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https://www.readbyqxmd.com/read/29774053/postherpes-simplex-encephalitis-a-case-series-of-viral-triggered-autoimmunity-synaptic-autoantibodies-and-response-to-therapy
#1
Harry Alexopoulos, Sofia Akrivou, Sotiria Mastroyanni, Maria Antonopoulou, Argirios Dinopoulos, Melpo Giorgi, Kostas Konstantinou, Evangelos Kouremenos, Maria Lariou, Dimitrios Naoumis, Efterpi Pavlidou, Evaggelos Pavlou, Konstantinos Voudris, Panayotis Vlachoyiannopoulos, Marinos C Dalakas
Background: Recent evidence suggests that patients with herpes simplex virus (HSV) encephalitis may relapse because of autoimmunity against the N-methyl-D-aspartate receptor (NMDAR). We present a case series of post-HSV relapsing encephalopathy associated with antibodies to central nervous system (CNS) synaptic antigens. Patient/Methods: Sera and cerebrospinal fluid (CSF) from five patients with HSV encephalitis who relapsed after antiviral therapy were tested for anti-NMDAR, gamma-aminobutyric acid b receptor (GABAbR), α-amino-3-hydroxy-5-methyl-4-isoxazolepropionic acid receptor (AMPAR), Leucine-rich, glioma inactivated 1 (LGI1), anti -contactin-associated protein-like 2 (CASPR2) and dipeptidyl-peptidase-like protein-6 (DDPX) antibodies using cell-based assays...
2018: Therapeutic Advances in Neurological Disorders
https://www.readbyqxmd.com/read/29771820/effect-of-lgi1-antibody-positive-igg-on-hippocampal-neuron-survival-a-preliminary-study
#2
Neşe Ayşit-Altuncu, Canan Ulusoy, Gürkan Öztürk, Erdem Tüzün
Anti-leucine-rich glioma inactivated 1 (anti-LGI1) encephalitis is one of the most frequently encountered forms of autoimmune encephalitis. Many patients with anti-LGI1 encephalitis develop permanent hippocampal neuron loss and chronic neuropsychiatric symptoms, suggesting that LGI antibodies (Ab) might have a neurotoxic action. To investigate this hypothesis, purified serum IgG of three patients with anti-LGI1 encephalitis and six healthy controls were incubated with cultured primary hippocampal neurons obtained from newborn mice...
May 15, 2018: Neuroreport
https://www.readbyqxmd.com/read/29761127/elevated-lgi1-igg-csf-index-predicts-worse-neurological-outcome
#3
Avi Gadoth, Anastasia Zekeridou, Christopher J Klein, Colton J Thoreson, Masoud Majed, Divyanshu Dubey, Eoin P Flanagan, Andrew McKeon, Sarah M Jenkins, Vanda A Lennon, Sean J Pittock
To determine whether CSF leucine-rich glioma-inactivated 1(LGI1)-IgG titer, index or IgG subclass has prognostic significance, we tested serum and CSF specimens collected concomitantly from 39 seropositive patients. LGI1-IgG index was elevated (>1) in 21 patients (54%), suggesting intrathecal synthesis. Patients with worse outcome at last follow-up (modified Rankin Scale >2) had significantly higher index (median 6.57 vs. 0.5, P  = 0.048) compared to those with better outcome. Higher CSF LGI1-IgG4 subclass-specific titer and index correlated with worse outcome ( P  < 0...
May 2018: Annals of Clinical and Translational Neurology
https://www.readbyqxmd.com/read/29756462/lgi1-antibody-encephalitis-and-psychosis
#4
Dahai Wang, Qinjian Hao, Lan He, Qiang Wang
OBJECTIVE: To describe a case of leucine-rich, glioma inactivated 1 antibody-encephalitis presenting with psychosis. METHODS: Case report. RESULTS: A young man with leucine-rich, glioma inactivated 1-antibody encephalitis initially presented with acute psychotic symptoms, short-term memory loss and faciobrachial dystonic seizures. Magnetic resonance imaging revealed hippocampal lesions. Electroencephalography revealed frontotemporal slowing of background activity...
May 1, 2018: Australasian Psychiatry: Bulletin of Royal Australian and New Zealand College of Psychiatrists
https://www.readbyqxmd.com/read/29731274/development-of-moyamoya-disease-after-non-herpetic-acute-limbic-encephalitis-a-case-report
#5
Yasuhiro Takahashi, Takeshi Mikami, Hime Suzuki, Katsuya Komatsu, Daisuke Yamamoto, Shun Shimohama, Kiyohiro Houkin, Shintaro Sugita, Tadashi Hasegawa, Nobuhiro Mikuni
We report a case of moyamoya disease (MMD), which developed after non-herpetic acute limbic encephalitis (NHALE) associated with anti-leucine-rich glioma-inactivated 1 (LGI1) antibody. The patient's mother had a history of MMD. No vascular lesions were identified at the time of the NHALE. Nine years later, the patient visited our hospital due to memory disturbances and repeated transient ischemic attacks affecting the right limb. Diffusion-weighted magnetic resonance imaging revealed scattered areas of signal hyperintensity, and the patient was ultimately diagnosed with MMD based on angiography...
May 3, 2018: Journal of Clinical Neuroscience: Official Journal of the Neurosurgical Society of Australasia
https://www.readbyqxmd.com/read/29723732/lgi1-tumor-tissue-expression-and-serum-autoantibodies-in-patients-with-primary-malignant-glioma
#6
Emanuela Dazzo, Elena Pasini, Sandra Furlan, Dario de Biase, Matteo Martinoni, Roberto Michelucci, Carlo Nobile
OBJECTIVES: The Leucine-rich glioma inactivated 1 (LGI1) protein is thought to be implicated in malignant progression of glioma tumors, and mutations in the encoding gene, LGI1, cause autosomal dominant lateral temporal epilepsy, a genetic focal epilepsy syndrome. The aim of this study was to investigate the possible involvement of LGI1 in high-grade glioma-associated epilepsy by analyzing its expression in tumor specimens of patients with and without epilepsy and by searching for LGI1 autoantibodies in the sera these patients...
April 14, 2018: Clinical Neurology and Neurosurgery
https://www.readbyqxmd.com/read/29670100/structural-basis-of-epilepsy-related-ligand-receptor-complex-lgi1-adam22
#7
Atsushi Yamagata, Yuri Miyazaki, Norihiko Yokoi, Hideki Shigematsu, Yusuke Sato, Sakurako Goto-Ito, Asami Maeda, Teppei Goto, Makoto Sanbo, Masumi Hirabayashi, Mikako Shirouzu, Yuko Fukata, Masaki Fukata, Shuya Fukai
Epilepsy is a common brain disorder throughout history. Epilepsy-related ligand-receptor complex, LGI1-ADAM22, regulates synaptic transmission and has emerged as a determinant of brain excitability, as their mutations and acquired LGI1 autoantibodies cause epileptic disorders in human. Here, we report the crystal structure of human LGI1-ADAM22 complex, revealing a 2:2 heterotetrameric assembly. The hydrophobic pocket of the C-terminal epitempin-repeat (EPTP) domain of LGI1 binds to the metalloprotease-like domain of ADAM22...
April 18, 2018: Nature Communications
https://www.readbyqxmd.com/read/29620086/drop-attacks-a-clinical-manifestation-of-lgi1-encephalitis
#8
Ana Vives-Rodriguez, Adithya Sivaraju, Elan D Louis
No abstract text is available yet for this article.
October 2017: Neurology. Clinical Practice
https://www.readbyqxmd.com/read/29572931/genetic-predisposition-in-anti-lgi1-and-anti-nmda-receptor-encephalitis
#9
Stefanie H Mueller, Anna Färber, Harald Prüss, Nico Melzer, Kristin S Golombeck, Tania Kümpfel, Franziska Thaler, Martin Elisak, Jan Lewerenz, Max Kaufmann, Kurt-Wolfram Sühs, Marius Ringelstein, Christoph Kellinghaus, Christian G Bien, Andrea Kraft, Uwe K Zettl, Sven Ehrlich, Robert Handreka, Kevin Rostásy, Florian Then Bergh, Jürgen H Faiss, Wolfgang Lieb, Andre Franke, Gregor Kuhlenbäumer, Klaus-Peter Wandinger, Frank Leypoldt
We performed a genome-wide-association study in 1,194 controls and 150 patients with anti-N-methyl-D-aspartate receptor (anti-NMDAR, n=96) or anti-leucine-rich glioma-inactivated1 (anti-LGI1, n=54) autoimmune encephalitis. Anti-LGI1 encephalitis was highly associated with 27 SNPs in the HLA-II region (leading SNP rs2858870 P=1.22x10-17 , OR=13.66 [7.50-24.87]). Potential associations, below genome-wide significance, were found with rs72961463 close to the doublecortin-like kinase 2 gene (DCLK2) and rs62110161 in a cluster of zink-finger genes...
March 23, 2018: Annals of Neurology
https://www.readbyqxmd.com/read/29561731/paraneoplastic-movement-disorders
#10
Karolina Popławska-Domaszewicz, Jolanta Florczak-Wyspiańska, Wojciech Kozubski, Sławomir Michalak
Paraneoplastic movement disorders are rare, autoimmune-mediated, nonmetastatic complications of malignant neoplasms. Common paraneoplastic movement disorders include paraneoplastic chorea, dystonia, cerebellar degeneration, different types of encephalitis, opsoclonus-myoclonus syndrome, stiff person syndrome, and neuromyotonia. Syndromes usually develop before tumor diagnosis, have subacute onset, and are associated with serum or cerebrospinal fluid antibodies. Two types of antibodies can be distinguished: antibodies against nuclear and cytoplasmic neuronal antigens (anti-Hu, anti-Ri, anti-Yo, anti-Ma, anti-CV2/CRMP5, anti-Gephrin, and anti-GABATRAP) and antibodies recently identified against cell surface and synaptic proteins (anti-NMDAR, anti-LGI1, and anti-Caspr2)...
March 21, 2018: Reviews in the Neurosciences
https://www.readbyqxmd.com/read/29520216/a-nogo-like-signaling-perspective-from-birth-to-adulthood-and-in-old-age-brain-expression-patterns-of-ligands-receptors-and-modulators
#11
Gabriella Smedfors, Lars Olson, Tobias E Karlsson
An appropriate strength of Nogo-like signaling is important to maintain synaptic homeostasis in the CNS. Disturbances have been associated with schizophrenia, MS and other diseases. Blocking Nogo-like signaling may improve recovery after spinal cord injury, stroke and traumatic brain injury. To understand the interacting roles of an increasing number of ligands, receptors and modulators engaged in Nogo-like signaling, the transcriptional activity of these genes in the same brain areas from birth to old age in the normal brain is needed...
2018: Frontiers in Molecular Neuroscience
https://www.readbyqxmd.com/read/29501086/psychiatric-symptoms-delay-the-diagnosis-of-anti-lgi1-encephalitis
#12
Yoonhyuk Jang, Soon-Tae Lee, Jung-Ah Lim, Tae-Joon Kim, Jin-Sun Jun, Jangsup Moon, Jun-Sang Sunwoo, Keun-Hwa Jung, Kyung-Il Park, Ki-Young Jung, Manho Kim, Sang Kun Lee, Kon Chu
The aim of this study was to analyze the detailed characteristics of the psychiatric symptoms in patients with anti-LGI1 encephalitis. Of 16 patients, ten showed psychiatric symptoms as the initial manifestations. All 10 patients experienced mood-related symptoms. The time to immune therapy was longer in those with initial psychiatric symptoms compared to those without them. Initial manifestation of psychiatric symptoms in patients with anti-LGI1 encephalitis may be a poor prognostic factor, at least in the short term, in that it misleads both the patients and the clinicians to neglect the typically accompanied symptoms of the disease such as faciobrachial dystonic seizure, delaying the timing of immune therapy...
April 15, 2018: Journal of Neuroimmunology
https://www.readbyqxmd.com/read/29491011/celecoxib-ameliorates-seizure-susceptibility-in-autosomal-dominant-lateral-temporal-epilepsy
#13
Lin Zhou, Liang Zhou, Li-da Su, Sheng-Long Cao, Ya-Jun Xie, Na Wang, Chong-Yu Shao, Ya-Nan Wang, Jia-Huan Zhou, John K Cowell, Ying Shen
Autosomal dominant lateral temporal epilepsy (ADLTE) is an inherited syndrome caused by mutations in the leucine-rich glioma inactivated 1 (LGI1) gene. It is known that glutamatergic transmission is altered in LGI1 mutant mice, and seizures can be reduced by restoring LGI1 function. Yet, the mechanism underlying ADLTE is unclear. Here, we propose that seizures in male LGI1 -/- mice are due to nonsynaptic epileptiform activity in cortical neurons. We examined the intrinsic excitability of pyramidal neurons in the temporal cortex of male LGI1 -/- mice and found that the voltage-gated K+ channel Kv1...
March 28, 2018: Journal of Neuroscience: the Official Journal of the Society for Neuroscience
https://www.readbyqxmd.com/read/29406902/autoimmune-and-paraneoplastic-movement-disorders-an-update
#14
REVIEW
José Fidel Baizabal-Carvallo, Joseph Jankovic
Movement disorders (MDs) are common in patients with autoimmune disorders affecting the central and peripheral nervous system. They may be observed in autoimmune disorders triggered by an infectious agent, such as streptococcus in Sydenham's chorea, or in basal ganglia encephalitis with antibodies against the dopamine-D2 receptors. In these patients chorea or dystonia are usually the most prominent hyperkinetic MDs. MDs are also observed in patients with diffuse or limbic encephalitis with antibodies directed against neuronal cell-surface antigens...
February 15, 2018: Journal of the Neurological Sciences
https://www.readbyqxmd.com/read/29395322/thymoma-associated-myasthenia-gravis-and-lgi1-encephalitis-with-nephrotic-syndrome-post-thymectomy
#15
Jyh Yung Hor, Thien Thien Lim, Mei Chih Cheng, Yuen Kang Chia, Chee Keong Wong, Su Min Lim, Chun Fai Cheah, Kenny Tan, P E Samuel Easaw, M Isabel Leite
Thymoma is associated with a wide spectrum of autoimmune paraneoplastic syndromes, though it is uncommon for multiple paraneoplastic syndromes to be present in a single individual. We report a rare case of an elderly gentleman who was found to have thymoma-associated myasthenia gravis and LGI1-encephalitis with myokymia, who presented with nephrotic syndrome (minimal change glomerulopathy) after thymectomy. The latter two paraneoplastic syndromes had manifested when prednisolone was tapered down to low dose...
April 15, 2018: Journal of Neuroimmunology
https://www.readbyqxmd.com/read/29394500/mutations-in-mical-1cause-autosomal-dominant-lateral-temporal-epilepsy
#16
Emanuela Dazzo, Kati Rehberg, Roberto Michelucci, Daniela Passarelli, Clementina Boniver, Valeria Vianello Dri, Pasquale Striano, Salvatore Striano, R Jeroen Pasterkamp, Carlo Nobile
OBJECTIVE: Autosomal-dominant lateral temporal epilepsy (ADLTE) is a genetic focal epilepsy characterized by auditory symptoms. Two genes, LGI1 and RELN, encoding secreted proteins, are implicated in the etiology of ADLTE, but half of the affected families remain genetically unsolved, and the underlying molecular mechanisms are yet to be clarified. We aimed to identify additional genes causing ADLTE to better understand the genetic basis and molecular pathway underlying this epileptic disorder...
March 2018: Annals of Neurology
https://www.readbyqxmd.com/read/29332416/-autoimmune-encephalitis-possibilities-in-the-laboratory-investigation
#17
Katalin Böröcz, Zsófia Hayden, Viktória Mészáros, Zsuzsanna Csizmadia, Kornélia Farkas, Zoltán Kellermayer, Péter Balogh, Ferenc Nagy, Tímea Berki
INTRODUCTION: The role of autoimmune responses against central nervous system (CNS) antigens in encephalitis presenting with non-classified neurologic or psychiatric symptoms has been appreciated in the past decade. Paraneoplastic limbic encephalitis has a poor prognosis and is most commonly associated with lung, ovarium, and testicular neoplasms, leading to immune reactions against intracellular antigens (anti-Hu/ANNA1, anti-Ri/ANNA2, anti-CV2/CRMP5 and anti-Ma2/Ta). In contrast, the recently described autoimmune encephalitis subtypes present with a broad spectrum of symptoms, respond to autoimmune therapies well and usually associate with autoantibodies against neuronal cell surface receptors (NMDAR, GABAB R, AMPAR) or synaptic proteins (LGI1, CASPR2)...
January 2018: Orvosi Hetilap
https://www.readbyqxmd.com/read/29325310/-clinical-characteristics-and-whole-exon-sequence-study-of-a-chinese-family-with-autosomal-dominant-lateral-temporal-lobe-epilepsy
#18
C Y Li, H H Yang, R J Lyu, Q Wang
Objective: To explore the genetic characteristics in a Chinese family with autosomal dominant lateral temporal lobe epilepsy (ADLTE) and analyze the correlation between genotype and phenotype. Methods: The natural history, clinical data and peripheral blood sample were collected in all patients and two healthy members of this ADLTE family. Whole exon sequence (WES) analysis strategy was used to explore the underlying mutations. Possible causative genetic variation was further confirmed by direct PCR and Sanger sequencing...
January 1, 2018: Zhonghua Nei Ke za Zhi [Chinese Journal of Internal Medicine]
https://www.readbyqxmd.com/read/29318331/-severe-hyponatremia-as-precursor-of-lgi1-autoimmune-encephalitis
#19
P Muhr, U Goldammer, C G Bien, C Bien, E Sindern
No abstract text is available yet for this article.
January 9, 2018: Der Nervenarzt
https://www.readbyqxmd.com/read/29237452/molecular-cloning-and-characterization-of-the-family-of-feline-leucine-rich-glioma-inactivated-lgi-genes-and-mutational-analysis-in-familial-spontaneous-epileptic-cats
#20
Yoshihiko Yu, Daisuke Hasegawa, Aki Fujiwara-Igarashi, Yuji Hamamoto, Shunta Mizoguchi, Takayuki Kuwabara, Michio Fujita
BACKGROUND: Leucine-rich glioma-inactivated (LGI) proteins play a critical role in synaptic transmission. Dysfunction of these genes and encoded proteins is associated with neurological disorders such as genetic epilepsy or autoimmune limbic encephalitis in animals and human. Familial spontaneous epileptic cats (FSECs) are the only feline strain and animal model of familial temporal lobe epilepsy. The seizure semiology of FSECs comprises recurrent limbic seizures with or without evolution into generalized epileptic seizures, while cats with antibodies against voltage-gated potassium channel complexed/LGI1 show limbic encephalitis and recurrent limbic seizures...
December 13, 2017: BMC Veterinary Research
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