The Antibody Assay in Suspected Autoimmune Encephalitis From Positive Rate to Test Strategies

Deng, Qun; Liu, Ye; Mao, Zhifeng; Chen, Yun; Yue, Ping; Zhu, Guoqiang; Zhao, Weiqing; Xiao, Hu; Zhou, Hao

doi:10.3389/fimmu.2022.803854

Cited by 4 publications

(16 citation statements)

References 32 publications

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“…CASPR2 antibodies in CSF and serum have diagnostic value for identifying individuals with CASPR2 antibody-associated AEs [ 7 , 8 , 46 , 47 ]. Since serum CASPR2 antibodies are common in pediatric AE patients, the detection rate in CSF is low [ 48 ]. Therefore, we need to pay more attention to the diagnostic value of serum antibodies.…”

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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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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“…Samples from each patient in our centre were sent to Guangzhou KingMed Diagnostics Group Co., Ltd for testing. 3 Positive results were evaluated at least in duplicate on the samples. The intensity of the surface immunofluorescence, which was directly compared to control samples and samples lacking transfected cells, was used by two independent, masked assessors to determine whether a sample was positive or negative.…”

Section: Detection Of Ae-absmentioning

confidence: 99%

“…2 Since the first study on antibody (specifically immunoglobulin-G [IgG]) biomarkers was published in 2007, autoimmune encephalitis research has rapidly advanced.Forty intracellular antibodies and more than 20 extracellular antibodies have been identified so far. 3 Recently, a large cohort study has shown a seroprevalence of 3.6% for autoimmune encephalitis antibody biomarkers (AE-Abs) in adults, with the most common AE-Abs being N-methyl-Daspartate receptor (NMDAR-IgG), glutamic acid decarboxylase 65 (GAD65-IgG), and leucine-rich glioma-inactivated 1 (LGI1-IgG). 4 Neuronal antibodies identify autoimmune encephalitis subtypes and help clinicians identify cases with…”

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confidence: 99%

“…With the recent increase in autoimmune encephalitis awareness, 9 the prevalence of antibody testing has become more common, resulting in a severe financial burden for patients. 3,10 Early clinical characterization can assist in the development of accurate antibody testing strategies. However, more research is needed to determine the clinical features in patients with autoimmune encephalitis that indicate the presence of AE-Abs.…”

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confidence: 99%

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Autoimmune encephalitis antibody profiles and clinical characteristics of children with suspected autoimmune encephalitis

Yang,

Luo,

Ding

et al. 2023

Develop Med Child Neuro

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AimTo identify the spectrum of autoimmune encephalitis antibody biomarkers (AE‐Abs) in children with suspected autoimmune encephalitis and explore the clinical features indicating AE‐Abs presence.MethodWe included children with suspected autoimmune encephalitis who underwent AE‐Abs tests at the Children's Hospital of Chongqing Medical University between June 2020 and June 2022. Clinical features suggestive of AE‐Abs were analysed based on AE‐Abs test results.ResultsA total of 392 children were tested for AE‐Abs with suspected autoimmune encephalitis. Of these, 49.5% were male, with a median age of 7 years 11 months (6 months–17 years 11 months); 93.6% (367/392) of all patients had both serum and cerebrospinal fluid (CSF) tests performed. The antibody‐positive rate in the cohort was 23.7% (93/392), the serum antibody‐positive rate was 21.9% (84/384), and the CSF antibody‐positive rate was 20.8% (78/375). Eleven different AE‐Abs were detected. Serum analysis revealed that N‐methyl‐D‐aspartate receptor immunoglobulin‐G (NMDAR‐IgG) (15.1%) was greater than myelin oligodendrocyte glycoprotein (MOG)‐IgG (14.6%) and glial fibrillary acidic protein (GFAP)‐IgG (3.3%). CSF analysis revealed that NMDAR‐IgG (16.3%) was greater than MOG‐IgG (13.8%) and GFAP‐IgG (3.3%). Compared with antibody‐negative patients, antibody‐positive patients were more often female (odds ratio [OR] 1.86, p = 0.03), with memory impairment (OR 2.91, p = 0.01) and sleep disorders (OR 2.08, p = 0.02).InterpretationIn children, the most frequent AE‐Abs detected were NMDAR‐IgG and MOG‐IgG. Female sex, memory impairment, and sleep disorders predict a higher likelihood of AE‐Abs.

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“…Since 2007, an increasing number of new specific anti-neuronal antibodies have been reported, and the study of AE has markedly expanded; in total, more than twenty extracellular antibodies and forty intracellular antibodies have been identified (2,3). The estimated prevalence of AE in the United States and China is 0.8/ 100,000 and is increasing rapidly (4,5). Previous studies have demonstrated that approximately 50% of AE patients require intensive care unit (ICU) treatment (6).…”

Section: Introductionmentioning

confidence: 99%

Performance of the clinical assessment scale for autoimmune encephalitis in a pediatric autoimmune encephalitis cohort

et al. 2022

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BackgroundThe Clinical Assessment Scale for Autoimmune Encephalitis (CASE), a new scale used for rating the severity of autoimmune encephalitis (AE), has demonstrated good validity and reliability in adults with AE, but there is a shortage of data on its performance in children with AE. This study aimed to assess the reliability and validity of the CASE in a cohort of children with AE.MethodsForty-seven pediatric inpatients with AE who visited Guizhou Provincial People’s Hospital between January 1, 2017, and October 31, 2021, were enrolled in the study. The CASE and mRS scores were obtained through a review of detailed medical records from the Health Information System by two pediatric neurologists. Finally, the performance of the CASE in this pediatric AE cohort was analyzed.ResultsThe results showed that anti-NMDA receptor encephalitis was the most common (61.70%) type of AE in children. The most common clinical manifestations were language problems (85.1%), psychiatric symptoms (80.9%), and dyskinesia/dystonia (78.7%). The CASE had good item reliability and interevaluator reliability; the Cronbach’s alpha value of the total score was 0.825, and the intraclass correlation (ICC) was 0.980. The Cronbach’s alpha value by item ranged from 0.16 to 0.406; items 1 and 9 had the lowest and highest Cronbach’s alpha values, respectively. The criterion validity between CASE and mRS total scores, as quantified by Pearson correlation, was 0.459, indicating slight to good criterion validity. The area under the curve (AUC) was 0.992 (95% confidence interval: 0.974-1.00). A cutoff value of 14 was selected to determine whether a patient needed admission to the ICU; this cutoff had a sensitivity of 100% and a specificity of 92%. The changes in EEG, MRI, and antibody titers were not related to the severity of AE. A CASE score cutoff of 9 was selected to indicate whether second-line treatment would be needed.ConclusionThe CASE has good reliability and validity in children with AE; however, some items of the CASE may not apply to this population. Thus, an in-depth study of the CASE is needed in children with AE.

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The Antibody Assay in Suspected Autoimmune Encephalitis From Positive Rate to Test Strategies

Cited by 4 publications

References 32 publications

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

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

Autoimmune encephalitis antibody profiles and clinical characteristics of children with suspected autoimmune encephalitis

Performance of the clinical assessment scale for autoimmune encephalitis in a pediatric autoimmune encephalitis cohort

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