Clinical Characterization of Definite Autoimmune Limbic Encephalitis: A 30-case Series.
Adult
Age of Onset
Atrophy
/ immunology
Autoantibodies
/ metabolism
Autoimmune Diseases
/ ethnology
Child, Preschool
Cognitive Dysfunction
/ immunology
Female
Humans
Immunotherapy
/ statistics & numerical data
Japan
/ ethnology
Limbic Encephalitis
/ ethnology
Magnetic Resonance Imaging
Male
Middle Aged
Paraneoplastic Syndromes
/ ethnology
Potassium Channels, Voltage-Gated
/ immunology
Receptors, N-Methyl-D-Aspartate
/ immunology
Retrospective Studies
Temporal Lobe
/ immunology
Treatment Outcome
Young Adult
NMDAR
VGKC
autoimmune limbic encephalitis
immunotherapy
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
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
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