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@ARTICLE{Mori:138773,
author = {Mori, Kohji and Nihei, Yoshihiro and Arzberger, Thomas and
Zhou, Qihui and Mackenzie, Ian R and Hermann, Andreas and
Hanisch, Frank and Degeneration, German Consortium for
Frontotemporal Lobar and Alliance, Bavarian Brain Banking
and Kamp, Frits and Nuscher, Brigitte and Orozco, Denise and
Edbauer, Dieter and Haass, Christian},
title = {{R}educed hn{RNPA}3 increases {C}9orf72 repeat {RNA} levels
and dipeptide-repeat protein deposition.},
journal = {EMBO reports},
volume = {17},
number = {9},
issn = {1469-221X},
address = {Hoboken, NJ [u.a.]},
publisher = {Wiley},
reportid = {DZNE-2020-05095},
pages = {1314-1325},
year = {2016},
abstract = {Intronic hexanucleotide (G4C2) repeat expansions in C9orf72
are genetically associated with frontotemporal lobar
degeneration (FTLD) and amyotrophic lateral sclerosis (ALS).
The repeat RNA accumulates within RNA foci but is also
translated into disease characterizing dipeptide repeat
proteins (DPR). Repeat-dependent toxicity may affect nuclear
import. hnRNPA3 is a heterogeneous nuclear
ribonucleoprotein, which specifically binds to the G4C2
repeat RNA We now report that a reduction of nuclear hnRNPA3
leads to an increase of the repeat RNA as well as DPR
production and deposition in primary neurons and a novel
tissue culture model that reproduces features of the C9orf72
pathology. In fibroblasts derived from patients carrying
extended C9orf72 repeats, nuclear RNA foci accumulated upon
reduction of hnRNPA3. Neurons in the hippocampus of C9orf72
patients are frequently devoid of hnRNPA3. Reduced nuclear
hnRNPA3 in the hippocampus of patients with extended C9orf72
repeats correlates with increased DPR deposition. Thus,
reduced hnRNPA3 expression in C9orf72 cases leads to
increased levels of the repeat RNA as well as enhanced
production and deposition of DPR proteins and RNA foci.},
keywords = {Animals / Brain: metabolism / C9orf72 Protein / Dipeptides:
metabolism / Fibroblasts / Gene Expression Regulation / Gene
Knockdown Techniques / HeLa Cells / Heterogeneous-Nuclear
Ribonucleoprotein Group A-B: metabolism / Humans / Neurons:
metabolism / Protein Binding / Protein Transport / Proteins:
genetics / Pyramidal Cells: metabolism / RNA Transport /
RNA, Messenger: genetics / RNA, Small Interfering: genetics
/ Rats / C9orf72 Protein (NLM Chemicals) / C9orf72 protein,
human (NLM Chemicals) / Dipeptides (NLM Chemicals) / HNRNPA3
protein, human (NLM Chemicals) / Heterogeneous-Nuclear
Ribonucleoprotein Group A-B (NLM Chemicals) / Proteins (NLM
Chemicals) / RNA, Messenger (NLM Chemicals) / RNA, Small
Interfering (NLM Chemicals)},
cin = {AG Levin / AG Edbauer / AG Haass / AG Teipel / AG Zhou},
ddc = {570},
cid = {I:(DE-2719)1111016 / I:(DE-2719)1110004 /
I:(DE-2719)1110007 / I:(DE-2719)1510100 /
I:(DE-2719)5000080},
pnm = {344 - Clinical and Health Care Research (POF3-344) / 342 -
Disease Mechanisms and Model Systems (POF3-342)},
pid = {G:(DE-HGF)POF3-344 / G:(DE-HGF)POF3-342},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:27461252},
pmc = {pmc:PMC5007570},
doi = {10.15252/embr.201541724},
url = {https://pub.dzne.de/record/138773},
}