Anti-GABA-A Receptor Antibody-Mediated Epilepsia Partialis Continua After Treatment With Alemtuzumab: A Case Report.

Female Humans Epilepsia Partialis Continua / etiology Alemtuzumab / adverse effects Receptors, GABA-A Status Epilepticus Encephalitis Multiple Sclerosis / complications

Journal

Neurology(R) neuroimmunology & neuroinflammation
ISSN: 2332-7812
Titre abrégé: Neurol Neuroimmunol Neuroinflamm
Pays: United States
ID NLM: 101636388

Informations de publication

Date de publication:
07 2023
Historique:
received: 18 10 2022
accepted: 20 03 2023
medline: 22 5 2023
pubmed: 19 5 2023
entrez: 18 5 2023
Statut: epublish

Résumé

Patients with anti-GABA-A receptor encephalitis characteristically experience therapy-refractory epileptic seizures. General anesthesia is often required to terminate refractory status epilepticus. The immunologic mechanisms leading to antibody formation remain to be elucidated. Described triggers of anti-GABA-A autoimmunity are tumors, mainly thymomas, and herpes simplex encephalitis. We present a young woman with prediagnosis of relapse remitting multiple sclerosis (MS), treated with interferons, natalizumab, and alemtuzumab. Six months after one and only cycle of alemtuzumab, speech arrest and behavioral changes with aggressive and anxious traits appeared. She showed increasing motor convulsions resulting in focal status epilepticus. Anti-GABA-A receptor antibodies in CSF and serum were confirmed in different external laboratories, in a more extensive analysis after antibodies against NMDAR, CASPR2, LGI1, GABABR, and AMPAR were ruled out during in-house examination. Clinical condition improved temporarily with cortisone therapy, plasmapheresis, and IVIG but deteriorated rapidly after steroid discontinuation, resulting in brain biopsy. On histopathologic confirmation consistent with anti-GABA-A receptor antibody-associated CNS inflammation, completing the first rituximab cycle, continuing oral corticosteroids and supplementing immunosuppression with cyclosporine A led to quick recovery. Our case describes a severe autoantibody-induced encephalitis in a young patient with MS, with alemtuzumab as a potential trigger for anti-GABA-A receptor encephalitis.

Sections du résumé

BACKGROUND AND OBJECTIVES
Patients with anti-GABA-A receptor encephalitis characteristically experience therapy-refractory epileptic seizures. General anesthesia is often required to terminate refractory status epilepticus. The immunologic mechanisms leading to antibody formation remain to be elucidated. Described triggers of anti-GABA-A autoimmunity are tumors, mainly thymomas, and herpes simplex encephalitis.
METHODS
We present a young woman with prediagnosis of relapse remitting multiple sclerosis (MS), treated with interferons, natalizumab, and alemtuzumab. Six months after one and only cycle of alemtuzumab, speech arrest and behavioral changes with aggressive and anxious traits appeared. She showed increasing motor convulsions resulting in focal status epilepticus.
RESULTS
Anti-GABA-A receptor antibodies in CSF and serum were confirmed in different external laboratories, in a more extensive analysis after antibodies against NMDAR, CASPR2, LGI1, GABABR, and AMPAR were ruled out during in-house examination. Clinical condition improved temporarily with cortisone therapy, plasmapheresis, and IVIG but deteriorated rapidly after steroid discontinuation, resulting in brain biopsy. On histopathologic confirmation consistent with anti-GABA-A receptor antibody-associated CNS inflammation, completing the first rituximab cycle, continuing oral corticosteroids and supplementing immunosuppression with cyclosporine A led to quick recovery.
DISCUSSION
Our case describes a severe autoantibody-induced encephalitis in a young patient with MS, with alemtuzumab as a potential trigger for anti-GABA-A receptor encephalitis.

Identifiants

DOI: 10.1212/NXI.0000000000200123 PMID: 37202183 PMC: PMC10195099
pubmed: 37202183
pii: 10/4/e200123
doi: 10.1212/NXI.0000000000200123
pmc: PMC10195099
pii:
doi:

Substances chimiques

Alemtuzumab 3A189DH42V
Receptors, GABA-A 0

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Informations de copyright

Copyright © 2023 The Author(s). Published by Wolters Kluwer Health, Inc. on behalf of the American Academy of Neurology.

Références

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JAMA Neurol. 2017 Aug 1;74(8):961-969
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pubmed: 28202703
Neurology. 2020 Sep 1;95(9):399-401
pubmed: 32651290
Lancet Neurol. 2016 Apr;15(4):391-404
pubmed: 26906964
N Engl J Med. 2018 Mar 1;378(9):840-851
pubmed: 29490181
Mult Scler Relat Disord. 2019 Feb;28:31-33
pubmed: 30553166

Auteurs

Dominica Ratuszny (D)

From the Department of Neurology (D.R., T. Skripuletz, E.V., K.-W.S.); Clinic for Anesthesiology and Intensive Care (T. Stüber), Hannover Medical School; Outpatient Clinic of Neurology (K.G.), Neurozentrum, Itzehoe; Departments of Neurosurgery (P.E.), Nuclear Medicine (J.A.M.), Diagnostic and Interventional Neuroradiology (M.P.W.), and Neuropathology (F.F.), Institute of Pathology, Hannover Medical School, Germany. ratuszny.dominica@mh-hannover.de.

Thomas Skripuletz (T)

From the Department of Neurology (D.R., T. Skripuletz, E.V., K.-W.S.); Clinic for Anesthesiology and Intensive Care (T. Stüber), Hannover Medical School; Outpatient Clinic of Neurology (K.G.), Neurozentrum, Itzehoe; Departments of Neurosurgery (P.E.), Nuclear Medicine (J.A.M.), Diagnostic and Interventional Neuroradiology (M.P.W.), and Neuropathology (F.F.), Institute of Pathology, Hannover Medical School, Germany.
ORCID: 0000-0001-8550-335X

Thomas Stüber (T)

From the Department of Neurology (D.R., T. Skripuletz, E.V., K.-W.S.); Clinic for Anesthesiology and Intensive Care (T. Stüber), Hannover Medical School; Outpatient Clinic of Neurology (K.G.), Neurozentrum, Itzehoe; Departments of Neurosurgery (P.E.), Nuclear Medicine (J.A.M.), Diagnostic and Interventional Neuroradiology (M.P.W.), and Neuropathology (F.F.), Institute of Pathology, Hannover Medical School, Germany.

Emil Valizada (E)

From the Department of Neurology (D.R., T. Skripuletz, E.V., K.-W.S.); Clinic for Anesthesiology and Intensive Care (T. Stüber), Hannover Medical School; Outpatient Clinic of Neurology (K.G.), Neurozentrum, Itzehoe; Departments of Neurosurgery (P.E.), Nuclear Medicine (J.A.M.), Diagnostic and Interventional Neuroradiology (M.P.W.), and Neuropathology (F.F.), Institute of Pathology, Hannover Medical School, Germany.

Klaus Gehring (K)

From the Department of Neurology (D.R., T. Skripuletz, E.V., K.-W.S.); Clinic for Anesthesiology and Intensive Care (T. Stüber), Hannover Medical School; Outpatient Clinic of Neurology (K.G.), Neurozentrum, Itzehoe; Departments of Neurosurgery (P.E.), Nuclear Medicine (J.A.M.), Diagnostic and Interventional Neuroradiology (M.P.W.), and Neuropathology (F.F.), Institute of Pathology, Hannover Medical School, Germany.

Philipp Ertl (P)

From the Department of Neurology (D.R., T. Skripuletz, E.V., K.-W.S.); Clinic for Anesthesiology and Intensive Care (T. Stüber), Hannover Medical School; Outpatient Clinic of Neurology (K.G.), Neurozentrum, Itzehoe; Departments of Neurosurgery (P.E.), Nuclear Medicine (J.A.M.), Diagnostic and Interventional Neuroradiology (M.P.W.), and Neuropathology (F.F.), Institute of Pathology, Hannover Medical School, Germany.

Jörg Andreas Müller (JA)

From the Department of Neurology (D.R., T. Skripuletz, E.V., K.-W.S.); Clinic for Anesthesiology and Intensive Care (T. Stüber), Hannover Medical School; Outpatient Clinic of Neurology (K.G.), Neurozentrum, Itzehoe; Departments of Neurosurgery (P.E.), Nuclear Medicine (J.A.M.), Diagnostic and Interventional Neuroradiology (M.P.W.), and Neuropathology (F.F.), Institute of Pathology, Hannover Medical School, Germany.

Mike P Wattjes (MP)

From the Department of Neurology (D.R., T. Skripuletz, E.V., K.-W.S.); Clinic for Anesthesiology and Intensive Care (T. Stüber), Hannover Medical School; Outpatient Clinic of Neurology (K.G.), Neurozentrum, Itzehoe; Departments of Neurosurgery (P.E.), Nuclear Medicine (J.A.M.), Diagnostic and Interventional Neuroradiology (M.P.W.), and Neuropathology (F.F.), Institute of Pathology, Hannover Medical School, Germany.

Friedrich Feuerhake (F)

From the Department of Neurology (D.R., T. Skripuletz, E.V., K.-W.S.); Clinic for Anesthesiology and Intensive Care (T. Stüber), Hannover Medical School; Outpatient Clinic of Neurology (K.G.), Neurozentrum, Itzehoe; Departments of Neurosurgery (P.E.), Nuclear Medicine (J.A.M.), Diagnostic and Interventional Neuroradiology (M.P.W.), and Neuropathology (F.F.), Institute of Pathology, Hannover Medical School, Germany.

Kurt-Wolfram Sühs (KW)

From the Department of Neurology (D.R., T. Skripuletz, E.V., K.-W.S.); Clinic for Anesthesiology and Intensive Care (T. Stüber), Hannover Medical School; Outpatient Clinic of Neurology (K.G.), Neurozentrum, Itzehoe; Departments of Neurosurgery (P.E.), Nuclear Medicine (J.A.M.), Diagnostic and Interventional Neuroradiology (M.P.W.), and Neuropathology (F.F.), Institute of Pathology, Hannover Medical School, Germany.

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Classifications MeSH

questionsmedicales.fr › Eucaryotes › Animaux › Chordés › Vertébrés › Mammifères › Eutheria › Primates › Haplorhini › Catarrhini › Hominidae › Humains › Anti-GABA-A Receptor Antibody-Mediated...
questionsmedicales.fr › États, signes et symptômes pathologiques › Signes et symptômes › Manifestations neurologiques › Crises épileptiques › État de mal épileptique › Épilepsie partielle continue › Anti-GABA-A Receptor Antibody-Mediated...
questionsmedicales.fr › Acides aminés, peptides et protéines › Protéines › Globulines › Sérum-globulines › Immunoglobulines › Anticorps › Anticorps monoclonaux › Anticorps monoclonaux humanisés › Alemtuzumab › Anti-GABA-A Receptor Antibody-Mediated...
questionsmedicales.fr › Acides aminés, peptides et protéines › Protéines › Protéines membranaires › Récepteurs de surface cellulaire › Récepteurs aux neuromédiateurs › Récepteurs aux acides aminés › Récepteurs GABA › Récepteurs GABA-A › Anti-GABA-A Receptor Antibody-Mediated...
questionsmedicales.fr › États, signes et symptômes pathologiques › Signes et symptômes › Manifestations neurologiques › Crises épileptiques › État de mal épileptique › Anti-GABA-A Receptor Antibody-Mediated...
questionsmedicales.fr › Maladies du système nerveux › Encéphalite › Anti-GABA-A Receptor Antibody-Mediated...
questionsmedicales.fr › Maladies du système immunitaire › Maladies auto-immunes › Maladies auto-immunes du système nerveux › Maladies démyélinisantes auto-immunes du SNC › Sclérose en plaques › Anti-GABA-A Receptor Antibody-Mediated...