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Auteur Floor E. SCHEEPERS |
Documents disponibles écrits par cet auteur (2)
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Effects of bumetanide on neurodevelopmental impairments in patients with tuberous sclerosis complex: an open-label pilot study / Dorinde M. VAN ANDEL in Molecular Autism, 11 (2020)
[article]
Titre : Effects of bumetanide on neurodevelopmental impairments in patients with tuberous sclerosis complex: an open-label pilot study Type de document : Texte imprimé et/ou numérique Auteurs : Dorinde M. VAN ANDEL, Auteur ; Jan J. SPRENGERS, Auteur ; Bob ORANJE, Auteur ; Floor E. SCHEEPERS, Auteur ; Floor E. JANSEN, Auteur ; Hilgo BRUINING, Auteur Article en page(s) : 30 p. Langues : Anglais (eng) Mots-clés : Bumetanide Erp Irritability NKCC1 antagonist Neurocognitive task Open-label Tand Tuberous sclerosis complex Index. décimale : PER Périodiques Résumé : BACKGROUND: Tuberous sclerosis complex (TSC) is an autosomal dominant disease that affects multiple organs including the brain. TSC is strongly associated with broad neurodevelopmental disorders, including autism spectrum disorder symptomatology. Preclinical TSC studies have indicated altered neuronal chloride homeostasis affecting the polarity of ?-aminobutyric acid (GABA) ergic transmission as a potential treatment target. Bumetanide, a selective NKCC1 chloride importer antagonist, may attenuate depolarizing GABA action, and in that way reduce disease burden. In this open-label pilot study, we tested the effect of bumetanide on a variety of neurophysiological, cognitive, and behavioral measures in children with TSC. METHODS: Participants were treated with bumetanide (2dd 0.5-1.0?mg) for 13?weeks in an open-label trial. The Aberrant Behavior Checklist-Irritability (ABC-I) subscale was chosen as the primary endpoint. Secondary endpoints included other behavioral questionnaires in addition to event-related potentials (ERP) and neuropsychological tests if tolerated. Additionally, the treatment effect on seizure frequency and quality of life was assessed. Endpoint data were collected at baseline, after 91?days of treatment and after a 28-day wash-out period. RESULTS: Fifteen patients (8-21-years old) with TSC were included of which 13 patients completed the study. Treatment was well-tolerated with only expected adverse events due to the diuretic effects of bumetanide. Irritable behavior (ABC-I) showed significant improvement after treatment in 11 out of 13 patients (t(12) = 4.41, p = .001, d = .773). A favorable effect was also found for social behavior (Social Responsiveness Scale) (t(11) = 4.01, p = .002, d = .549) and hyperactive behavior (ABC-hyperactivity subscale) (t(12) = 3.65, p = .003, d = .686). Moreover, patients rated their own health-related quality of life higher after treatment. At baseline, TSC patients showed several atypical ERPs versus typically developing peers of which prepulse inhibition was significantly decreased in the TSC group. Neuropsychological measurements showed no change and bumetanide had no effect on seizure frequency. LIMITATIONS: The sample size and open-label design of this pilot study warrant caution when interpreting outcome measures. CONCLUSIONS: Bumetanide treatment is a potential treatment to alleviate the behavioral burden and quality of life associated with TSC. More elaborate trials are needed to determine the application and effect size of bumetanide for the TSC population. Trial registration EU Clinical Trial Register, EudraCT 2016-002408-13 (www.clinicaltrialsregister.eu/ctr-search/trial/2016-002408-13/NL). Registered 25 July 2016. En ligne : http://dx.doi.org/10.1186/s13229-020-00335-4 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=427
in Molecular Autism > 11 (2020) . - 30 p.[article] Effects of bumetanide on neurodevelopmental impairments in patients with tuberous sclerosis complex: an open-label pilot study [Texte imprimé et/ou numérique] / Dorinde M. VAN ANDEL, Auteur ; Jan J. SPRENGERS, Auteur ; Bob ORANJE, Auteur ; Floor E. SCHEEPERS, Auteur ; Floor E. JANSEN, Auteur ; Hilgo BRUINING, Auteur . - 30 p.
Langues : Anglais (eng)
in Molecular Autism > 11 (2020) . - 30 p.
Mots-clés : Bumetanide Erp Irritability NKCC1 antagonist Neurocognitive task Open-label Tand Tuberous sclerosis complex Index. décimale : PER Périodiques Résumé : BACKGROUND: Tuberous sclerosis complex (TSC) is an autosomal dominant disease that affects multiple organs including the brain. TSC is strongly associated with broad neurodevelopmental disorders, including autism spectrum disorder symptomatology. Preclinical TSC studies have indicated altered neuronal chloride homeostasis affecting the polarity of ?-aminobutyric acid (GABA) ergic transmission as a potential treatment target. Bumetanide, a selective NKCC1 chloride importer antagonist, may attenuate depolarizing GABA action, and in that way reduce disease burden. In this open-label pilot study, we tested the effect of bumetanide on a variety of neurophysiological, cognitive, and behavioral measures in children with TSC. METHODS: Participants were treated with bumetanide (2dd 0.5-1.0?mg) for 13?weeks in an open-label trial. The Aberrant Behavior Checklist-Irritability (ABC-I) subscale was chosen as the primary endpoint. Secondary endpoints included other behavioral questionnaires in addition to event-related potentials (ERP) and neuropsychological tests if tolerated. Additionally, the treatment effect on seizure frequency and quality of life was assessed. Endpoint data were collected at baseline, after 91?days of treatment and after a 28-day wash-out period. RESULTS: Fifteen patients (8-21-years old) with TSC were included of which 13 patients completed the study. Treatment was well-tolerated with only expected adverse events due to the diuretic effects of bumetanide. Irritable behavior (ABC-I) showed significant improvement after treatment in 11 out of 13 patients (t(12) = 4.41, p = .001, d = .773). A favorable effect was also found for social behavior (Social Responsiveness Scale) (t(11) = 4.01, p = .002, d = .549) and hyperactive behavior (ABC-hyperactivity subscale) (t(12) = 3.65, p = .003, d = .686). Moreover, patients rated their own health-related quality of life higher after treatment. At baseline, TSC patients showed several atypical ERPs versus typically developing peers of which prepulse inhibition was significantly decreased in the TSC group. Neuropsychological measurements showed no change and bumetanide had no effect on seizure frequency. LIMITATIONS: The sample size and open-label design of this pilot study warrant caution when interpreting outcome measures. CONCLUSIONS: Bumetanide treatment is a potential treatment to alleviate the behavioral burden and quality of life associated with TSC. More elaborate trials are needed to determine the application and effect size of bumetanide for the TSC population. Trial registration EU Clinical Trial Register, EudraCT 2016-002408-13 (www.clinicaltrialsregister.eu/ctr-search/trial/2016-002408-13/NL). Registered 25 July 2016. En ligne : http://dx.doi.org/10.1186/s13229-020-00335-4 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=427 The cognitive and neural correlates of psychopathy and especially callous–unemotional traits in youths: A systematic review of the evidence / Pierre C. M. HERPERS in Development and Psychopathology, 26-1 (February 2014)
[article]
Titre : The cognitive and neural correlates of psychopathy and especially callous–unemotional traits in youths: A systematic review of the evidence Type de document : Texte imprimé et/ou numérique Auteurs : Pierre C. M. HERPERS, Auteur ; Floor E. SCHEEPERS, Auteur ; Daniëlle M. A. BONS, Auteur ; Jan K. BUITELAAR, Auteur ; Nanda N. ROMMELSE, Auteur Article en page(s) : p.245-273 Langues : Français (fre) Index. décimale : PER Périodiques Résumé : It is unclear whether the concepts and findings of the underlying neurobiology of adult psychopathy apply to youths as well. If so, a life span approach to treatment should be taken. Because youths’ brains are still developing, interventions at an early age may be far more effective in the long run. The aim of this systematic review is to examine whether the neurocognitive and neurobiological factors that underlie juvenile psychopathy, and specifically callous–unemotional (CU) traits, are similar to those underlying adult psychopathy. The results show that youths with CU traits show lower levels of prosocial reasoning, lower emotional responsivity, and decreased harm avoidance. Brain imaging studies in youths with CU traits are still rare. Available studies suggest specific neural correlates, such as a reduced response of the amygdala and a weaker functional connectivity between the amygdala and the ventromedial prefrontal cortex. These findings are largely in line with existing theories of adult psychopathy, such as the dual-hormone serotonergic hypothesis and the integrated emotions systems theory. We recommend that future studies investigate the role of oxytocin, invest in the study of neural mechanisms, and study the precursors, risk factors, and correlates of CU traits in early infancy and in longitudinal designs. En ligne : http://dx.doi.org/10.1017/S0954579413000527 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=224
in Development and Psychopathology > 26-1 (February 2014) . - p.245-273[article] The cognitive and neural correlates of psychopathy and especially callous–unemotional traits in youths: A systematic review of the evidence [Texte imprimé et/ou numérique] / Pierre C. M. HERPERS, Auteur ; Floor E. SCHEEPERS, Auteur ; Daniëlle M. A. BONS, Auteur ; Jan K. BUITELAAR, Auteur ; Nanda N. ROMMELSE, Auteur . - p.245-273.
Langues : Français (fre)
in Development and Psychopathology > 26-1 (February 2014) . - p.245-273
Index. décimale : PER Périodiques Résumé : It is unclear whether the concepts and findings of the underlying neurobiology of adult psychopathy apply to youths as well. If so, a life span approach to treatment should be taken. Because youths’ brains are still developing, interventions at an early age may be far more effective in the long run. The aim of this systematic review is to examine whether the neurocognitive and neurobiological factors that underlie juvenile psychopathy, and specifically callous–unemotional (CU) traits, are similar to those underlying adult psychopathy. The results show that youths with CU traits show lower levels of prosocial reasoning, lower emotional responsivity, and decreased harm avoidance. Brain imaging studies in youths with CU traits are still rare. Available studies suggest specific neural correlates, such as a reduced response of the amygdala and a weaker functional connectivity between the amygdala and the ventromedial prefrontal cortex. These findings are largely in line with existing theories of adult psychopathy, such as the dual-hormone serotonergic hypothesis and the integrated emotions systems theory. We recommend that future studies investigate the role of oxytocin, invest in the study of neural mechanisms, and study the precursors, risk factors, and correlates of CU traits in early infancy and in longitudinal designs. En ligne : http://dx.doi.org/10.1017/S0954579413000527 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=224