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@ARTICLE{Madlener:1005758,
author = {Madlener, Marie and Strippel, Christine and Thaler,
Franziska S. and Doppler, Kathrin and Wandinger, Klaus P.
and Lewerenz, Jan and Ringelstein, Marius and Roessling,
Rosa and Menge, Til and Wickel, Jonathan and Kellingshaus,
Christoph and Mues, Sigrid and Kraft, Andrea and Linsa,
Andreas and Tauber, Simone C. and Berg, Florian Then and
Gerner, Stefan T. and Paliantonis, Asterios and Finke,
Alexander and Priller, Josef and Schirotzek, Ingo and
Süße, Marie and Sühs, Kurt W. and Urbanek, Christian and
Senel, Makbule and Sommer, Claudia and Kuempfel, Tania and
Pruess, Harald and Fink, Gereon R. and Leypoldt, Frank and
Melzer, Nico and Malter, Michael P.},
title = {{G}lutamic acid decarboxylase antibody-associated
neurological syndromes: {C}linical and antibody
characteristics and therapy response},
journal = {Journal of the neurological sciences},
volume = {445},
issn = {0022-510x},
address = {Amsterdam [u.a.]},
publisher = {Elsevier Science},
reportid = {FZJ-2023-01612},
pages = {120540 -},
year = {2023},
abstract = {Background: Antibodies against glutamic acid decarboxylase
(GAD-abs) at high serum levels are associated with diverse
autoimmune neurological syndromes (AINS), including
cerebellar ataxia, epilepsy, limbic encephalitis and
stiff-person syndrome. The impact of low serum GAD-ab levels
in patients with suspected AINS remains controversial.
Specific intrathecal GAD-ab synthesis may serve as a marker
for GAD-ab-associated nervous system autoimmunity. We
present characteristics of a multicentric patient cohort
with suspected AINS associated with GAD antibodies
(SAINS-GAD+) and explore the relevance of serum GAD-ab
levels and intrathecal GAD-ab synthesis. Methods: All
patients with SAINS-GAD+ included in the registry of the
German Network for Research on Autoimmune Encephalitis
(GENERATE) from 2011 to 2019 were analyzed. High serum
GAD-ab levels were defined as RIA>2000 U/mL, ELISA>1000
U/mL, or as a positive staining pattern on cell-based
assays. Results: One-hundred-one patients were analyzed. In
descending order they presented with epilepsy/limbic
encephalitis $(39\%),$ cerebellar ataxia $(28\%),$ stiff
person syndrome $(22\%),$ and overlap syndrome $(12\%).$
Immunotherapy was administered in $89\%$ of cases with
improvements in $46\%.$ $35\%$ of SAINS-GAD+ patients had
low GAD-ab serum levels. Notably, unmatched oligoclonal
bands in CSF but not in serum were more frequent in patients
with low GAD-ab serum levels. GAD-ab-levels (high/low) and
intrathecal GAD-ab synthesis (present or not) did not impact
clinical characteristics and outcome. Conclusions: Overall,
immunotherapy in SAINS-GAD+ was moderately effective. Serum
GAD-ab levels and the absence or presence of intrathecal
GAD-ab synthesis did not predict clinical characteristics or
outcomes in SAINS-GAD+. The detection of unmatched
oligoclonal bands might outweigh low GAD-ab serum levels.},
cin = {INM-3},
ddc = {610},
cid = {I:(DE-Juel1)INM-3-20090406},
pnm = {5251 - Multilevel Brain Organization and Variability
(POF4-525)},
pid = {G:(DE-HGF)POF4-5251},
typ = {PUB:(DE-HGF)16},
pubmed = {36608627},
UT = {WOS:000923071300001},
doi = {10.1016/j.jns.2022.120540},
url = {https://juser.fz-juelich.de/record/1005758},
}