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REVIEW
Giovanna Scorrano, Fedele Dono, Clarissa Corniello, Stefano Consoli, Giacomo Evangelista, Armando Di Ludovico, Francesco Chiarelli, Francesca Anzellotti, Angelo Di Iorio, Stefano L Sensi
INTRODUCTION: Anti-N-methyl-D-aspartate receptor (NMDAr) antibody encephalitis is an autoimmune disorder characterized by synaptic NMDAr current disruption and receptor hypofunction, often affecting women during pregnancy. Clinical manifestations associated with anti-NMDAr encephalitis can occur both in the mother and fetus. METHODS: We generated a systematic search of the literature to identify epidemiological, clinical, and serological data related to pregnant women with anti-NMDAr encephalitis and their children, analyzing the fetal outcomes...
April 24, 2024: Neurological Sciences
#2
JOURNAL ARTICLE
Li Li, Hongmei Liao, Xiaojun Kuang, Ke Jin
Apart from the typical respiratory symptoms, coronavirus disease 2019 (COVID-19) also affects the central nervous system, leading to central disorders such as encephalopathy and encephalitis. However, knowledge of pediatric COVID-19-associated encephalopathy is limited, particularly regarding specific subtypes of encephalopathy. This study aimed to assess the features of COVID-19-associated encephalopathy/encephalitis in children. We retrospectively analyzed a single cohort of 13 hospitalized children with COVID-19-associated encephalopathy...
April 3, 2024: Journal of Neurovirology
#3
JOURNAL ARTICLE
Trevor Rafferty, Anna Koeppen-Babcock, Srikanth Muppidi, Yi Li, Scheherazade Le
PURPOSE: Immune-mediated seizures are rare but are increasingly recognized as an etiology of seizures resistant to anti-seizure medications (ASMs). Antibody Prevalence in Epilepsy 2 (APE2) and Response to Immunotherapy in Epilepsy 2 (RITE2) scores were developed recently to identify patients who may be seropositive for serum central nervous system (CNS) specific antibodies (Ab) and may benefit from immunotherapy (Dubey et al. 2018). The goal of this study was to apply APE2 and RITE2 scores to an independent cohort of patients with seizures secondary to autoimmune encephalitis (AE) and to further verify the sensitivity and specificity of the scores...
March 21, 2024: Epilepsy & Behavior: E&B
#4
REVIEW
Barbara Wagner, Sarosh Irani
Seizures are a common feature of autoimmune encephalitis and are especially prevalent in patients with the commonest autoantibodies, against LGI1, CASPR2 and the NMDA, GABAB , and GABAA receptors. In this chapter, we discuss the classification, clinical, investigation, and treatment aspects of patients with these, and other autoantibody-mediated and -associated, illnesses. We highlight distinctive and common seizure semiologies which, often alongside other features we outline, can help the clinical diagnosis of an autoantibody-associated syndrome...
2024: Handbook of Clinical Neurology
#5
JOURNAL ARTICLE
Wei Peng, Minjin Wang, Wenyan Shi, Jierui Wang, Dong Zhou, Jinmei Li
BACKGROUND: The identification of patients with seizures of unknown etiology who would benefit from neural antibody testing necessitates effective assessment tools. The study aimed to compare the performance of the Antibody Prevalence in Epilepsy and Encephalopathy (APE2) score and the "Obvious" Indications for Neural Antibody Testing in Epilepsy or Seizures (ONES) checklist. We also intended to evaluate whether the performance of the tools varied by types of antibody. METHODS: Patients diagnosed with epilepsy, seizures, or status epilepticus of unknown etiology at West China Hospital from January 2019 to December 2021 were included...
February 26, 2024: International Immunopharmacology
#6
JOURNAL ARTICLE
Lin Bai, Weiying Di, Zucai Xu, Bin Liu, Nan Lin, Siyuan Fan, Haitao Ren, Qiang Lu, Jiawei Wang, Hongzhi Guan
OBJECTIVE: To summarize the clinical characteristics and prognosis of febrile infection-related epilepsy syndrome with claustrum lesions (FIRES-C). METHOD: Clinical data of FIRES-C patients were collected retrospectively. The study reviewed and analyzed their clinical manifestations, treatment strategies, and prognosis. RESULT: Twenty patients were enrolled, including 13 females and 7 males, with a median onset age of 20.5 years. All patients developed seizures after fever, with a median interval of 5 days...
February 12, 2024: Neurological Sciences
#7
JOURNAL ARTICLE
Friederike A Arlt, Ramona Miske, Marie-Luise Machule, Peter Broegger Christensen, Swantje Mindorf, Bianca Teegen, Kathrin Borowski, Maria Buthut, Rosa Rößling, Elisa Sanchez-Sendin, Scott van Hoof, César Cordero-Gómez, Isabel Bünger, Helena Radbruch, Andrea Kraft, Ilya Ayzenberg, Jaqueline Klausewitz, Niels Hansen, Charles Timäus, Peter Körtvelyessy, Thomas Postert, Kirsten Baur-Seack, Constanze Rost, Robert Brunkhorst, Kathrin Doppler, Niklas Haigis, Gerhard Hamann, Albrecht Kunze, Alexandra Stützer, Matthias Maschke, Nico Melzer, Felix Rosenow, Kai Siebenbrodt, Christian Stenør, Martin Dichgans, Marios K Georgakis, Rong Fang, Gabor C Petzold, Michael Görtler, Inga Zerr, Silke Wunderlich, Ivan Mihaljevic, Paul Turko, Marianne Schmidt Ettrup, Emilie Buchholz, Helle Foverskov Rasmussen, Mahoor Nasouti, Ivan Talucci, Hans M Maric, Stefan H Heinemann, Matthias Endres, Lars Komorowski, Harald Prüss
BACKGROUND: Autoantibodies against the potassium voltage-gated channel subfamily A member 2 (KCNA2) have been described in a few cases of neuropsychiatric disorders, but their diagnostic and pathophysiological role is currently unknown, imposing challenges to medical practice. DESIGN / METHODS: We retrospectively collected comprehensive clinical and paraclinical data of 35 patients with KCNA2 IgG autoantibodies detected in cell-based and tissue-based assays. Patients' sera and cerebrospinal fluid (CSF) were used for characterization of the antigen, clinical-serological correlations, and determination of IgG subclasses...
February 1, 2024: Brain, Behavior, and Immunity
#8
Garrett Friedman, James Hammock, Niki Holtzman-Hayes, Lauren Gluck
Anti-NMDA Receptor (NMDAR) Encephalitis (NMDARE) is an autoimmune disorder that is often debilitating and difficult to diagnose. Patients, especially those with underlying neuropsychiatric disorders, may experience delayed or misdiagnosis of NMDARE. Here, we report on a patient with known congenital leukodystrophy (CLD) and epilepsy with a challenging diagnosis of NMDARE. The patient first presented with progressive behavior changes and seizure-like episodes. Initial workup, including video EEG and brain MRI, were mostly unremarkable, and the patient's symptoms were resistant to treatment with multiple anti-epileptic drugs...
January 2024: Neurohospitalist
#9
REVIEW
Tamang Sapana, Wei Li, Fengyan Tian, Wenhao Yan, Binghua Dou, Shuang Hua, Zhihong Zhuo
BACKGROUND: Glutamic acid decarboxylase (GAD) is the rate-limiting enzyme for the synthesis of gamma-aminobutyric acid (GABA), the major inhibitory neurotransmitter in the central nervous system. Antibodies against glutamic acid decarboxylase (GAD) are associated with various neurologic conditions described in patients, including stiff person syndrome, cerebellar ataxia, refractory epilepsy, and limbic and extra limbic encephalitis. While there are few case reports and research on anti-GAD65 antibody-associated encephalitis in adults, such cases are extremely rare in pediatric cases...
2023: Frontiers in Immunology
#10
JOURNAL ARTICLE
Motoki Inaji, Taketoshi Maehara
Status epilepticus(SE)is defined as a prolonged seizure and is a common neurological emergency with high morbidity and mortality rates. As uncontrolled SE causes irreversible neurological damage, prompt diagnosis and treatment are required. If anti-seizure medications and benzodiazepines, which are initial treatments for SE, are not effective and SE deteriorates to refractory, anesthetic drugs are needed to suppress seizure activity under electroencephalogram(EEG)monitoring. Continuous EEG monitoring is useful not only for evaluating the control of SE but also for diagnosing non-convulsive SE(NCSE)and psychogenic non-epileptic seizures...
November 2023: No Shinkei Geka. Neurological Surgery
#11
JOURNAL ARTICLE
Si-Qi Xia, Hao-Nan Fan, Lin-Feng Fan, Wu Xia, Gao Chen
RATIONALE: Autoimmunity targeting glutamic acid decarboxylase 65 (GAD65) is associated with type 1 diabetes mellitus as well as various neurological diseases. In the central nervous system, GAD65 autoimmunity usually presents with limbic encephalitis, whereas extralimbic encephalitis (ELE) has only been reported in a few cases. Moreover, anti-GAD65 ELE in the paraneoplastic context has not yet been reported. PATIENT CONCERNS: A 60-year-old man presented with intermittent cough and sputum for 10 years, with no other diseases...
November 3, 2023: Medicine (Baltimore)
#12
JOURNAL ARTICLE
M J Zhang, L Lin, W H Wang, W H Li, C J Wei, H Xie, Q P Zhang, Y Wu, H Xiong, S Z Zhou, B Yang, X H Bao
Objective: To explore the clinical and imaging features of acute encephalopathy with biphasic seizures and late reduced diffusion(AESD) in children. Methods: For the case series study, 21 children with AESD from Peking University First Hospital, Provincial Children's Hospital Affiliated to Anhui Medical University, Children 's Hospital of Fudan University, and Shanxi Children's Hospital who were diagnosed and treated from October 2021 to July 2023 were selected. Clinical data were collected to summarize their clinical information, imaging, and laboratory tests, as well as treatment and prognostic characteristics...
November 2, 2023: Zhonghua Er Ke za Zhi. Chinese Journal of Pediatrics
#13
JOURNAL ARTICLE
Zhang Kai-Jing, Lv Xiao-Juan, Huang Xiao-Hui
In young women with anti-N-methyl-D-aspartate receptor (anti-NMDAR) autoimmune encephalitis (AE), co-occurrence with ovarian teratoma is common. While the management of mature teratoma with AE is well documented, literature on managing immature teratoma (IT) in tandem with AE is relatively scarce. Here, we report a case of a female patient in her early adolescence who presented with abdominal pain and was diagnosed with grade 3 IT combined with anti-NMDAR AE after an ovarian tumour was discovered and resected...
October 29, 2023: BMJ Case Reports
#14
Shaikha Alsuwaidi, Hamda Kamalboor, Tarig Khairi, Abubaker Almadani
Anti-Ma2 encephalitis is an autoimmune disorder that typically involves the brainstem, limbic system, and diencephalon. It can be paraneoplastic and is more common in males. We describe an unusual presentation of anti-Ma2 encephalitis in a patient with an XY chromosome and a female phenotype. She experienced various neurological symptoms, including olfactory hallucinations, episodic nausea, per-ictal water drinking, and hypersomnolence, that were poorly controlled by antiseizure medications (ASMs) and immunotherapy...
2023: Epilepsy & behavior reports
#15
JOURNAL ARTICLE
Abrar A Khan, Waleed M Alzahrani, Reem M Bagadood, Baker M Alweshah, Jwana F Alsulaimi, Hanadi M Abualela
OBJECTIVES: To evaluate the clinical and electrographic features of patients with autoimmune epilepsy and assess the influence of early diagnosis and treatment on reducing seizure frequency. METHODS: A retrospective observational case series was conducted utilizing medical records from King Abdullah Medical Hospital between 2017 and 2022. Cases of newly diagnosed seizures were chosen based on laboratory-proven autoimmunity. RESULTS: Five female inpatients were identified, primarily presenting with seizures suggestive of an autoimmune origin...
October 2023: Neurosciences: the Official Journal of the Pan Arab Union of Neurological Sciences
#16
REVIEW
Milena M Andzelm, Coral M Stredny
There has been increasing understanding of the role of inflammation in seizures and epilepsy, as well as targeted immunomodulatory treatments. In children, immune-mediated seizures often present acutely in the setting of autoimmune encephalitis and are very responsive to immunotherapy with low rates of subsequent epilepsy. Conversely, seizures in autoimmune-associated epilepsies, such as Rasmussen syndrome, can remain refractory to multimodal therapy, including immunomodulation. In this review, the authors discuss the presentations of immune-mediated seizures in children, underlying mechanisms, and emerging therapies...
November 2023: Rheumatic Diseases Clinics of North America
#17
JOURNAL ARTICLE
Spencer R Osbourn, Keith E Ryan, Kevin R Cannard, Yitao Ma
New onset refractory status epilepticus (NORSE) is a rare but critical condition characterized by refractory status epilepticus (RSE) in an individual without prior history of epilepsy or known structural, toxic, or metabolic cause. Postinfectious immune activation is an important cause of NORSE. Early testing for autoimmune antibodies is strongly recommended (Wickstrom et al., 2022). We report a case of NORSE triggered by Japanese encephalitis (JE) in an unvaccinated US adult traveler. Her CSF later revealed positive anti-N-methyl-d-aspartate (NMDA)-receptor antibody...
October 15, 2023: Journal of Neuroimmunology
#18
Lu Wang, Hong-Jun Su, Guan-Jie Song
BACKGROUND: Anti-N-methyl-D-aspartate receptor (anti-NMDAR) encephalitis is a rare autoimmune disorder. The symptoms of anti-NMDAR encephalitis include behavioral problems, speech problems, psychosis, seizures, and memory deficits, among others. However, laryngospasm is rare. We present the case of a patient with anti-NMDAR antibodies and severe laryngospasms. CASE SUMMARY: The patient was a 15-year-old female with normal psychomotor development. She was initially admitted to our neurological intensive care unit with seizures...
July 16, 2023: World Journal of Clinical Cases
#19
JOURNAL ARTICLE
Mimics of Autoimmune Encephalitis: Validation of the 2016 Clinical Autoimmune Encephalitis Criteria.
Robin W Van Steenhoven, Juna M de Vries, Arlette L Bruijstens, Manuela Paunovic, Mariska M Nagtzaam, Suzanne C Franken, Anna E Bastiaansen, Marienke A De Bruijn, Agnes Van Sonderen, Marco W J Schreurs, Mayke Gardeniers, Robert M Verdijk, Rutger K Balvers, Peter A Sillevis Smitt, Rinze F Neuteboom, Maarten J Titulaer
BACKGROUND AND OBJECTIVES: The clinical criteria for autoimmune encephalitis (AE) were proposed by Graus et al. in 2016. In this study, the AE criteria were validated in the real world, and common AE mimics were described. In addition, criteria for probable anti-LGI1 encephalitis were proposed and validated. METHODS: In this retrospective cohort study, patients referred to our national referral center with suspicion of AE and specific neuroinflammatory disorders with similar clinical presentations were included from July 2016 to December 2019...
November 2023: Neurology® Neuroimmunology & Neuroinflammation
#20
JOURNAL ARTICLE
Yan-Ting Lu, Chih-Hsiang Lin, Chen-Jui Ho, Shih-Ying Chen, Meng-Han Tsai
Autoimmune encephalitis (AE) is a neurological emergency. We aimed to analyze the application and effectiveness of the currently available prediction tools for AE patients in Taiwan. We retrospectively collected 27 AE patients between January 2008 and December 2019. Antibody Prevalence in Epilepsy (APE) score, Response to Immunotherapy in Epilepsy (RITE) score, and anti-NMDAR Encephalitis One Year Functional Status (NEOS) score were applied to validate their usability. Based on the defined cutoff values, the sensitivity and specificity of each score were calculated...
July 5, 2023: Biomedicines
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