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@ARTICLE{Scheibe:163492,
author = {Scheibe, Franziska and Ostendorf, Lennard and Prüss,
Harald and Radbruch, Helena and Aschman, Tom and Hoffmann,
Sarah and Blau, Igor-Wolfgang and Meisel, Christian and
Alexander, Tobias and Meisel, Andreas},
title = {{D}aratumumab for treatment-refractory antibody-mediated
diseases in neurology.},
journal = {European journal of neurology},
volume = {29},
number = {6},
issn = {1351-5101},
address = {Oxford},
publisher = {Blackwell Science},
reportid = {DZNE-2022-00252},
pages = {1847-1854},
year = {2022},
note = {ISSN 1468-1331 not unique: **2 hits**. (CC BY-NC-ND)},
abstract = {A fraction of patients with antibody-mediated autoimmune
diseases remain unresponsive to first-/second-line and
sometimes even to escalation immunotherapies. Because these
patients are still affected by poor outcome and increased
mortality, we investigated the safety and efficacy of the
plasma cell-depleting anti-CD38 antibody daratumumab in
life-threatening, antibody-mediated autoimmune diseases.In
this retrospective, single-center case series, seven
patients with autoantibody-driven neurological autoimmune
diseases (autoimmune encephalitis, n = 5; neurofascin
antibody-associated chronic inflammatory demyelinating
polyneuropathy associated with sporadic late onset nemaline
myopathy, n = 1; seronegative myasthenia gravis, n = 1)
unresponsive to a median of four (range = 4-9)
immunotherapies were treated with four to 20 cycles of 16
mg/kg daratumumab.Daratumumab allowed a substantial clinical
improvement in all patients, as measured by modified Rankin
Scale (mRS; before treatment: mRS =5, n = 7; after
treatment: median mRS =4, range = 0-5), Clinical Assessment
Scale in Autoimmune Encephalitis (from median 21 to 3
points, n = 5), Inflammatory Neuropathy Cause and Treatment
disability score (from 7 to 0 points, n = 1), and
Quantitative Myasthenia Gravis score (from 16 to 8 points, n
= 1). Daratumumab induced a substantial reduction of
disease-specific autoreactive antibodies, total IgG (serum,
$66\%,$ n = 7; cerebrospinal fluid, $58\%,$ n = 5), and
vaccine-induced titers for rubella $(50\%)$ and tetanus
toxoid $(74\%).$ Treatment-related toxicities Grade 3 or
higher occurred in five patients, including one death.Our
findings suggest that daratumumab provided a clinically
relevant depletion of autoreactive long-lived plasma cells,
identifying plasma cell-targeted therapies as promising
escalation therapy for highly active, otherwise
treatment-refractory autoantibody-mediated neurological
diseases.},
keywords = {Antibodies, Monoclonal / Autoantibodies / Encephalitis /
Hashimoto Disease / Humans / Myasthenia Gravis / Nervous
System Diseases: drug therapy / Neurology / Retrospective
Studies / CIDP (Other) / autoimmune encephalitis (Other) /
daratumumab (Other) / myasthenia gravis (Other) / sporadic
late onset nemaline myopathy (Other)},
cin = {AG Prüß},
ddc = {610},
cid = {I:(DE-2719)1810003},
pnm = {353 - Clinical and Health Care Research (POF4-353)},
pid = {G:(DE-HGF)POF4-353},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:35098616},
doi = {10.1111/ene.15266},
url = {https://pub.dzne.de/record/163492},
}