Abstract | BACKGROUND: CASE PRESENTATION: We report of a 25-year-old male patient with epilepsy since the age of 18 and displaying clinical signs of LE and a high titer of GAD65 ABs in cerebrospinal fluid (CSF) and serum. Monthly, repetitive, intravenous cortisone pulse therapies that were initially administered for 6 months failed to improve his condition. Subsequent flow-cytometry analysis of CSF showed especially an increased fraction of activated HLA-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/month was started. After 3 months, the fCD8(+)TL in the CSF normalized; after 6 months, the psychological impulse-control deficits normalized; and after 11 months the patient was seizure free. However, 7 weeks later, seizures and, later on, psychological deficits recurred and fCD8(+)TL was once again present in the CSF. Flumazenil PET, magnetic resonance imaging-volumetry, and neuropsychological changes during therapy are described. CONCLUSION: The correlation of the fCD8(+)TL in the CSF with clinical and paraclinical measures of disease activity combined with the unambiguous response to basiliximab strongly 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 of human anti- drug ABs, a well-known complication of therapy with chimeric ABs.
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Authors | Guido Widman, Kristin Golombeck, Hubertus Hautzel, Catharina C Gross, Carlos M Quesada, Juri-Alexander Witt, Elena Rota-Kops, Johannes Ermert, Susanne Greschus, Rainer Surges, Christoph Helmstaedter, Heinz Wiendl, Nico Melzer, Christian E Elger |
Journal | Frontiers in neurology
(Front Neurol)
Vol. 6
Pg. 167
( 2015)
ISSN: 1664-2295 [Print] Switzerland |
PMID | 26284025
(Publication Type: Journal Article)
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