Cell Replacement Therapy Improves Pathological Hallmarks in a Mouse Model of Leukodystrophy Vanishing White Matter.
Animals
Astrocytes
/ pathology
Cell Differentiation
/ physiology
Cell- and Tissue-Based Therapy
/ methods
Disease Models, Animal
Female
Humans
Leukoencephalopathies
/ pathology
Male
Mice
Mice, Inbred C57BL
Myelin Sheath
/ pathology
Neuroglia
/ pathology
Stem Cell Transplantation
/ methods
Stem Cells
/ pathology
White Matter
/ pathology
astrocytes
cell replacement therapy
glial cells
leukodystrophy
oligodendrocytes
stem cells
vanishing white matter
white matter disorder
Journal
Stem cell reports
ISSN: 2213-6711
Titre abrégé: Stem Cell Reports
Pays: United States
ID NLM: 101611300
Informations de publication
Date de publication:
05 03 2019
05 03 2019
Historique:
received:
12
09
2017
revised:
18
01
2019
accepted:
21
01
2019
pubmed:
26
2
2019
medline:
7
3
2020
entrez:
26
2
2019
Statut:
ppublish
Résumé
Stem cell therapy has great prospects for brain white matter disorders, including the genetically determined disorders called leukodystrophies. We focus on the devastating leukodystrophy vanishing white matter (VWM). Patients with VWM show severe disability and early death, and treatment options are lacking. Previous studies showed successful cell replacement therapy in rodent models for myelin defects. However, proof-of-concept studies of allogeneic cell replacement in models representative of human leukodystrophies are lacking. We tested cell replacement in a mouse model representative of VWM. We transplanted different murine glial progenitor cell populations and showed improved pathological hallmarks and motor function. Improved mice showed a higher percentage of transplanted cells that differentiated into GFAP
Identifiants
pubmed: 30799272
pii: S2213-6711(19)30020-7
doi: 10.1016/j.stemcr.2019.01.018
pmc: PMC6411482
pii:
doi:
Types de publication
Journal Article
Research Support, Non-U.S. Gov't
Langues
eng
Sous-ensembles de citation
IM
Pagination
441-450Informations de copyright
Copyright © 2019 The Authors. Published by Elsevier Inc. All rights reserved.
Références
Behav Genet. 1990 Mar;20(2):213-34
pubmed: 1693848
Am J Hum Genet. 2017 Apr 6;100(4):617-634
pubmed: 28366443
Cell Stem Cell. 2013 Feb 7;12(2):252-64
pubmed: 23395447
Glob J Health Sci. 2015 Nov 03;8(7):41-6
pubmed: 26925900
Proc Natl Acad Sci U S A. 2017 Mar 14;114(11):E2243-E2252
pubmed: 28246330
N Engl J Med. 2017 Mar 16;376(11):1038-1046
pubmed: 28296613
Cell Stem Cell. 2008 Jun 5;2(6):553-65
pubmed: 18522848
J Clin Invest. 2016 Apr 1;126(4):1512-24
pubmed: 26974157
Mol Ther. 2016 Mar;24(3):430-46
pubmed: 26755333
Nat Genet. 2001 Dec;29(4):383-8
pubmed: 11704758
J Neurosci. 2014 Nov 26;34(48):16153-61
pubmed: 25429155
J Neurosci. 2014 Sep 3;34(36):11929-47
pubmed: 25186741
Cell. 2006 Aug 25;126(4):663-76
pubmed: 16904174
Nat Biotechnol. 2011 Sep 25;29(10):934-41
pubmed: 21947029
Stem Cell Reports. 2014 Apr 24;2(5):648-61
pubmed: 24936452
Nature. 1993 Apr 1;362(6419):453-5
pubmed: 8464477
Ann Neurol. 1999 Nov;46(5):716-22
pubmed: 10553988
Brain. 2013 Jan;136(Pt 1):209-22
pubmed: 23365098
Neuropathol Appl Neurobiol. 2018 Jun;44(4):391-403
pubmed: 28953319
Stem Cells Dev. 2012 Aug 10;21(12):2222-38
pubmed: 22085254
Lancet Neurol. 2006 May;5(5):413-23
pubmed: 16632312
Exp Neurol. 2017 May;291:74-86
pubmed: 28163160
J Neurosci. 2009 Mar 11;29(10):3276-87
pubmed: 19279265
Neurol Clin Pract. 2016 Dec;6(6):506-514
pubmed: 29849248
Nat Med. 2005 Sep;11(9):966-72
pubmed: 16086023
Ann Neurol. 2009 Sep;66(3):343-54
pubmed: 19798728
Sci Transl Med. 2012 Oct 10;4(155):155ra137
pubmed: 23052294
Sci Transl Med. 2012 Oct 10;4(155):155ra136
pubmed: 23052293
Stem Cells. 2016 Apr;34(4):984-96
pubmed: 26676415
Stem Cell Res Ther. 2015 Mar 14;6:30
pubmed: 25888852