Autoimmune encephalitis and CSF anti-AMPA GluR3 antibodies in childhood: a case report and literature review

Cucuzza, Maria Elena; Pavone, Piero; D'Ambra, Angela; Finocchiaro, Maria Carla; Greco, Filippo; Smilari, Pierluigi; Ruggieri, Martino; Stefano, Vita Antonella Di

doi:10.1007/s10072-022-06170-0

Cited by 7 publications

(6 citation statements)

References 16 publications

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“…Autoimmune encephalitis (AE) causes severe neurologic symptoms in patients of all ages, and advances in AE diagnosis have led to the identification of more antibodies causing brain inflammation, so antibodies to MOG and NMDAR have been reported with increasing frequency in AE ( 24 ). In this study, the incidence of anti-NMDAR antibody positivity was 8.5%, which is similar to that reported in previous studies ( 25 ), and none of them met the diagnostic criteria for anti-NMDAR AE.…”

Section: Discussionmentioning

confidence: 99%

Clinical features of patients with MOG-IgG associated disorders and analysis of the relationship between fibrinogen-to-albumin ratio and the severity at disease onset

Wang

et al. 2023

Front. Neurol.

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ObjectiveThe study aimed to investigate the differences in clinical features between pediatric and adult patients with first-episode MOG-IgG associated disorders (MOGAD) and evaluate the relationship between the fibrinogen-to-albumin ratio (FAR) and the severity of neurological deficits at disease onset.MethodsWe retrospectively collected and analyzed biochemical test results, imaging characteristics, clinical manifestations, expanded disability status scale (EDSS) score, and FAR. The Spearman correlation analysis and logistic regression models were used to examine the association between FAR and severity. Receiver operating characteristic (ROC) curve analysis was to analyze the predictive ability of FAR for the severity of neurological deficits.ResultsFever (50.0%), headache (36.1%), and blurred vision (27.8%) were the most common clinical manifestations in the pediatric group (<18 years old). However, in the adult group (≥18 years old), the most common symptoms were blurred vision (45.7%), paralysis (37.0%), and paresthesia (32.6%). Fever was more common in the pediatric group, while paresthesia was more common in the adult patients, with all differences statistically significant (P < 0.05). The most frequent clinical phenotype in the pediatric group was acute disseminated encephalomyelitis (ADEM; 41.7%), whereas optic neuritis (ON; 32.6%) and transverse myelitis (TM; 26.1%) were more common in the adult group. The differences in clinical phenotype between the two groups were statistically significant (P < 0.05). In both pediatric and adult patients, cortical/subcortical and brainstem lesions were the most common lesions on cranial magnetic resonance imaging (MRI), whereas, for spinal MRI, cervical and thoracic spinal cord lesions were the most commonly observed. According to binary logistic regression analysis, FAR was an independent risk factor for the severity of neurological deficits (odds ratio = 1.717; 95% confidence interval = 1.191–2.477; P = 0.004). FAR (r = 0.359, P = 0.001) was positively correlated with the initial EDSS score. The area under the ROC curve was 0.749.ConclusionThe current study found age-dependent phenotypes in MOGAD patients as ADEM was more commonly observed in patients < 18 years old, while ON and TM were more frequently found in patients ≥18 years old. A high FAR level was an independent indicator for more severe neurological deficits at disease onset in patients with a first episode of MOGAD.

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Section: Discussionmentioning

confidence: 99%

Clinical features of patients with MOG-IgG associated disorders and analysis of the relationship between fibrinogen-to-albumin ratio and the severity at disease onset

Wang

et al. 2023

Front. Neurol.

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“…Examples include glutamic acid decarboxylase 65 (GAD65) and myelin oligodendrocyte glycoprotein (MOG); even more rarely reported autoantibodies include glycine receptor and GABA A receptor [17]. A number of further rarer antibodies have recently been identified in paediatric encephalitis as well [17,18].…”

Section: Autoimmune Encephalitismentioning

confidence: 99%

Neuroinflammatory syndromes in children

Hauptman

Ferrafiat

2023

Current Opinion in Psychiatry

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Purpose of reviewNeuropsychiatric symptoms due to paediatric neuroinflammatory diseases are increasingly recognized and reported. Psychiatrists are crucial in front-lines identification, diagnosis and care of individuals with disorders such as autoimmune encephalitis and management of long-term neurobehavioral sequelae. This review summarizes recent literature on autoimmune and post-infectious encephalitis, discusses special considerations in children with neurodevelopmental conditions and presents a paradigm for evaluation and management. Recent findingsThere is a growing body of evidence on neuropsychiatric symptom burdens of paediatric neuroinflammatory diseases. A particular development is the evolution of diagnostic and treatment guidelines for conditions such as autoimmune encephalitis, which take into account phenotypes of acute, short-term and long-term sequelae. Interest in inflammatory sequelae of viral illness, such as SARS-CoV-2, in children remains in early development. SummaryNeuroimmunological disease data are constantly evolving. New recommendations exist for multiple common neuroimmunological disorders with behavioural, emotional, cognitive and neurological sequelae. Anti-NMDA receptor encephalitis now has well-recognized patterns of symptom semiology, diagnostic and treatment recommendations, and outcome patterns. Recognizing psychiatric symptoms heralding autoimmune brain disease and understanding neuropsychiatric sequelae are now a crucial skill set for paediatric psychiatrists. Exploration of inflammatory features of other diseases, such as genetic syndromes, is a burgeoning research area.

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“…13 Also, antigen-specific memory B cell clones are capable to activate memory T cells with encephalitogenic features in subjects with RRMS ex vivo, suggesting that disease-driving cells mainly belong to the memory B cell subtype. 14 Therefore, a personalized retreatment depending on levels of circulating memory B cells may be sufficient to control disease activity. This strategy has been similarly suggested for the treatment of neuromyelitis optica with RTX.…”

Section: Introductionmentioning

confidence: 99%

Memory B cell–guided extended interval dosing of ocrelizumab in multiple sclerosis

Meng,

Sacco,

Disanto

et al. 2024

Mult Scler

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Background: Ocrelizumab (OCR) is an anti-CD20 monoclonal antibody approved for the treatment of relapsing-remitting and primary-progressive multiple sclerosis (MS). We aimed to evaluate the effectiveness of an individualized OCR extended interval dosing (EID), after switching from standard interval dosing (SID). Methods: This was a retrospective, observational, single-centre study including MS patients regularly followed at the Neurocenter of Southern Switzerland. After a cumulative OCR dose ⩾1200 mg, stable patients were switched to EID (OCR infusions following CD19+ 27+ memory B cell repopulation). Results: A total of 128 patients were included in the study, and 113 (88.3%) were switched to EID with a median interval of 9.9 (8.8–11.8) months between infusions. No clinical relapses occurred; 2 (1.8%) patients experienced disability worsening. Three (2.7%) and 2 (1.8%) patients experienced new T2 brain and spinal lesions, respectively. There was a mild decrease in IgG and IgM concentrations during both SID and EID OCR regimens (β = −0.23, p = 0.001 and β = −0.07, p < 0.001, respectively). Conclusion: Switch to personalized dosing of OCR based on CD19+ 27+ memory B cell repopulation led to a great extension of the interval between infusions, with maintained clinical and radiological efficacy. Given the potential advantages in terms of safety and health costs, EID OCR regimens should be further investigated.

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Autoimmune encephalitis and CSF anti-AMPA GluR3 antibodies in childhood: a case report and literature review

Cited by 7 publications

References 16 publications

Clinical features of patients with MOG-IgG associated disorders and analysis of the relationship between fibrinogen-to-albumin ratio and the severity at disease onset

Clinical features of patients with MOG-IgG associated disorders and analysis of the relationship between fibrinogen-to-albumin ratio and the severity at disease onset

Neuroinflammatory syndromes in children

Memory B cell–guided extended interval dosing of ocrelizumab in multiple sclerosis

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