Caspr2 antibody-associated limbic encephalitis: contribution of visual aided analysis of 18F-FDG PET images using normal database comparison

Sánchez-Ordúz, Liset; Pérez-Larraya, Gállego; Grisanti, Fabiana; Centeno, María; Arbizu, Javier

doi:10.1016/j.remnie.2019.11.003

Cited by 6 publications

(7 citation statements)

References 10 publications

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“…Molecular imaging such as 18 F-fluorodeoxi glucose positron emission tomography (FDG-PET) has been used recently to help identify anti-Caspr2 antibody-associated autoimmune encephalitis, and the most common abnormality was temporomesial hypometabolism. 27,28 Unfortunately, our patient could not afford the FDG-PET examination. In the future, more imaging techniques would be incorporated to present the detailed radiological features of autoimmune encephalitis associated with Caspr2-IgG.…”

Section: Discussionmentioning

confidence: 91%

Possible coexistence of MOG-IgG-associated disease and anti-Caspr2 antibody-associated autoimmune encephalitis: a first case report

Liu

Bai²,

et al. 2020

Ther Adv Neurol Disord

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Myelin oligodendrocyte glycoprotein antibody-associated disease has been proposed as a separate inflammatory demyelinating disease of the central nervous system (CNS) since the discovery of pathogenic antibodies against myelin oligodendrocyte glycoprotein (MOG-IgG). Antibodies targeting contactin-associated protein-like 2 (Caspr2), a component of voltage-gated potassium channel (VGKC) complex, have been documented to be associated with a novel autoimmune synaptic encephalitis with a low incidence. Herein, we reported an adult female with initial presentation of decreased vision in the right eye and subsequent episodes of neuropsychiatric disturbance including hypersomnia, agitation, apatheia, and memory impairment. Magnetic resonance imaging (MRI) revealed multiple lesions scattered in brain, brainstem, and cervical and thoracic spinal cord, showing hypointensity on T1-weighted images, hyperintensity on T2-weighted and fluid attenuated inversion recovery (FLAIR) images. Heterogenous patchy or ring-like enhancement was observed in the majority of lesions. The detection of low-titer MOG-IgG exclusively in cerebrospinal fluid (CSF; titer, 1:1) and Caspr2-IgG in both serum and CSF (titers, 1:100 and 1:1) led to a possible diagnosis of coexisting MOG-IgG-associated disease (MOGAD) and anti-Caspr2 antibody-associated autoimmune encephalitis. The patient was treated with immunosuppressive agents including corticosteroids and immunoglobulin, and achieved a sustained remission. To the best of our knowledge, this is the first report on the possible coexistence of MOGAD and anti-Caspr2 antibody-associated autoimmune encephalitis, which advocates for the recommendation of a broad spectrum screening for antibodies against well-defined CNS antigens in suspected patients with autoimmune-mediated diseases of the CNS.

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Section: Discussionmentioning

confidence: 91%

Possible coexistence of MOG-IgG-associated disease and anti-Caspr2 antibody-associated autoimmune encephalitis: a first case report

Liu

Bai²,

et al. 2020

Ther Adv Neurol Disord

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“…In a study by Bien et al, MRI findings enhanced the predictive accuracy in the titer range between 1:64 and 1:512 [ 46 ]. In addition, FDG-PET is also currently being applied as a diagnostic tool for these patients [ 6 , 52 , 53 ]. Although detecting MRI abnormalities in CASPR2 antibody-associated AE in children is limited, we need to combine MRI (other modern diagnostic tools) and antibodies to improve the effectiveness of diagnostic validity.…”

Section: Discussionmentioning

confidence: 99%

Contactin-associated protein-like 2 antibody-associated autoimmune encephalitis in children: case reports and systematic review of literature

et al. 2023

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Objectives To ascertain the clinical characteristics of pediatric patients with contactin-associated protein-like 2 (CASPR2) antibody-associated autoimmune encephalitis (AEs). Methods Two cases of CASPR2 antibody-associated AEs have been reported. In addition, a systematic search of literature published between January 2010 and March 2022 through six online databases was conducted to identify the pediatric patients with CASPR2 antibody-associated AEs. Data on demographics, clinical symptoms, laboratory examinations, imaging, treatment, and outcome were collected. Results Our updated literature search yielded 1,837 publications, of which 21 were selected, and 40 patients in this study met the diagnostic criteria for AE. There were 25 males and 15 females with a mean age of 9.2 years. The most common presenting symptoms are psychiatric symptoms (72.5%), sleep changes (62.5%), and movement disorders (60%). The psychiatric symptoms included mood changes (39.1%), behavior changes (25%), and hallucination (7.5%). In total, 23 cases (57.5%) combined with autonomic dysfunction, such as gastrointestinal dysmotility, cardiovascular-related symptoms, and sweating. No tumors were observed in children. Thirty-eight patients received first-line immunotherapy, and eight received first-line and second-line immunotherapy. All patients had a good clinical response to immune therapy. Mean mRS at onset was 3.4; It was 0.88 at the last follow-up. There was no recurrence during follow-up. Conclusion Psychiatric symptoms, sleep disorders, movement disorders, and cardiovascular-related symptoms are the most common presentation in pediatric patients with CASPR2 antibody-associated AEs. Tumor, particularly with thymoma, is uncommon in children diagnosed with CASPR2 antibody-associated AEs. In addition, prompt diagnosis and immunotherapy can relieve symptoms and improve the prognosis. Supplementary Information The online version contains supplementary material available at 10.1007/s13760-023-02174-5.

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“…Abnormal FDG‐PET brain scans have been reported in anti‐NMDAR encephalitis patients but metabolic patterns have been inconsistent across different regions. 4 , 5 , 6 , 7 , 8 , 9 , 10 , 11 , 12 , 13 , 14 , 15 , 16 , 17 , 18 , 19 , 20 , 25 , 27 , 28 , 29 , 30 In this study, we investigated the application of FDG‐PET during different stages of anti‐NMDAR encephalitis disease progression. First, notably (but unsurprisingly) we found that consistent with other diseases, the brain‐glucose metabolic patterns changed during different stages ( p < 0.05).…”

Section: Discussionmentioning

confidence: 99%

“…These data imply that earlier treatment results in a better prognosis. Moreover, our results were similar to those of other studies in general, 6 , 7 , 9 , 11 , 25 , 27 , 28 , 29 , 30 but were inconsistent in detail. We propose that this is due to the differences between PET intervals in each report.…”

Section: Discussionmentioning

confidence: 99%

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Hypometabolism of the left middle/medial frontal lobe onFDG‐PETin anti–NMDAreceptor encephalitis: Comparison withMRIandEEGfindings

et al. 2023

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Objectives To investigate changes in brain‐glucose metabolism in anti‐N‐methyl‐D‐aspartate receptor (NMDAR) encephalitis, and compare results with MRI and electroencephalography (EEG) findings at different disease stages. Methods The clinical data of 18 patients (median age, 35 years; 11 men) were retrospectively collected. Patients were divided into groups based on the time of symptom onset to examination, (≤1 month, >1 but ≤3 months, >3 months). Two‐sample t ‐test results were compared with age and sex‐paired healthy controls using statistical parametric mapping and verified using a NeuroQ software normal database with a discriminating z‐score of 2. Results Abnormal patterns on FDG‐PET differed over time ( T = 3.21–8.74, Z = 2.68–4.23, p < 0.005). Regional analysis showed hypometabolic left middle or medial frontal cortex in 4/5, 5/7, and 5/6 patients, respectively. Time‐subgroup analysis revealed hypermetabolic supertemporal cortex in 4/5, 5/7, and 2/6, patients, respectively. MRI and EEG abnormalities in any region and stage occurred in 10/18 and 10/16 patients, respectively. MRI and EEG time‐subgroup analysis showed abnormalities in 5/9, 4/5, and 1/4, and 1/3, 6/7, and 3/6 patients, respectively. Abnormal temporal lobes were detected most frequently in MRI analyses and occurred in 3/10 patients. Conclusions Decreased left middle/medial frontal metabolism could be common to all stages. Metabolism in other regions, MRI, and EEG results were associated with the progression of anti‐NMDAR encephalitis. The sensitivity rate of FDG‐PET was superior to that of MRI and EEG.

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Caspr2 antibody-associated limbic encephalitis: contribution of visual aided analysis of 18F-FDG PET images using normal database comparison

Cited by 6 publications

References 10 publications

Possible coexistence of MOG-IgG-associated disease and anti-Caspr2 antibody-associated autoimmune encephalitis: a first case report

Possible coexistence of MOG-IgG-associated disease and anti-Caspr2 antibody-associated autoimmune encephalitis: a first case report

Contactin-associated protein-like 2 antibody-associated autoimmune encephalitis in children: case reports and systematic review of literature

Hypometabolism of the left middle/medial frontal lobe onFDG‐PETin anti–NMDAreceptor encephalitis: Comparison withMRIandEEGfindings

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