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Lennox Gastaut Syndrome

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What has been the impact of new drug treatments on epilepsy?

Zhibin Chen, Martin J Brodie, Patrick Kwan,

PURPOSE OF REVIEW:Nearly two dozen antiseizure medications (ASMs) with different mechanisms of action have been introduced over the past three decades with the aim of providing better efficacy or safety profile than the previous drugs. Several new ASMs with improvement on a classic drug family or have novel mechanisms of ... Read more >>

Curr. Opin. Neurol. (Current opinion in neurology)
[2020, 33(2):185-190]

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EEF1A2 mutations in epileptic encephalopathy/intellectual disability: Understanding the potential mechanism of phenotypic variation.

Kexin Long, Hua Wang, Zhanyi Song, Xiaomeng Yin, Yaqin Wang,

EEF1A2 encodes protein elongation factor 1-alpha 2, which is involved in Guanosine triphosphate (GTP)-dependent binding of aminoacyl-transfer RNA (tRNA) to the A-site of ribosomes during protein biosynthesis and is highly expressed in the central nervous system. De novo mutations in EEF1A2 have been identified in patients with extensive neurological deficits, ... Read more >>

Epilepsy Behav (Epilepsy & behavior : E&B)
[2020, 105:106955]

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The cannabidiol conundrum: potential benefits and risks of cannabidiol products for children.

Lauren Singer, Hannah Tokish, Faith Park, Christine Campisi, Ruth L Milanaik,

PURPOSE OF REVIEW:Since the federal ban on hemp products was lifted in December 2018, cannabidiol (CBD), a nonpsychoactive cannabinoid derived from hemp, has become increasingly popular and accessible. CBD is sold in the form of oils, tablets, and foods in locations including gas stations, cafés, and drug stores. Despite a ... Read more >>

Curr. Opin. Pediatr. (Current opinion in pediatrics)
[2020, 32(1):198-205]

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Open-label, uncontrolled retrospective study of perampanel in adults with Lennox-Gastaut syndrome.

Arielle Crespel, Ngoc Phuong Loc Tang, Greta Macorig, Philippe Gelisse, Pierre Genton,

PURPOSE:Perampanel (PER) was added to the anticonvulsant regimen of 71 patients with Lennox-Gastaut Syndrome (LGS) to evaluate its efficacy against seizures and its tolerability. METHOD:We evaluated at 3-month intervals 62 with pure LGS and 9 with LGS-like epileptic encephalopathy (28 females, 43 males, mean age 40.1 ± 11.5 yrs, median ... Read more >>

Seizure (Seizure)
[2020, 75:66-69]

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Asymmetric Slow-Spike-Wave Patterns with Maximal Discharges Contralateral to MRI Lesions Predict Better Surgical Prognosis in Symptomatic Lennox-Gastaut Syndrome or Lennox-Gastaut Phenotypes.

Mei-Hua Yang, Juan Liu, Yi-Ling Zhou, Hui Yang, Fang-Cheng Cai, John Zempel, Qing-Wu Yang, Shi-Yong Liu,

INTRODUCTION:Lennox-Gastaut syndrome (LGS) is a severe subtype of childhood-onset epileptic encephalopathy with drug-resistant and poor surgical prognosis. However, electroencephalogram (EEG) patterns of symptomatic LGS or LG phenotypes with structural brain lesions including focal abnormalities or asymmetric slow-spike-wave (SSW) patterns remain largely unknown. Due to the contradictory lateralization difference between MRI ... Read more >>

Pediatr Neurosurg (Pediatric neurosurgery)
[2020, 55(1):26-35]

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Automatic detection of generalized paroxysmal fast activity in Lennox-Gastaut syndrome using a bi-modal EEG time-frequency feature

Amir Omidvarnia, Aaron E.L. Warren, Linda Dalic, Mangor Pedersen, John Archer, Graeme Jackson,

Objective : Mark-up of generalized interictal epileptiform discharges (IEDs) on EEG is an important step in the diagnosis and characterization of epilepsy. However, manual EEG mark-up is a time-consuming, subjective, and highly specialized task where the human reviewer needs to visually inspect a large amount of data to facilitate accurate ... Read more >>

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Adjunctive Rufinamide in Children with Lennox-Gastaut Syndrome: A Literature Review.

Ganna Balagura, Antonella Riva, Francesca Marchese, Alberto Verrotti, Pasquale Striano,

Lennox-Gastaut syndrome (LGS) is a severe, childhood-onset, developmental epileptic encephalopathy, with different etiologies and co-morbidities. Seizure treatment in LGS represents a major challenge; new antiepileptic drugs (AEDs) are developed to especially address seizures resulting in high morbidity and mortality, such as drop seizures. Rufinamide (RFN) is one of the latest ... Read more >>

Neuropsychiatr Dis Treat (Neuropsychiatric disease and treatment)
[2020, 16:369-379]

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Effective and safe diet therapies for Lennox-Gastaut syndrome with mitochondrial dysfunction.

Ji-Hoon Na, Heung-Dong Kim, Young-Mock Lee,

Background:Lennox-Gastaut syndrome (LGS) is a typical intractable form of epilepsy that most often occurs between the second and sixth year of life. This study aimed to evaluate the clinical efficacy and safety of ketogenic diet therapies (DTs) for LGS with mitochondrial dysfunction. Methods:This was a retrospective study involving 20 LGS ... Read more >>

Ther Adv Neurol Disord (Therapeutic advances in neurological disorders)
[2020, 13:1756286419897813]

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Clinical features and electroclinical evolution in 22 cases with epileptic spasms without hypsarrhythmia.

Reiko Koichihara, Shin-Ichiro Hamano, Atsuro Daida, Hazuki Nonoyama, Satoru Ikemoto, Yuko Hirata, Ryuki Matsuura,

This study aimed to investigate the general presentation of epileptic spasms without hypsarrhythmia (ESwoH) and retrospectively determine whether there are differences in treatment effects related to ACTH therapy, long-term seizure outcome, and evolution of EEG features according to pre-treatment EEG patterns. According to the pattern of background activity, we divided ... Read more >>

Epileptic Disord (Epileptic disorders : international epilepsy journal with videotape)
[2020, 22(1):73-82]

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The role of cannabinoids in epilepsy treatment: a critical review of efficacy results from clinical trials.

Rima Nabbout, Elizabeth A Thiele,

CBD was shown to have anti-seizure activity based on in vitro and in vivo models. However, several reports of small series or case reports of the use of cannabis extracts in epilepsy yielded contradictory results and the efficacy of cannabis use in patients with epilepsy have also been inconclusive. In ... Read more >>

Epileptic Disord (Epileptic disorders : international epilepsy journal with videotape)
[2020, 22(S1):23-28]

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Clinical trial simulations of the interaction between cannabidiol and clobazam and effect on drop-seizure frequency.

Kirsten Riber Bergmann, Karen Broekhuizen, Geert Jan Groeneveld,

With this study, we aim to test the hypothesis that the effect of cannabidiol on drop-seizure frequency in patients with Lennox-Gastaut syndrome and Dravet syndrome could be attributed to a drug-drug interaction with clobazam. We performed clinical trial simulations for the effect of 20 mg/kg/day cannabidiol on drop-seizure frequency in patients ... Read more >>

Br J Clin Pharmacol (British journal of clinical pharmacology)
[2020, 86(2):380-385]

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Source of cannabinoids: what is available, what is used, and where does it come from?

Nicola Specchio, Nicola Pietrafusa, Helen J Cross,

Cannabis sativa L. is an ancient medicinal plant wherefrom over 120 cannabinoids are extracted. In the past two decades, there has been increasing interest in the therapeutic potential of cannabis-based treatments for neurological disorders such as epilepsy, and there is now evidence for the medical use of cannabis and its ... Read more >>

Epileptic Disord (Epileptic disorders : international epilepsy journal with videotape)
[2020, 22(S1):1-9]

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Cannabidiol with clobazam for seizures associated with Dravet and Lennox-Gastaut syndromes.

Alan Lamb, Dalia Dawoud, Ross Dent, Nicole Elliott, Amanda I Adler,

Lancet Neurol (The Lancet. Neurology)
[2020, 19(4):290-291]

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A Novel X-Linked Variant of IQSEC2 is Associated with Lennox-Gastaut Syndrome and Mild Intellectual Disability in Three Generations of a Korean Family.

Min-Hyuk Choi, Jin Ok Yang, Ju-Sik Min, Jeong-Ju Lee, Soo-Young Jun, Yong-Jae Lee, Ji-Yong Yoon, Su-Jin Jeon, Iksu Byeon, Joon-Won Kang, Nam-Soon Kim,

Aim: Lennox-Gastaut syndrome (LGS) is a severe type of childhood-onset epilepsy with multiple types of seizures, specific discharges on electroencephalography, and intellectual disability. However, LGS-related genes are largely unknown. To identify causative genes related to LGS, we collected and analyzed data from a three-generation Korean family in which one member ... Read more >>

Genet Test Mol Biomarkers (Genetic testing and molecular biomarkers)
[2020, 24(1):54-58]

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Targeting the centromedian thalamic nucleus for deep brain stimulation.

Aaron E L Warren, Linda J Dalic, Wesley Thevathasan, Annie Roten, Kristian J Bulluss, John Archer,

OBJECTIVES:Deep brain stimulation (DBS) of the centromedian thalamic nucleus (CM) is an emerging treatment for multiple brain diseases, including the drug-resistant epilepsy Lennox-Gastaut syndrome (LGS). We aimed to improve neurosurgical targeting of the CM by: (1) developing a structural MRI approach for CM visualisation, (2) identifying the CM's neurophysiological characteristics ... Read more >>

J. Neurol. Neurosurg. Psychiatry (Journal of neurology, neurosurgery, and psychiatry)
[2020, 91(4):339-349]

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Epilepsy and cannabidiol: a guide to treatment.

Alexis Arzimanoglou, Ulrich Brandl, J Helen Cross, Antonio Gil-Nagel, Lieven Lagae, Cecilie Johannessen Landmark, Nicola Specchio, Rima Nabbout, Elizabeth A Thiele, Oliver Gubbay, The Cannabinoids International Experts Panel, ,

The growing interest in cannabidiol (CBD), specifically a pure form of CBD, as a treatment for epilepsy, among other conditions, is reflected in recent changes in legislation in some countries. Although there has been much speculation about the therapeutic value of cannabis-based products as an anti-seizure treatment for some time, ... Read more >>

Epileptic Disord (Epileptic disorders : international epilepsy journal with videotape)
[2020, 22(1):1-14]

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Adverse effects of cannabinoids.

Carla Anciones, Antonio Gil-Nagel,

Cannabidiol is a cannabinoid-derived product that has recently been approved for the treatment of pharmacoresistant seizures in patients with epileptic encephalopathies such as Dravet Syndrome and Lennox-Gastaut Syndrome. Short-term side effects of cannabidiol are well know and well-documented in the clinical trials that lead to its approval. Generally, is a ... Read more >>

Epileptic Disord (Epileptic disorders : international epilepsy journal with videotape)
[2020, 22(S1):29-32]

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From Standard of Care to Personalized (Art of) Medicine: Two Novel GABA-A Receptor β3 Subunit Mutations Associated With Epilepsy Syndromes.

Libor Velisek,

[Box: see text]. ... Read more >>

Epilepsy Curr (Epilepsy currents)
[2020, 20(1):45-47]

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Cognitive and functional status in late-onset Lennox-Gastaut syndrome: Variation on a classic phenotype.

Nitish Chourasia, Atul Maheshwari, Giridhar Kalamangalam,

Lennox-Gastaut syndrome (LGS) denotes a refractory epileptic encephalopathy of childhood onset with the triad of generalized slow spike-wave (GSSW) on interictal scalp electroencephalogram (EEG), multiple seizure types, and intellectual impairment. The neurobiology of LGS is said to sustain abnormal patterns of brain activity and connectivity that ultimately impair normal cerebral ... Read more >>

Epilepsy Behav (Epilepsy & behavior : E&B)
[2020, 102:106660]

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Epilepsy in Christianson syndrome: Two cases of Lennox-Gastaut syndrome and a review of literature.

Azusa Ikeda, Ayako Yamamoto, Kazushi Ichikawa, Yu Tsuyusaki, Megumi Tsuji, Mizue Iai, Yumi Enomoto, Hiroaki Murakami, Kenji Kurosawa, Satoko Miyatake, Naomichi Matsumoto, Tomohide Goto,

Christianson syndrome (CS) is an X-linked intellectual disorder caused by mutations in the SLC9A6 gene. Clinical features of CS include an inability to speak, truncal ataxia, postnatal microcephaly, hyperkinesis, and epilepsy. Almost all patients with CS develop drug-resistant epilepsy-its most serious complication. We report two cases of CS with drug-resistant ... Read more >>

Epilepsy Behav Rep (Epilepsy & behavior reports)
[2020, 13:100349]

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Response to cannabidiol in epilepsy of infancy with migrating focal seizures associated with KCNT1 mutations: An open-label, prospective, interventional study.

Kelsey Poisson, Matthew Wong, Chon Lee, Maria Roberta Cilio,

Epilepsy of Infancy with Migrating Focal Seizures (EIMFS) is a rare, developmental and epileptic encephalopathy most commonly associated with mutations in KCNT1, a potassium channel. Polymorphous migrating focal seizures begin within 6 months of life and are pharmacoresistant to standard anticonvulsants. Additional therapies are needed to decrease seizure frequency and ... Read more >>

Eur. J. Paediatr. Neurol. (European journal of paediatric neurology : EJPN : official journal of the European Paediatric Neurology Society)
[2019, :]

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A reflection on the role of genetics in the concept of "epileptic encephalopathy", as emerged from the most recent ILEA classification of epilepsy.

Angelo Russo, Giuseppe Gobbi,

The International League Against Epilepsy (ILAE) has been working to standardize the epilepsy classifications for over a hundred years.The latest epilepsy classification has been recently carried out with a careful overview on several topics including the "epileptic encephalopathies" concept and several constructive discussions on this topic have taken place in ... Read more >>

Ital J Pediatr (Italian journal of pediatrics)
[2020, 46(1):2]

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Epidiolex (Cannabidiol) Primer: Frequently Asked Questions for Patients and Caregivers.

Renad Abu-Sawwa, Caitlin Stehling,

J Pediatr Pharmacol Ther (The journal of pediatric pharmacology and therapeutics : JPPT : the official journal of PPAG)
[2020, 25(1):75-77]

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Two Pediatric Cases of Successful Management of Postictal Transient Urinary Retention.

Ho Eun Park, Je-Sang Lee, Dong Min Kim, Yong Beom Shin,

We report two cases of postictal urinary retention in pediatric patients with cognitive impairment. Two girls with intellectual disabilities, concomitant cerebral palsy (case 1) and Rett syndrome (case 2), developed urinary retention following seizures. Their caregivers brought them to the hospital with complaints of abdominal distension. After excluding neurological progression, ... Read more >>

Ann Rehabil Med (Annals of rehabilitation medicine)
[2020, 44(1):90-93]

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Drug Development for Rare Paediatric Epilepsies: Current State and Future Directions.

Stéphane Auvin, Andreja Avbersek, Thomas Bast, Catherine Chiron, Renzo Guerrini, Rafal M Kaminski, Lieven Lagae, Pierandrea Muglia, J Helen Cross,

Rare diseases provide a challenge in the evaluation of new therapies. However, orphan drug development is of increasing interest because of the legislation enabling facilitated support by regulatory agencies through scientific advice, and the protection of the molecules with orphan designation. In the landscape of the rare epilepsies, very few ... Read more >>

Drugs (Drugs)
[2019, 79(18):1917-1935]

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