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@ARTICLE{Butzlaff:836327,
author = {Butzlaff, Malte and Hannan, Shabab B. and Karsten, Peter
and Lenz, Sarah and Ng, Josephine and Voßfeldt, Hannes and
Prüßing, Katja and Pflanz, Ralf and Schulz, Jörg B. and
Rasse, Tobias and Voigt, Aaron},
title = {{I}mpaired retrograde transport by the {D}ynein/{D}ynactin
complex contributes to {T}au-induced toxicity},
journal = {Human molecular genetics},
volume = {24},
number = {13},
issn = {1460-2083},
address = {Oxford},
publisher = {Oxford Univ. Press},
reportid = {FZJ-2017-05448},
pages = {3623 - 3637},
year = {2015},
abstract = {The gene mapt codes for the microtubule-associated protein
Tau. The R406W amino acid substitution in Tau is associated
with frontotemporal dementia with parkinsonism linked to
chromosome 17 (FTDP-17) characterized by Tau-positive
filamentous inclusions. These filamentous Tau inclusions are
present in a group of neurodegenerative diseases known as
tauopathies, including Alzheimer's disease (AD). To gain
more insights into the pathomechanism of tauopathies, we
performed an RNAi-based large-scale screen in Drosophila
melanogaster to identify genetic modifiers of
Tau[R406W]-induced toxicity. A collection of RNAi lines,
putatively silencing more than 7000 genes, was screened for
the ability to modify Tau[R406W]-induced toxicity in vivo.
This collection covered more than $50\%$ of all protein
coding fly genes and more than $90\%$ of all fly genes known
to have a human ortholog. Hereby, we identified 62 genes
that, when silenced by RNAi, modified Tau-induced toxicity
specifically. Among these 62 modifiers were three subunits
of the Dynein/Dynactin complex. Analysis on segmental nerves
of fly larvae showed that pan neural Tau[R406W] expression
and concomitant silencing of Dynein/Dynactin complex members
synergistically caused strong pathological changes within
the axonal compartment, but only minor changes at synapses.
At the larval stage, these alterations did not cause
locomotion deficits, but became evident in adult flies. Our
data suggest that Tau-induced detrimental effects most
likely originate from axonal rather than synaptic
dysfunction and that impaired retrograde transport
intensifies detrimental effects of Tau in axons. In
conclusion, our findings contribute to the elucidation of
disease mechanisms in tauopathies like FTDP-17 or AD.},
cin = {INM-11},
ddc = {570},
cid = {I:(DE-Juel1)INM-11-20170113},
pnm = {572 - (Dys-)function and Plasticity (POF3-572)},
pid = {G:(DE-HGF)POF3-572},
typ = {PUB:(DE-HGF)16},
UT = {WOS:000357523900003},
pubmed = {pmid:25794683},
doi = {10.1093/hmg/ddv107},
url = {https://juser.fz-juelich.de/record/836327},
}