Autoimmune Encephalitis With mGluR1 Antibodies Presenting With Epilepsy, but Without Cerebellar Signs: A Case Report.

Autor: Vinke AM; From the Department of Neurology (A.M.V., J.H.J., R.P.W.R.), Maastricht University Medical Center; Department of Psychiatry and Neuropsychology (S.Z., C.C.-H., M.M.-D., P.M., M.L., P.M.M.), Maastricht University; School for Mental Health and Neuroscience (S.Z., C.C.-H., M.M.-D., P.M., M.L., P.M.M., R.P.W.R.), Maastricht University; Department of Biomedical Sciences and Medicine (C.C.-H.), University of Algarve, Faro, Portugal; Central Diagnostic Laboratory (J.G.M.C.D.), Maastricht University Medical Center; Department of Neurology (J.M.V.), Erasmus MC University Medical Center, Rotterdam; Department of Neurology (D.P.), Zuyderland Medical Center, Heerlen; and Academic Center for Epileptology Kempenhaeghe/MUMC+ (R.P.W.R.), Maastricht, the Netherlands., Zong S; From the Department of Neurology (A.M.V., J.H.J., R.P.W.R.), Maastricht University Medical Center; Department of Psychiatry and Neuropsychology (S.Z., C.C.-H., M.M.-D., P.M., M.L., P.M.M.), Maastricht University; School for Mental Health and Neuroscience (S.Z., C.C.-H., M.M.-D., P.M., M.L., P.M.M., R.P.W.R.), Maastricht University; Department of Biomedical Sciences and Medicine (C.C.-H.), University of Algarve, Faro, Portugal; Central Diagnostic Laboratory (J.G.M.C.D.), Maastricht University Medical Center; Department of Neurology (J.M.V.), Erasmus MC University Medical Center, Rotterdam; Department of Neurology (D.P.), Zuyderland Medical Center, Heerlen; and Academic Center for Epileptology Kempenhaeghe/MUMC+ (R.P.W.R.), Maastricht, the Netherlands., Janssen JH; From the Department of Neurology (A.M.V., J.H.J., R.P.W.R.), Maastricht University Medical Center; Department of Psychiatry and Neuropsychology (S.Z., C.C.-H., M.M.-D., P.M., M.L., P.M.M.), Maastricht University; School for Mental Health and Neuroscience (S.Z., C.C.-H., M.M.-D., P.M., M.L., P.M.M., R.P.W.R.), Maastricht University; Department of Biomedical Sciences and Medicine (C.C.-H.), University of Algarve, Faro, Portugal; Central Diagnostic Laboratory (J.G.M.C.D.), Maastricht University Medical Center; Department of Neurology (J.M.V.), Erasmus MC University Medical Center, Rotterdam; Department of Neurology (D.P.), Zuyderland Medical Center, Heerlen; and Academic Center for Epileptology Kempenhaeghe/MUMC+ (R.P.W.R.), Maastricht, the Netherlands., Correia-Hoffmann C; From the Department of Neurology (A.M.V., J.H.J., R.P.W.R.), Maastricht University Medical Center; Department of Psychiatry and Neuropsychology (S.Z., C.C.-H., M.M.-D., P.M., M.L., P.M.M.), Maastricht University; School for Mental Health and Neuroscience (S.Z., C.C.-H., M.M.-D., P.M., M.L., P.M.M., R.P.W.R.), Maastricht University; Department of Biomedical Sciences and Medicine (C.C.-H.), University of Algarve, Faro, Portugal; Central Diagnostic Laboratory (J.G.M.C.D.), Maastricht University Medical Center; Department of Neurology (J.M.V.), Erasmus MC University Medical Center, Rotterdam; Department of Neurology (D.P.), Zuyderland Medical Center, Heerlen; and Academic Center for Epileptology Kempenhaeghe/MUMC+ (R.P.W.R.), Maastricht, the Netherlands., Mané-Damas M; From the Department of Neurology (A.M.V., J.H.J., R.P.W.R.), Maastricht University Medical Center; Department of Psychiatry and Neuropsychology (S.Z., C.C.-H., M.M.-D., P.M., M.L., P.M.M.), Maastricht University; School for Mental Health and Neuroscience (S.Z., C.C.-H., M.M.-D., P.M., M.L., P.M.M., R.P.W.R.), Maastricht University; Department of Biomedical Sciences and Medicine (C.C.-H.), University of Algarve, Faro, Portugal; Central Diagnostic Laboratory (J.G.M.C.D.), Maastricht University Medical Center; Department of Neurology (J.M.V.), Erasmus MC University Medical Center, Rotterdam; Department of Neurology (D.P.), Zuyderland Medical Center, Heerlen; and Academic Center for Epileptology Kempenhaeghe/MUMC+ (R.P.W.R.), Maastricht, the Netherlands., Damoiseaux JGMC; From the Department of Neurology (A.M.V., J.H.J., R.P.W.R.), Maastricht University Medical Center; Department of Psychiatry and Neuropsychology (S.Z., C.C.-H., M.M.-D., P.M., M.L., P.M.M.), Maastricht University; School for Mental Health and Neuroscience (S.Z., C.C.-H., M.M.-D., P.M., M.L., P.M.M., R.P.W.R.), Maastricht University; Department of Biomedical Sciences and Medicine (C.C.-H.), University of Algarve, Faro, Portugal; Central Diagnostic Laboratory (J.G.M.C.D.), Maastricht University Medical Center; Department of Neurology (J.M.V.), Erasmus MC University Medical Center, Rotterdam; Department of Neurology (D.P.), Zuyderland Medical Center, Heerlen; and Academic Center for Epileptology Kempenhaeghe/MUMC+ (R.P.W.R.), Maastricht, the Netherlands., de Vries JM; From the Department of Neurology (A.M.V., J.H.J., R.P.W.R.), Maastricht University Medical Center; Department of Psychiatry and Neuropsychology (S.Z., C.C.-H., M.M.-D., P.M., M.L., P.M.M.), Maastricht University; School for Mental Health and Neuroscience (S.Z., C.C.-H., M.M.-D., P.M., M.L., P.M.M., R.P.W.R.), Maastricht University; Department of Biomedical Sciences and Medicine (C.C.-H.), University of Algarve, Faro, Portugal; Central Diagnostic Laboratory (J.G.M.C.D.), Maastricht University Medical Center; Department of Neurology (J.M.V.), Erasmus MC University Medical Center, Rotterdam; Department of Neurology (D.P.), Zuyderland Medical Center, Heerlen; and Academic Center for Epileptology Kempenhaeghe/MUMC+ (R.P.W.R.), Maastricht, the Netherlands., Pröpper D; From the Department of Neurology (A.M.V., J.H.J., R.P.W.R.), Maastricht University Medical Center; Department of Psychiatry and Neuropsychology (S.Z., C.C.-H., M.M.-D., P.M., M.L., P.M.M.), Maastricht University; School for Mental Health and Neuroscience (S.Z., C.C.-H., M.M.-D., P.M., M.L., P.M.M., R.P.W.R.), Maastricht University; Department of Biomedical Sciences and Medicine (C.C.-H.), University of Algarve, Faro, Portugal; Central Diagnostic Laboratory (J.G.M.C.D.), Maastricht University Medical Center; Department of Neurology (J.M.V.), Erasmus MC University Medical Center, Rotterdam; Department of Neurology (D.P.), Zuyderland Medical Center, Heerlen; and Academic Center for Epileptology Kempenhaeghe/MUMC+ (R.P.W.R.), Maastricht, the Netherlands., Molenaar P; From the Department of Neurology (A.M.V., J.H.J., R.P.W.R.), Maastricht University Medical Center; Department of Psychiatry and Neuropsychology (S.Z., C.C.-H., M.M.-D., P.M., M.L., P.M.M.), Maastricht University; School for Mental Health and Neuroscience (S.Z., C.C.-H., M.M.-D., P.M., M.L., P.M.M., R.P.W.R.), Maastricht University; Department of Biomedical Sciences and Medicine (C.C.-H.), University of Algarve, Faro, Portugal; Central Diagnostic Laboratory (J.G.M.C.D.), Maastricht University Medical Center; Department of Neurology (J.M.V.), Erasmus MC University Medical Center, Rotterdam; Department of Neurology (D.P.), Zuyderland Medical Center, Heerlen; and Academic Center for Epileptology Kempenhaeghe/MUMC+ (R.P.W.R.), Maastricht, the Netherlands., Losen M; From the Department of Neurology (A.M.V., J.H.J., R.P.W.R.), Maastricht University Medical Center; Department of Psychiatry and Neuropsychology (S.Z., C.C.-H., M.M.-D., P.M., M.L., P.M.M.), Maastricht University; School for Mental Health and Neuroscience (S.Z., C.C.-H., M.M.-D., P.M., M.L., P.M.M., R.P.W.R.), Maastricht University; Department of Biomedical Sciences and Medicine (C.C.-H.), University of Algarve, Faro, Portugal; Central Diagnostic Laboratory (J.G.M.C.D.), Maastricht University Medical Center; Department of Neurology (J.M.V.), Erasmus MC University Medical Center, Rotterdam; Department of Neurology (D.P.), Zuyderland Medical Center, Heerlen; and Academic Center for Epileptology Kempenhaeghe/MUMC+ (R.P.W.R.), Maastricht, the Netherlands., Martinez Martinez P; From the Department of Neurology (A.M.V., J.H.J., R.P.W.R.), Maastricht University Medical Center; Department of Psychiatry and Neuropsychology (S.Z., C.C.-H., M.M.-D., P.M., M.L., P.M.M.), Maastricht University; School for Mental Health and Neuroscience (S.Z., C.C.-H., M.M.-D., P.M., M.L., P.M.M., R.P.W.R.), Maastricht University; Department of Biomedical Sciences and Medicine (C.C.-H.), University of Algarve, Faro, Portugal; Central Diagnostic Laboratory (J.G.M.C.D.), Maastricht University Medical Center; Department of Neurology (J.M.V.), Erasmus MC University Medical Center, Rotterdam; Department of Neurology (D.P.), Zuyderland Medical Center, Heerlen; and Academic Center for Epileptology Kempenhaeghe/MUMC+ (R.P.W.R.), Maastricht, the Netherlands., Rouhl RPW; From the Department of Neurology (A.M.V., J.H.J., R.P.W.R.), Maastricht University Medical Center; Department of Psychiatry and Neuropsychology (S.Z., C.C.-H., M.M.-D., P.M., M.L., P.M.M.), Maastricht University; School for Mental Health and Neuroscience (S.Z., C.C.-H., M.M.-D., P.M., M.L., P.M.M., R.P.W.R.), Maastricht University; Department of Biomedical Sciences and Medicine (C.C.-H.), University of Algarve, Faro, Portugal; Central Diagnostic Laboratory (J.G.M.C.D.), Maastricht University Medical Center; Department of Neurology (J.M.V.), Erasmus MC University Medical Center, Rotterdam; Department of Neurology (D.P.), Zuyderland Medical Center, Heerlen; and Academic Center for Epileptology Kempenhaeghe/MUMC+ (R.P.W.R.), Maastricht, the Netherlands.
Jazyk: angličtina
Zdroj: Neurology(R) neuroimmunology & neuroinflammation [Neurol Neuroimmunol Neuroinflamm] 2022 Apr 21; Vol. 9 (4). Date of Electronic Publication: 2022 Apr 21 (Print Publication: 2022).
DOI: 10.1212/NXI.0000000000001171
Abstrakt: Objective: To describe the unique case history of a patient with mGluR1 antibodies, with mainly limbic and without cerebellar symptoms.
Methods: A 50-year-old woman initially presented with focal seizures with epigastric rising and déjà-vu sensations, next to cognitive complaints, and musical auditory hallucinations. MRI, EEG, and neuronal autoantibody tests were performed.
Results: EEG findings showed slow and sharp activity (sharp waves and sharp-wave-slow-wave complex) in the left temporal lobe. A test for autoantibodies was negative initially. Because of persistent symptoms, serum and CSF were tested 4 years later and found positive for mGluR1 antibodies. Treatment started with monthly IV immunoglobulins and azathioprine that was replaced by mycophenolate mofetil later. Especially cognitive symptoms and hallucinations did not respond well to the treatment. During treatment, mGluR1 antibodies remained present in CSF.
Discussion: Whereas cerebellar symptoms are present in 97% of mGluR1-positive cases, our patient presented without ataxia. Therefore, we suggest that the clinical presentation of patients with mGluR1 antibodies is probably more diverse than previously described. Testing for mGluR1 antibodies should be considered in patients with limbic encephalitis and epilepsy, especially when negative for more common antibodies.
(Copyright © 2022 The Author(s). Published by Wolters Kluwer Health, Inc. on behalf of the American Academy of Neurology.)
Databáze: MEDLINE