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@ARTICLE{Miedema:164917,
author = {Miedema, Suzanne S M and Mol, Merel O and Koopmans, Frank T
W and Hondius, David C and van Nierop, Pim and Menden, Kevin
and de Veij Mestdagh, Christina F and van Rooij, Jeroen and
Ganz, Andrea B and Paliukhovich, Iryna and Melhem, Shamiram
and Li, Ka Wan and Holstege, Henne and Rizzu, Patrizia and
van Kesteren, Ronald E and van Swieten, John C and Heutink,
Peter and Smit, August B},
title = {{D}istinct cell type-specific protein signatures in {GRN}
and {MAPT} genetic subtypes of frontotemporal dementia.},
journal = {Acta Neuropathologica Communications},
volume = {10},
issn = {2051-5960},
address = {London},
publisher = {Biomed Central},
reportid = {DZNE-2022-01328},
pages = {100},
year = {2022},
note = {CC BY},
abstract = {Frontotemporal dementia is characterized by progressive
atrophy of frontal and/or temporal cortices at an early age
of onset. The disorder shows considerable clinical,
pathological, and genetic heterogeneity. Here we
investigated the proteomic signatures of frontal and
temporal cortex from brains with frontotemporal dementia due
to GRN and MAPT mutations to identify the key cell types and
molecular pathways in their pathophysiology. We compared
patients with mutations in the GRN gene (n = 9) or with
mutations in the MAPT gene (n = 13) with non-demented
controls (n = 11). Using quantitative proteomic analysis on
laser-dissected tissues we identified brain region-specific
protein signatures for both genetic subtypes. Using
published single cell RNA expression data resources we
deduced the involvement of major brain cell types in driving
these different protein signatures. Subsequent gene ontology
analysis identified distinct genetic subtype- and cell
type-specific biological processes. For the GRN subtype, we
observed a distinct role for immune processes related to
endothelial cells and for mitochondrial dysregulation in
neurons. For the MAPT subtype, we observed distinct
involvement of dysregulated RNA processing, oligodendrocyte
dysfunction, and axonal impairments. Comparison with an
in-house protein signature of Alzheimer's disease brains
indicated that the observed alterations in RNA processing
and oligodendrocyte function are distinct for the
frontotemporal dementia MAPT subtype. Taken together, our
results indicate the involvement of different brain cell
types and biological mechanisms in genetic subtypes of
frontotemporal dementia. Furthermore, we demonstrate that
comparison of proteomic profiles of different disease
entities can separate general neurodegenerative processes
from disease-specific pathways, which may aid the
development of disease subtype-specific treatment
strategies.},
keywords = {Endothelial Cells: metabolism / Frontotemporal Dementia:
genetics / Frontotemporal Dementia: pathology / Humans /
Intercellular Signaling Peptides and Proteins: genetics /
Mutation: genetics / Pick Disease of the Brain /
Progranulins: genetics / Proteomics / tau Proteins: genetics
/ tau Proteins: metabolism / Cell type enrichment (Other) /
Frontotemporal dementia (Other) / GRN (Other) / Human brain
proteomics (Other) / MAPT (Other) / GRN protein, human (NLM
Chemicals) / Intercellular Signaling Peptides and Proteins
(NLM Chemicals) / MAPT protein, human (NLM Chemicals) /
Progranulins (NLM Chemicals) / tau Proteins (NLM Chemicals)},
cin = {AG Heutink 1 / AG Rizzu},
ddc = {610},
cid = {I:(DE-2719)1210002 / I:(DE-2719)1210009},
pnm = {354 - Disease Prevention and Healthy Aging (POF4-354)},
pid = {G:(DE-HGF)POF4-354},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:35799292},
pmc = {pmc:PMC9261008},
doi = {10.1186/s40478-022-01387-8},
url = {https://pub.dzne.de/record/164917},
}