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@ARTICLE{Mori:137126,
author = {Mori, Kohji and Arzberger, Thomas and Grässer, Friedrich A
and Gijselinck, Ilse and May, Stephanie and Rentzsch,
Kristin and Weng, Shih-Ming and Schludi, Martin H and van
der Zee, Julie and Cruts, Marc and Van Broeckhoven,
Christine and Kremmer, Elisabeth and Kretzschmar, Hans A and
Haass, Christian and Edbauer, Dieter},
title = {{B}idirectional transcripts of the expanded {C}9orf72
hexanucleotide repeat are translated into aggregating
dipeptide repeat proteins.},
journal = {Acta neuropathologica},
volume = {126},
number = {6},
issn = {0001-6322},
address = {Heidelberg},
publisher = {Springer},
reportid = {DZNE-2020-03448},
pages = {881-893},
year = {2013},
abstract = {Massive GGGGCC repeat expansion in the first intron of the
gene C9orf72 is the most common known cause of familial
frontotemporal lobar degeneration (FTLD) and amyotrophic
lateral sclerosis (ALS). Despite its intronic localization
and lack of an ATG start codon, the repeat region is
translated in all three reading frames into aggregating
dipeptide-repeat (DPR) proteins, poly-(Gly-Ala),
poly-(Gly-Pro) and poly-(Gly-Arg). We took an antibody-based
approach to further validate the translation of DPR
proteins. To test whether the antisense repeat RNA
transcript is also translated, we raised antibodies against
the predicted products, poly-(Ala-Pro) and poly-(Pro-Arg).
Both antibodies stained p62-positive neuronal cytoplasmic
inclusions throughout the cerebellum and hippocampus
indicating that not only sense but also antisense strand
repeats are translated into DPR proteins in the absence of
ATG start codons. Protein products of both strands
co-aggregate suggesting concurrent translation of both
strands. Moreover, an antibody targeting the putative
carboxyl terminus of DPR proteins can detect inclusion
pathology in C9orf72 repeat expansion carriers suggesting
that the non-ATG translation continues through the entire
repeat and beyond. A highly sensitive monoclonal antibody
against poly-(Gly-Arg), visualized abundant inclusion
pathology in all cortical regions and some inclusions also
in motoneurons. Together, our data show that the GGGGCC
repeat is bidirectionally translated into five distinct DPR
proteins that co-aggregate in the characteristic
p62-positive TDP-43 negative inclusions found in FTLD/ALS
cases with C9orf72 repeat expansion. Novel monoclonal
antibodies against poly-(Gly-Arg) will facilitate
pathological diagnosis of C9orf72 FTLD/ALS.},
keywords = {Amyotrophic Lateral Sclerosis: diagnosis / Amyotrophic
Lateral Sclerosis: genetics / Amyotrophic Lateral Sclerosis:
metabolism / Brain: metabolism / C9orf72 Protein / DNA
Repeat Expansion / Frontotemporal Lobar Degeneration:
diagnosis / Frontotemporal Lobar Degeneration: genetics /
Frontotemporal Lobar Degeneration: metabolism / Humans /
Protein Biosynthesis / Proteins: genetics / Proteins:
metabolism / C9orf72 Protein (NLM Chemicals) / C9orf72
protein, human (NLM Chemicals) / Proteins (NLM Chemicals)},
cin = {AG Levin / AG Edbauer / Ext HZM / AG Haass},
ddc = {610},
cid = {I:(DE-2719)1111016 / I:(DE-2719)1110004 /
I:(DE-2719)5000050 / I:(DE-2719)1110007},
pnm = {342 - Disease Mechanisms and Model Systems (POF3-342) / 344
- Clinical and Health Care Research (POF3-344)},
pid = {G:(DE-HGF)POF3-342 / G:(DE-HGF)POF3-344},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:24132570},
doi = {10.1007/s00401-013-1189-3},
url = {https://pub.dzne.de/record/137126},
}