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@ARTICLE{Dchs:258682,
author = {Düchs, Matthias and Blazevic, Dragica and Rechtsteiner,
Philipp and Kenny, Cynthia and Lamla, Thorsten and Low,
Sarah and Savistchenko, Jimmy and Neumann, Manuela and
Melki, Ronald and Schönberger, Tanja and Stierstorfer,
Birgit and Wyatt, David and Igney, Frederik and Ciossek,
Thomas},
title = {{AAV}-mediated expression of a new conformational
anti-aggregated α-synuclein antibody prolongs survival in a
genetic model of α-synucleinopathies.},
journal = {npj Parkinson's Disease},
volume = {9},
number = {1},
issn = {2373-8057},
address = {London [u.a.]},
publisher = {Nature Publ. Group},
reportid = {DZNE-2023-00655},
pages = {91},
year = {2023},
abstract = {Prion-like transmission of pathology in
α-synucleinopathies like Parkinson's disease or multiple
system atrophy is increasingly recognized as one potential
mechanism to address disease progression. Active and passive
immunotherapies targeting insoluble, aggregated α-synuclein
are already being actively explored in the clinic with mixed
outcomes so far. Here, we report the identification of
306C7B3, a highly selective, aggregate-specific α-synuclein
antibody with picomolar affinity devoid of binding to the
monomeric, physiologic protein. 306C7B3 binding is
Ser129-phosphorylation independent and shows high affinity
to several different aggregated α-synuclein polymorphs,
increasing the likelihood that it can also bind to the
pathological seeds assumed to drive disease progression in
patients. In support of this, highly selective binding to
pathological aggregates in postmortem brains of MSA patients
was demonstrated, with no staining in samples from other
human neurodegenerative diseases. To achieve CNS exposure of
306C7B3, an adeno-associated virus (AAV) based approach
driving expression of the secreted antibody within the brain
of (Thy-1)-[A30P]-hα-synuclein mice was used. Widespread
central transduction after intrastriatal inoculation was
ensured by using the AAV2HBKO serotype, with transduction
being spread to areas far away from the inoculation site.
Treatment of (Thy-1)-[A30P]-hα-synuclein mice at the age of
12 months demonstrated significantly increased survival,
with 306C7B3 concentration reaching 3.9 nM in the
cerebrospinal fluid. These results suggest that AAV-mediated
expression of 306C7B3, targeting extracellular, presumably
disease-propagating aggregates of α-synuclein, has great
potential as a disease-modifying therapy for
α-synucleinopathies as it ensures CNS exposure of the
antibody, thereby mitigating the selective permeability of
the blood-brain barrier.},
cin = {AG Neumann},
ddc = {610},
cid = {I:(DE-2719)1210003},
pnm = {352 - Disease Mechanisms (POF4-352)},
pid = {G:(DE-HGF)POF4-352},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:37322068},
pmc = {pmc:PMC10272115},
doi = {10.1038/s41531-023-00542-9},
url = {https://pub.dzne.de/record/258682},
}