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@PHDTHESIS{Kreye:144861,
author = {Kreye, Jakob},
title = {{R}epertoireanalyse monoklonaler {A}ntikörper des
{L}iquors von {P}atienten mit
anti-{NMDA}-{R}ezeptor-{E}nzephalitis},
school = {Charité Universitätsmedizin Berlin},
type = {Habilitationsschrift},
publisher = {Charité - Universitätsmedizin Berlin},
reportid = {DZNE-2020-00286},
pages = {28},
year = {2017},
note = {Habilitationsschrift, Charité Universitätsmedizin Berlin,
2017},
abstract = {The anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis
is the most common form of autoimmune mediated brain
inflammation. Typically patients initially develop psychotic
changes such as substantive thought disorders and
hallucinations, followed by neurological symptoms like
impaired consciousness, dyskinesia, seizures, autonomic
dysregulations and central hypoventilation. The detection of
antibodies against the NR1 subunit of the NMDAR in the
cerebrospinal fluid (CSF) is the most important diagnostic
criterion. Previous works with extracted whole
immunoglobulins from patients‘ CSF and serum have shown
synaptic dysfunction in vitro and in vivo. Thereby an
influence of further possible autoantibodies could not be
eliminated and the proof of pathogenicity of NR1-specific
antibodies was pending. Therefore we generated monoclonal
recombinant human antibodies from CSF samples from eight
patients with NMDAR encephalitis. We isolated single memory
B cells and plasma cells via flow cytometry. The variable
gene regions coding the heavy and light immunoglobulin
chains were amplified by polymerase chain reaction. These
were cloned into vectors, containing the constant gene
sequence of human antibodies. We co-transfected human
embryonic kidney (HEK) cells with the respective pair of
heavy and light immunoglobulin chains and harvested
monoclonal antibodies from the culture medium. Six percent
of the generated monoclonal recombinant human antibodies
showed reactivity to NR1-transfected HEK cells, likewise the
typical NR1-specific staining pattern on hippocampal neurons
and mouse brain sections. All NR1-specific antibodies were
from the IgG isotype and have been isolated from plasma
cells as well as memory B cells. Last-mentioned ones might
be clinically relevant as possible origin of future
relapses. Several of the NR1-specific antibodies showed
clonal expansion with 100 percent identical clones and few
somatic hypermutations, indicating a recent peripheral
immune reaction. Furthermore we found three completely
unmutated NR1-specific antibodies („naturally occurring
antibodies“), suggesting incomplete immune tolerance
against NR1 in these patients. Incubating hippocampal
neurons with monoclonal NR1-specific antibodies caused a
reduction of the NMDAR density and NMDAR-specific currents.
After intravenous injections into mice, antibody enrichment
in the hippocampus and cerebellum was detected. More than 95
percent of the non- NR1-binding antibodies showed specific
reactivity on mouse brain sections, amongst others to
neuronal surfaces, glia cells and endothelium. With these
data we proved that monoclonal NR1-specific antibodies alone
are neurotoxic. Therewith, any detection of these
autoantibodies needs to be considered as a risk factor for
neuro-psychiatric symptoms. Future work will clarify whether
the presence of different NR1-specific antibodies is of
prognostic relevance and whether further autoantibodies
contribute to the pathophysiology.},
keywords = {anti-NMDA-receptor-Encephalitis (Other) / monoclonal
antibodies (Other) / autoantibodies (Other) / cerebrospinal
fluid (Other) / electrophysiology (Other) / 600 Technik,
Medizin, angewandte Wissenschaften::610 Medizin und
Gesundheit (Other)},
cin = {AG Prüß},
cid = {I:(DE-2719)1810003},
pnm = {342 - Disease Mechanisms and Model Systems (POF3-342)},
pid = {G:(DE-HGF)POF3-342},
typ = {PUB:(DE-HGF)13},
urn = {urn:nbn:de:kobv:188-fudissthesis000000105850-6},
doi = {10.17169/REFUBIUM-10947},
url = {https://pub.dzne.de/record/144861},
}