Clinical Characterization of Definite Autoimmune Limbic Encephalitis: A 30-case Series.


Journal

Internal medicine (Tokyo, Japan)
ISSN: 1349-7235
Titre abrégé: Intern Med
Pays: Japan
ID NLM: 9204241

Informations de publication

Date de publication:
01 Dec 2019
Historique:
pubmed: 23 8 2019
medline: 3 3 2020
entrez: 23 8 2019
Statut: ppublish

Résumé

Objective Limbic encephalitis (LE) is an inflammatory condition of the limbic system that has an acute or subacute onset. Several types of antibodies are related to the onset of LE, including anti-N-methyl D-aspartate receptor (NMDAR) antibodies and voltage-gated potassium channel (VGKC)-complex antibodies. However, the characteristics and prevalence of LE remain unclear, especially in Asian cohorts, due to the rarity. We aimed to survey their characteristics. Materials and Methods Data of 30 cases clinically defined as "definite autoimmune LE" (based on the standard criteria) were retrospectively collected. These patients were categorized into four subtypes: NMDAR (+) (n=8), VGKC (+) (n=2), antibodies related to paraneoplastic syndrome (n=2), and an antibody-negative group (uncategorized) (n=18). Results LE is rare in Japan, and affected only 30 of 16,759 hospital patients (0.2%) over a ten-year period. The NMDAR (+) group showed distinctive symptoms, while the other three groups had similar indications. Brain MRI indicated significant medial temporal lobe atrophy at one year follow up after discharge. The prevalence of cognitive dysfunction as a complication was 64% (9/14). First-line immunotherapy resulted in a good outcome. A drastic improvement was seen from 4.0±1.1 to 1.1+ on the modified Rankin Scale. A good treatment outcome was observed in all groups (NMDAR, VGKC, and uncategorized), suggesting the importance of an early clinical diagnosis and the early initiation of treatment. Furthermore, we reviewed 26 cases that were clinically diagnosed as definitive autoimmune LE in previous case reports. Conclusion Our findings show that the establishment of a clinical diagnosis based on the clinical criteria of definitive autoimmune LE is important for the initiation of immunotherapy.

Identifiants

pubmed: 31434821
doi: 10.2169/internalmedicine.3029-19
pmc: PMC6928500
doi:

Substances chimiques

Autoantibodies 0
Potassium Channels, Voltage-Gated 0
Receptors, N-Methyl-D-Aspartate 0

Types de publication

Journal Article Multicenter Study

Langues

eng

Sous-ensembles de citation

IM

Pagination

3369-3378

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Auteurs

Yuri Shojima (Y)

Department of Neurology, Juntendo University School of Medicine, Japan.

Kenya Nishioka (K)

Department of Neurology, Juntendo University School of Medicine, Japan.

Masao Watanabe (M)

Department of Neurology, Juntendo University Urayasu Hospital, Japan.

Takayuki Jo (T)

Department of Neurology, Juntendo Shizuoka Hospital, Japan.

Keiko Tanaka (K)

Department of Cellular Neurobiology, Brain Research Institute, Niigata University, Japan.

Hiroshi Takashima (H)

Department of Neurology and Geriatrics, Kagoshima University Graduate School of Medical and Dental Sciences, Japan.

Kazuyuki Noda (K)

Department of Neurology, Juntendo Shizuoka Hospital, Japan.

Yasuyuki Okuma (Y)

Department of Neurology, Juntendo Shizuoka Hospital, Japan.

Takao Urabe (T)

Department of Neurology, Juntendo University Urayasu Hospital, Japan.

Kazumasa Yokoyama (K)

Department of Neurology, Juntendo University School of Medicine, Japan.

Nobutaka Hattori (N)

Department of Neurology, Juntendo University School of Medicine, Japan.

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Classifications MeSH