Autoimmune inflammatory meningoencephalitis in a patient negative for glial fibrillary acidic protein-specific immunoglobulin G.


Journal

Multiple sclerosis and related disorders
ISSN: 2211-0356
Titre abrégé: Mult Scler Relat Disord
Pays: Netherlands
ID NLM: 101580247

Informations de publication

Date de publication:
Feb 2019
Historique:
received: 05 08 2018
revised: 08 11 2018
accepted: 28 12 2018
pubmed: 4 1 2019
medline: 14 6 2019
entrez: 4 1 2019
Statut: ppublish

Résumé

In this study, we describe clinical findings in a patient with autoimmune inflammatory meningoencephalitis who was negative for antibodies against glial fibrillary acidic protein (GFAP-IgG). Serum and cerebral spinal fluid (CSF) samples were collected from the patient as part of a study of 520 patients with neurological syndromes. Antibodies against GFAP and other proteins associated with neurological disorders were measured by rat brain- and cell-based indirect immunofluorescence assays. A 42-year-old female was diagnosed with autoimmune inflammatory meningoencephalitis. She experienced a subacute and relapsing course with decreased vision, fever, headache, ataxia, hemiplegia, and disturbance of consciousness. Brain magnetic resonance imaging showed extensive lesions in the white matter along the ventricle, brainstem, right internal capsule, and meninges. The patient responded well to steroid treatment. Examination of CSF revealed a normal white blood cell count and protein level. Serum and CSF were negative for GFAP-specific antibodies and all other autoantibodies tested. Immunohistochemical staining of a brain biopsy collected during relapse revealed chronic inflammation and severe edema. Extensive and strong staining of CD163+ macrophages were evident throughout the lesions; however, CD3+ cells were rare and CD138+ and CD20+ cells were absent. We describe a case of subacute corticosteroid-responsive nonvasculitic autoimmune inflammatory meningoencephalitis in the absence of GFAP-IgG. The pathological features were distinct from those of patients with GFAP-IgG-positive meningoencephalitis, suggesting that nonvasculitic autoimmune inflammatory meningoencephalitis is a heterogeneous neurological syndrome.

Sections du résumé

BACKGROUND BACKGROUND
In this study, we describe clinical findings in a patient with autoimmune inflammatory meningoencephalitis who was negative for antibodies against glial fibrillary acidic protein (GFAP-IgG).
METHODS METHODS
Serum and cerebral spinal fluid (CSF) samples were collected from the patient as part of a study of 520 patients with neurological syndromes. Antibodies against GFAP and other proteins associated with neurological disorders were measured by rat brain- and cell-based indirect immunofluorescence assays.
RESULTS RESULTS
A 42-year-old female was diagnosed with autoimmune inflammatory meningoencephalitis. She experienced a subacute and relapsing course with decreased vision, fever, headache, ataxia, hemiplegia, and disturbance of consciousness. Brain magnetic resonance imaging showed extensive lesions in the white matter along the ventricle, brainstem, right internal capsule, and meninges. The patient responded well to steroid treatment. Examination of CSF revealed a normal white blood cell count and protein level. Serum and CSF were negative for GFAP-specific antibodies and all other autoantibodies tested. Immunohistochemical staining of a brain biopsy collected during relapse revealed chronic inflammation and severe edema. Extensive and strong staining of CD163+ macrophages were evident throughout the lesions; however, CD3+ cells were rare and CD138+ and CD20+ cells were absent.
CONCLUSION CONCLUSIONS
We describe a case of subacute corticosteroid-responsive nonvasculitic autoimmune inflammatory meningoencephalitis in the absence of GFAP-IgG. The pathological features were distinct from those of patients with GFAP-IgG-positive meningoencephalitis, suggesting that nonvasculitic autoimmune inflammatory meningoencephalitis is a heterogeneous neurological syndrome.

Identifiants

pubmed: 30605794
pii: S2211-0348(18)30568-6
doi: 10.1016/j.msard.2018.12.037
pii:
doi:

Substances chimiques

Anti-Inflammatory Agents 0
Biomarkers 0
Methylprednisolone X4W7ZR7023

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

167-171

Informations de copyright

Copyright © 2018. Published by Elsevier B.V.

Auteurs

Longchang Xie (L)

Department of Neurology, the Second Affiliated Hospital of GuangZhou Medical University, 250# Changgang east Road, Guangzhou 510260, Guangdong Province, China; Institute of Neuroscience and The Second Affiliated Hospital of Guangzhou Medical University, Key Laboratory of Neurogenetics and Channelopathies of Guangdong Province and the Ministry of Education of China, Collaborative Innovation Center for Neurogenetics and Channelopathies, 250# Changgang east Road, Guangzhou 510260, Guangdong Province, China.

Tianni Liu (T)

Department of Neurology, the Second Affiliated Hospital of GuangZhou Medical University, 250# Changgang east Road, Guangzhou 510260, Guangdong Province, China; Institute of Neuroscience and The Second Affiliated Hospital of Guangzhou Medical University, Key Laboratory of Neurogenetics and Channelopathies of Guangdong Province and the Ministry of Education of China, Collaborative Innovation Center for Neurogenetics and Channelopathies, 250# Changgang east Road, Guangzhou 510260, Guangdong Province, China.

Haiyan Yao (H)

Department of Neurology, the Second Affiliated Hospital of GuangZhou Medical University, 250# Changgang east Road, Guangzhou 510260, Guangdong Province, China; Institute of Neuroscience and The Second Affiliated Hospital of Guangzhou Medical University, Key Laboratory of Neurogenetics and Channelopathies of Guangdong Province and the Ministry of Education of China, Collaborative Innovation Center for Neurogenetics and Channelopathies, 250# Changgang east Road, Guangzhou 510260, Guangdong Province, China.

Linzhan Wu (L)

Department of Neurology, the Second Affiliated Hospital of GuangZhou Medical University, 250# Changgang east Road, Guangzhou 510260, Guangdong Province, China; Institute of Neuroscience and The Second Affiliated Hospital of Guangzhou Medical University, Key Laboratory of Neurogenetics and Channelopathies of Guangdong Province and the Ministry of Education of China, Collaborative Innovation Center for Neurogenetics and Channelopathies, 250# Changgang east Road, Guangzhou 510260, Guangdong Province, China.

Wei Qiu (W)

Department of Neurology, The Third Affiliated Hospital of Guangzhou Medical University, 63# Duobao Road, Guangzhou 510150, China.

Baikeng Chen (B)

Department of Neurology, the Second Affiliated Hospital of GuangZhou Medical University, 250# Changgang east Road, Guangzhou 510260, Guangdong Province, China; Institute of Neuroscience and The Second Affiliated Hospital of Guangzhou Medical University, Key Laboratory of Neurogenetics and Channelopathies of Guangdong Province and the Ministry of Education of China, Collaborative Innovation Center for Neurogenetics and Channelopathies, 250# Changgang east Road, Guangzhou 510260, Guangdong Province, China.

Si Liu (S)

Department of Neurology, the Second Affiliated Hospital of GuangZhou Medical University, 250# Changgang east Road, Guangzhou 510260, Guangdong Province, China; Institute of Neuroscience and The Second Affiliated Hospital of Guangzhou Medical University, Key Laboratory of Neurogenetics and Channelopathies of Guangdong Province and the Ministry of Education of China, Collaborative Innovation Center for Neurogenetics and Channelopathies, 250# Changgang east Road, Guangzhou 510260, Guangdong Province, China.

Qingmei Huang (Q)

Department of Neurology, the Second Affiliated Hospital of GuangZhou Medical University, 250# Changgang east Road, Guangzhou 510260, Guangdong Province, China; Institute of Neuroscience and The Second Affiliated Hospital of Guangzhou Medical University, Key Laboratory of Neurogenetics and Channelopathies of Guangdong Province and the Ministry of Education of China, Collaborative Innovation Center for Neurogenetics and Channelopathies, 250# Changgang east Road, Guangzhou 510260, Guangdong Province, China.

Huacai Yang (H)

Department of Neurology, the Second Affiliated Hospital of GuangZhou Medical University, 250# Changgang east Road, Guangzhou 510260, Guangdong Province, China; Institute of Neuroscience and The Second Affiliated Hospital of Guangzhou Medical University, Key Laboratory of Neurogenetics and Channelopathies of Guangdong Province and the Ministry of Education of China, Collaborative Innovation Center for Neurogenetics and Channelopathies, 250# Changgang east Road, Guangzhou 510260, Guangdong Province, China.

Huiming Xu (H)

Department of Neurology, the Second Affiliated Hospital of GuangZhou Medical University, 250# Changgang east Road, Guangzhou 510260, Guangdong Province, China; Institute of Neuroscience and The Second Affiliated Hospital of Guangzhou Medical University, Key Laboratory of Neurogenetics and Channelopathies of Guangdong Province and the Ministry of Education of China, Collaborative Innovation Center for Neurogenetics and Channelopathies, 250# Changgang east Road, Guangzhou 510260, Guangdong Province, China.

Youming Long (Y)

Department of Neurology, the Second Affiliated Hospital of GuangZhou Medical University, 250# Changgang east Road, Guangzhou 510260, Guangdong Province, China; Institute of Neuroscience and The Second Affiliated Hospital of Guangzhou Medical University, Key Laboratory of Neurogenetics and Channelopathies of Guangdong Province and the Ministry of Education of China, Collaborative Innovation Center for Neurogenetics and Channelopathies, 250# Changgang east Road, Guangzhou 510260, Guangdong Province, China. Electronic address: youminglong@126.com.

Cong Gao (C)

Department of Neurology, the Second Affiliated Hospital of GuangZhou Medical University, 250# Changgang east Road, Guangzhou 510260, Guangdong Province, China; Institute of Neuroscience and The Second Affiliated Hospital of Guangzhou Medical University, Key Laboratory of Neurogenetics and Channelopathies of Guangdong Province and the Ministry of Education of China, Collaborative Innovation Center for Neurogenetics and Channelopathies, 250# Changgang east Road, Guangzhou 510260, Guangdong Province, China.

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