hertie Institute for Clinical Brain Research, University of Tübingen, Tübingen, Germany; German Center for Neurodegenerative Diseases (DZNE), Tübingen, Germany; Department of Neurology, University Hospital Tübingen, Tübingen, Germany.

Janani Raju (J)

Hertie Institute for Clinical Brain Research, University of Tübingen, Tübingen, Germany; German Center for Neurodegenerative Diseases (DZNE), Tübingen, Germany.

Andrés Lamsfus-Calle (A)

University Children's Hospital, Department of Pediatrics I, Hematology and Oncology, University of Tübingen, Germany.

Alberto Daniel-Moreno (A)

University Children's Hospital, Department of Pediatrics I, Hematology and Oncology, University of Tübingen, Germany.

Justin S Antony (JS)

University Children's Hospital, Department of Pediatrics I, Hematology and Oncology, University of Tübingen, Germany.

Markus Mezger (M)

University Children's Hospital, Department of Pediatrics I, Hematology and Oncology, University of Tübingen, Germany.

Ludger Schöls (L)

Hertie Institute for Clinical Brain Research, University of Tübingen, Tübingen, Germany; German Center for Neurodegenerative Diseases (DZNE), Tübingen, Germany; Department of Neurology, University Hospital Tübingen, Tübingen, Germany.

Stefan Hauser (S)

Hertie Institute for Clinical Brain Research, University of Tübingen, Tübingen, Germany; German Center for Neurodegenerative Diseases (DZNE), Tübingen, Germany.

Stefanie N Hayer (SN)

Hertie Institute for Clinical Brain Research, University of Tübingen, Tübingen, Germany; German Center for Neurodegenerative Diseases (DZNE), Tübingen, Germany; Department of Neurology, University Hospital Tübingen, Tübingen, Germany. Electronic address: stefanie.hayer@med.uni-tuebingen.de.

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Classifications MeSH