This title appears in the Scientific Report :
2016
Please use the identifier:
http://hdl.handle.net/2128/13263 in citations.
Please use the identifier: http://dx.doi.org/10.3389/fneur.2015.00167 in citations.
Treating a GAD65 Antibody-Associated Limbic Encephalitis with Basiliximab: A Case Study
Treating a GAD65 Antibody-Associated Limbic Encephalitis with Basiliximab: A Case Study
Background: Antibodies (ABs) against the 65-kDa isoform of the intracellular enzymeglutamate decarboxylase (GAD65) have been found in limbic encephalitis (LE) andother neurological conditions. The direct significance of anti-GAD65-ABs for epilepsyis unclear. However, in histological preparations fro...
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Personal Name(s): | Widman, Guido (Corresponding author) |
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Golombeck, Kristin / Hautzel, Hubertus / Gross, Catharina C. / Quesada, Carlos M. / Witt, Juri-Alexander / Rota Kops, Elena / Ermert, Johannes / Greschus, Susanne / Surges, Rainer / Helmstaedter, Christoph / Wiendl, Heinz / Melzer, Nico / Elger, Christian E. | |
Contributing Institute: |
Nuklearchemie; INM-5 Physik der Medizinischen Bildgebung; INM-4 Klinisch-Medizinische Einrichtungen (Univ. Düsseldorf); KME |
Published in: | Frontiers in neurology, 6 (2015) S. 167 |
Imprint: |
Lausanne
Frontiers Research Foundation
2015
|
PubMed ID: |
26284025 |
DOI: |
10.3389/fneur.2015.00167 |
Document Type: |
Journal Article |
Research Program: |
(Dys-)function and Plasticity |
Link: |
OpenAccess OpenAccess |
Publikationsportal JuSER |
Please use the identifier: http://dx.doi.org/10.3389/fneur.2015.00167 in citations.
Background: Antibodies (ABs) against the 65-kDa isoform of the intracellular enzymeglutamate decarboxylase (GAD65) have been found in limbic encephalitis (LE) andother neurological conditions. The direct significance of anti-GAD65-ABs for epilepsyis unclear. However, in histological preparations from biopsies of resective epilepsysurgeries, predominantly cytotoxic T-lymphocytes were detected making close contactsto neurons. Activated T-lymphocytes can, in turn, be selectively controlled by therapeuticinterleukin-2 receptor Abs, such as basiliximab.Case presentation: We report of a 25-year-old male patient with epilepsy since theage of 18 and displaying clinical signs of LE and a high titer of GAD65 ABs in cerebro-spinal fluid (CSF) and serum. Monthly, repetitive, intravenous cortisone pulse therapiesthat were initially administered for 6 months failed to improve his condition. Subsequentflow-cytometry analysis of CSF showed especially an increased fraction of activatedHLA-DR+CD8+T-lymphocytes (fCD8+TL) when compared to controls. Thus, a second,intravenous cortisone pulse therapy with an additional basiliximab dose of 20 mg/monthwas started. After 3 months, the fCD8+TL in the CSF normalized; after 6 months, thepsychological impulse-control deficits normalized; and after 11 months the patientwas seizure free. However, 7 weeks later, seizures and, later on, psychological deficitsrecurred and fCD8+TL was once again present in the CSF. Flumazenil PET, magneticresonance imaging-volumetry, and neuropsychological changes during therapy aredescribed.Conclusion: The correlation of the fCD8+TL in the CSF with clinical and paraclinical measures of disease activity combined with the unambiguous response to basiliximabstrongly argues in favor of the putative pathogenic role fCD8+TL in anti-GAD65 LE. The clinical relapse at the end of the observation period might be due to the formation ofhuman anti-drug ABs, a well-known complication of therapy with chimeric ABs. |