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https://www.readbyqxmd.com/read/28424564/new-developments-in-the-management-of-narcolepsy
#1
REVIEW
Vivien C Abad, Christian Guilleminault
Narcolepsy is a life-long, underrecognized sleep disorder that affects 0.02%-0.18% of the US and Western European populations. Genetic predisposition is suspected because of narcolepsy's strong association with HLA DQB1*06-02, and genome-wide association studies have identified polymorphisms in T-cell receptor loci. Narcolepsy pathophysiology is linked to loss of signaling by hypocretin-producing neurons; an autoimmune etiology possibly triggered by some environmental agent may precipitate hypocretin neuronal loss...
2017: Nature and Science of Sleep
https://www.readbyqxmd.com/read/28396432/serotonin-neurons-in-the-dorsal-raphe-mediate-the-anticataplectic-action-of-orexin-neurons-by-reducing-amygdala-activity
#2
Emi Hasegawa, Takashi Maejima, Takayuki Yoshida, Olivia A Masseck, Stefan Herlitze, Mitsuhiro Yoshioka, Takeshi Sakurai, Michihiro Mieda
Narcolepsy is a sleep disorder caused by the loss of orexin (hypocretin)-producing neurons and marked by excessive daytime sleepiness and a sudden weakening of muscle tone, or cataplexy, often triggered by strong emotions. In a mouse model for narcolepsy, we previously demonstrated that serotonin neurons of the dorsal raphe nucleus (DRN) mediate the suppression of cataplexy-like episodes (CLEs) by orexin neurons. Using an optogenetic tool, in this paper we show that the acute activation of DRN serotonin neuron terminals in the amygdala, but not in nuclei involved in regulating rapid eye-movement sleep and atonia, suppressed CLEs...
April 10, 2017: Proceedings of the National Academy of Sciences of the United States of America
https://www.readbyqxmd.com/read/28366327/underutilization-of-the-mslt-in-sleepy-patients-with-a-short-onset-rem-period-soremp-in-the-sleep-clinic
#3
Alyssa Cairns, Richard Bogan
OBJECTIVE/BACKGROUND: A nocturnal sleep onset REM period (defined as REM onset latency ≤ 15 min; SOREMP) occurs rarely and research has shown that the phenomenon is specific for type 1 and 2 narcolepsy. However, little is known about the meaningfulness of the phenotype in general sleep clinic patients because those that exhibit the phenomenon often present with few traditional narcolepsy symptoms. As such, this study aimed to (1) evaluate the rate of eventual MSLT testing for those with a SOREMP on routine PSG when the phenomenon occurred in the absence of potential explanatory factors and (2) quantify the stability of the SOREMP phenotype...
April 2017: Sleep Medicine
https://www.readbyqxmd.com/read/28366326/assessing-sleepiness-and-cataplexy-in-children-and-adolescents-with-narcolepsy-a-review-of-current-patient-reported-measures
#4
REVIEW
Khadra Benmedjahed, Y Grace Wang, Jérémy Lambert, Christopher Evans, Steve Hwang, Jed Black, Murray W Johns
OBJECTIVE: The objective of this study was to review patient-reported outcome measures assessing excessive daytime sleepiness (EDS) or cataplexy in children or adolescents to determine their usefulness and limitations in pediatric narcolepsy assessment. METHODS: Searches were performed in Embase and Medline for pediatric measures of EDS and cataplexy that are either patient- or proxy-reported, and searches of http://www.clinicaltrials.gov/ were conducted for studies in narcolepsy that included at least one patient-reported measure...
April 2017: Sleep Medicine
https://www.readbyqxmd.com/read/28364512/nocturnal-dynamics-of-sleep-wake-transitions-in-patients-with-narcolepsy
#5
Xiaozhe Zhang, Jan W Kantelhardt, Xiao Song Dong, Dagmar Krefting, Jing Li, Han Yan, Frank Pillmann, Ingo Fietze, Thomas Penzel, Long Zhao, Fang Han
Introduction: We investigate how characteristics of sleep-wake dynamics in humans are modified by narcolepsy, a clinical condition that is supposed to destabilize sleep-wake regulation. Subjects with and without cataplexy are considered separately. Differences in sleep scoring habits as a possible confounder have been examined. Aims and Methods: Four groups of subjects are considered: narcolepsy patients from China with (n = 88) and without (n = 15) cataplexy, healthy controls from China (n = 110) and from Europe (n = 187, 2 nights each)...
February 1, 2017: Sleep
https://www.readbyqxmd.com/read/28364477/temporal-changes-in-the-cerebrospinal-fluid-level-of-hypocretin-1-and-histamine-in-narcolepsy
#6
Régis Lopez, Lucie Barateau, Elisa Evangelista, Sofiene Chenini, Philippe Robert, Isabelle Jaussent, Yves Dauvilliers
Study Objectives: To follow the temporal changes of cerebrospinal fluid (CSF) biomarker levels in narcoleptic patients with unexpected hypocretin level at referral. Methods: From 2007 to 2015, 170 human leukocyte antigen (HLA) DQB1*06:02-positive patients with primary narcolepsy and definite (n = 155, 95 males, 60 females, 36 children) or atypical cataplexy (n = 15, 4 males, 3 children) were referred to our center. Cerebrospinal hypocretin deficiency was found in 95...
January 1, 2017: Sleep
https://www.readbyqxmd.com/read/28343142/circuit-mechanisms-of-sleepiness-and-cataplexy-in-narcolepsy
#7
REVIEW
Sara Pintwala, John Peever
Narcolepsy is a debilitating sleep disorder caused by loss of orexin neurons in the lateral hypothalamus. Excessive daytime sleepiness and cataplexy are the major complaints in narcolepsy, and are associated with impaired quality of life. Although it is unclear how orexin loss causes sleepiness and cataplexy, animal models have been instrumental in identifying the neurobiological underpinnings of narcolepsy because they reliably recapitulate disease symptoms. Current evidence indicates that orexin cell loss causes sleepiness and cataplexy by destabilizing the ability of the circuits that initiate and sustain normal levels of arousal and motor activity...
March 23, 2017: Current Opinion in Neurobiology
https://www.readbyqxmd.com/read/28332559/the-immunogenetics-of-narcolepsy-associated-with-a-h1n1-pdm09-vaccination-pandemrix-supports-a-potent-gene-environment-interaction
#8
I L Bomfim, F Lamb, K Fink, A Szakács, A Silveira, L Franzén, V Azhary, M Maeurer, N Feltelius, N Darin, T Hallböök, L Arnheim-Dahlström, I Kockum, T Olsson
The influenza A(H1N1)pdm09 vaccination campaign from 2009 to 2010 was associated with a sudden increase in the incidence of narcolepsy in several countries. Narcolepsy with cataplexy is strongly associated with the human leukocyte antigen (HLA) class II DQB1*06:02 allele, and protective associations with the DQB1*06:03 allele have been reported. Several non-HLA gene loci are also associated, such as common variants of the T-cell receptor-α (TRA), the purinergic receptor P2RY11, cathepsin H (CTSH) and TNFSF4/OX40L/CD252...
March 23, 2017: Genes and Immunity
https://www.readbyqxmd.com/read/28258080/measurement-of-narcolepsy-symptoms-the-narcolepsy-severity-scale
#9
Yves Dauvilliers, Severine Beziat, Carole Pesenti, Regis Lopez, Lucie Barateau, Bertrand Carlander, Gianina Luca, Mehdi Tafti, Charles M Morin, Michel Billiard, Isabelle Jaussent
OBJECTIVE: To validate the Narcolepsy Severity Scale (NSS), a brief clinical instrument to evaluate the severity and consequences of symptoms in patients with narcolepsy type 1 (NT1). METHODS: A 15-item scale to assess the frequency and severity of excessive daytime sleepiness, cataplexy, hypnagogic hallucinations, sleep paralysis, and disrupted nighttime sleep was developed and validated by sleep experts with patients' feedback. Seventy untreated and 146 treated adult patients with NT1 were evaluated and completed the NSS in a single reference sleep center...
March 3, 2017: Neurology
https://www.readbyqxmd.com/read/28235898/gabaergic-neurons-of-the-central-amygdala-promote-cataplexy
#10
Carrie E Mahoney, Lindsay J Agostinelli, Jessica N K Brooks, Bradford B Lowell, Thomas E Scammell
Narcolepsy is characterized by chronic sleepiness and cataplexy-sudden muscle paralysis triggered by strong, positive emotions. This condition is caused by a lack of orexin (hypocretin) signaling, but little is known about the neural mechanisms that mediate cataplexy. The amygdala regulates responses to rewarding stimuli and contains neurons active during cataplexy. In addition, lesions of the amygdala reduce cataplexy. Because GABAergic neurons of the central nucleus of the amygdala (CeA) target brainstem regions known to regulate muscle tone, we hypothesized that these cells promote emotion-triggered cataplexy...
April 12, 2017: Journal of Neuroscience: the Official Journal of the Society for Neuroscience
https://www.readbyqxmd.com/read/28215239/cataplexy-causing-subdural-hematomas
#11
Nuria Matos, Carles Gaig, Joan Santamaria, Alex Iranzo
No abstract text is available yet for this article.
February 2017: Sleep Medicine
https://www.readbyqxmd.com/read/28209737/gaba-cells-in-the-central-nucleus-of-the-amygdala-promote-cataplexy
#12
Matthew B Snow, Jimmy J Fraigne, Gabrielle Thibault-Messier, Victoria L Chuen, Aren Thomasian, Richard L Horner, John Peever
Cataplexy is a hallmark of narcolepsy characterized by the sudden uncontrollable onset of muscle weakness or paralysis during wakefulness. It can occur spontaneously, but is typically triggered by positive emotions such as laughter. Although cataplexy was identified >130 years ago, its neural mechanism remains unclear. Here, we show that a newly identified GABA circuit within the central nucleus of the amygdala (CeA) promotes cataplexy. We used behavioral, electrophysiological, immunohistochemical, and chemogenetic strategies to target and manipulate CeA activity selectively in narcoleptic (orexin(-/-)) mice to determine its functional role in controlling cataplexy...
April 12, 2017: Journal of Neuroscience: the Official Journal of the Society for Neuroscience
https://www.readbyqxmd.com/read/28179647/narcolepsy
#13
REVIEW
Birgitte R Kornum, Stine Knudsen, Hanna M Ollila, Fabio Pizza, Poul J Jennum, Yves Dauvilliers, Sebastiaan Overeem
Narcolepsy is a chronic sleep disorder that has a typical onset in adolescence and is characterized by excessive daytime sleepiness, which can have severe consequences for the patient. Problems faced by patients with narcolepsy include social stigma associated with this disease, difficulties in obtaining an education and keeping a job, a reduced quality of life and socioeconomic consequences. Two subtypes of narcolepsy have been described (narcolepsy type 1 and narcolepsy type 2), both of which have similar clinical profiles, except for the presence of cataplexy, which occurs only in patients with narcolepsy type 1...
February 9, 2017: Nature Reviews. Disease Primers
https://www.readbyqxmd.com/read/28162825/safety-of-quadrivalent-live-attenuated-influenza-vaccine-in-subjects-aged-2-49years
#14
Roger Baxter, Abigail Eaton, John Hansen, Laurie Aukes, Herve Caspard, Christopher S Ambrose
BACKGROUND: Quadrivalent live attenuated influenza vaccine (Q/LAIV) was licensed in 2012 and replaced trivalent live attenuated influenza vaccine in the United States during the 2013-2014 influenza season. This study assessed the safety of Q/LAIV in children and adults aged 2-49years. METHODS: This was a prospective observational cohort study using data collected from Kaiser Permanente Northern California. Post-vaccination events of interest were any hospitalization, hospitalization for lower respiratory tract infection, and the following medically attended events: hypersensitivity, seizures/convulsions, lower respiratory tract infection, wheezing, Guillain-Barré syndrome, Bell's palsy, encephalitis, neuritis, vasculitis, and narcolepsy/cataplexy...
March 1, 2017: Vaccine
https://www.readbyqxmd.com/read/28162143/patient-reported-measures-of-narcolepsy-the-need-for-better-assessment
#15
Ulf Kallweit, Markus Schmidt, Claudio L Bassetti
STUDY OBJECTIVES: Narcolepsy, a chronic disorder of the central nervous system, is clinically characterized by a symptom pentad that includes excessive daytime sleepiness, cataplexy, sleep paralysis, hypnopompic/hypnagogic hallucinations, and disrupted nighttime sleep. Ideally, screening and diagnosis instruments that assist physicians in evaluating a patient for type 1 or type 2 narcolepsy would be brief, easy for patients to understand and physicians to score, and would identify or rule out the need for electrophysiological testing...
January 31, 2017: Journal of Clinical Sleep Medicine: JCSM: Official Publication of the American Academy of Sleep Medicine
https://www.readbyqxmd.com/read/28139340/ultra-high-performance-liquid-chromatography-tandem-mass-spectrometry-determination-of-ghb-ghb-glucuronide-in-plasma-and-cerebrospinal-fluid-of-narcoleptic-patients-under-sodium-oxybate-treatment
#16
Roberta Tittarelli, Simona Pichini, Daniel S Pedersen, Roberta Pacifici, Monica Moresco, Fabio Pizza, Francesco Paolo Busardò, Giuseppe Plazzi
Sodium oxybate (Xyrem(®)), the sodium salt of γ- hydroxybutyric acid (GHB), is a first-line treatment of the symptoms induced by type 1 narcolepsy (NT1) and it is highly effective in improving sleep architecture, decreasing excessive daytime sleepiness and the frequency of cataplexy attacks. Using an ultra-high-performance liquid chromatography tandem mass spectrometry (UHPLC-MS/MS) validated method, GHB was determined together with its glucuronide (GHB-gluc), in plasma and cerebrospinal fluid (CSF) samples of NT1 patients under sodium oxybate treatment...
May 2017: Forensic Science International
https://www.readbyqxmd.com/read/28129985/safety-and-efficacy-of-pitolisant-on-cataplexy-in-patients-with-narcolepsy-a-randomised-double-blind-placebo-controlled-trial
#17
Zoltan Szakacs, Yves Dauvilliers, Vladimir Mikhaylov, Irina Poverennova, Sergei Krylov, Slavko Jankovic, Karel Sonka, Philippe Lehert, Isabelle Lecomte, Jeanne-Marie Lecomte, Jean-Charles Schwartz
BACKGROUND: Histaminergic neurons are crucial to maintain wakefulness, but their role in cataplexy is unknown. We assessed the safety and efficacy of pitolisant, a histamine H3 receptor inverse agonist, for treatment of cataplexy in patients with narcolepsy. METHODS: For this randomised, double-blind, placebo-controlled trial we recruited patients with narcolepsy from 16 sleep centres in nine countries (Bulgaria, Czech Republic, Hungary, Macedonia, Poland, Russia, Serbia, Turkey, and Ukraine)...
March 2017: Lancet Neurology
https://www.readbyqxmd.com/read/28129984/wide-implications-of-a-trial-on-pitolisant-for-cataplexy
#18
Christian R Baumann
No abstract text is available yet for this article.
March 2017: Lancet Neurology
https://www.readbyqxmd.com/read/28123336/narcolepsy-and-cataplexy-a-pediatric-case-report
#19
Tülin Savaş, Ilknur Erol, Semra Saygı, Mehmet Ali Habeşoğlu
Narcolepsy is characterized by excessive sleepiness, cataplexy, hypnagogic hallucinations, and sleep paralysis during the rapid eye movement period of sleep. Herein, we present a boy aged eight years who was diagnosed as having narcolepsy and cataplexy about thirteen months after his first presentation. He was admitted with symptoms of daytime sleepiness. In the follow-up, cataplexy in the form of head dropping attacks developed seven months after the first admission. The patient was investigated for different prediagnoses and was eventually diagnosed as having narcolepsy and cataplexy through polysomnography and multiple sleep latency tests thirteen months after the first presentation...
December 2016: Türk Pediatri Arşivi
https://www.readbyqxmd.com/read/28108192/epidemiology-and-pathophysiology-of-childhood-narcolepsy
#20
REVIEW
Thomas J Dye, Neepa Gurbani, Narong Simakajornboon
It is now recognized that there are two types of narcolepsy. Narcolepsy type I or Narcolepsy with cataplexy is caused by the loss of hypocretin or orexin neurons. Narcolepsy type II or narcolepsy without cataplexy has normal hypocretin and the etiology is unknown. Hypocretin is a neuropeptide produced by neurons in the lateral hypothalamus. Both genetic and environmental factors play a crucial role in the pathogenesis of narcolepsy. Most patients with narcolepsy type I and half of patients with narcolepsy type II carry HLA-DQB1*0602...
December 21, 2016: Paediatric Respiratory Reviews
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