Centre d'Information et de documentation du CRA Rhône-Alpes
CRA
Informations pratiques
-
Adresse
Centre d'information et de documentation
du CRA Rhône-Alpes
Centre Hospitalier le Vinatier
bât 211
95, Bd Pinel
69678 Bron CedexHoraires
Lundi au Vendredi
9h00-12h00 13h30-16h00Contact
Tél: +33(0)4 37 91 54 65
Mail
Fax: +33(0)4 37 91 54 37
-
Détail de l'auteur
Auteur Jan J. SPRENGERS |
Documents disponibles écrits par cet auteur (3)
Faire une suggestion Affiner la recherche
Effects of Bumetanide on Neurocognitive Functioning in Children with Autism Spectrum Disorder: Secondary Analysis of a Randomized Placebo-Controlled Trial / Dorinde M. VAN ANDEL in Journal of Autism and Developmental Disorders, 54-3 (March 2024)
[article]
Titre : Effects of Bumetanide on Neurocognitive Functioning in Children with Autism Spectrum Disorder: Secondary Analysis of a Randomized Placebo-Controlled Trial Type de document : Texte imprimé et/ou numérique Auteurs : Dorinde M. VAN ANDEL, Auteur ; Jan J. SPRENGERS, Auteur ; Marsh KÖNIGS, Auteur ; Maretha V. DE JONGE, Auteur ; Hilgo BRUINING, Auteur Article en page(s) : p.894-904 Langues : Anglais (eng) Index. décimale : PER Périodiques Résumé : We present the secondary-analysis of neurocognitive tests in the 'Bumetanide in Autism Medication and Biomarker' (BAMBI;EUDRA-CT-2014-001560-35) study, a randomized double-blind placebo-controlled (1:1) trial testing 3-months bumetanide treatment (??1 mg twice-daily) in unmedicated children 7-15 years with ASD. Children with IQ???70 were analyzed for baseline deficits and treatment-effects on the intention-to-treat-population with generalized-linear-models, principal component analysis and network analysis. Ninety-two children were allocated to treatment and 83 eligible for analyses. Heterogeneous neurocognitive impairments were found that were unaffected by bumetanide treatment. Network analysis showed higher modularity after treatment (mean difference:-0.165, 95%CI:-0.317 to ??0.013,p = .034) and changes in the relative importance of response inhibition in the neurocognitive network (mean difference:-0.037, 95%CI:-0.073 to ??0.001,p = .042). This study offers perspectives to include neurocognitive tests in ASD trials. En ligne : https://doi.org/10.1007/s10803-022-05841-3 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=524
in Journal of Autism and Developmental Disorders > 54-3 (March 2024) . - p.894-904[article] Effects of Bumetanide on Neurocognitive Functioning in Children with Autism Spectrum Disorder: Secondary Analysis of a Randomized Placebo-Controlled Trial [Texte imprimé et/ou numérique] / Dorinde M. VAN ANDEL, Auteur ; Jan J. SPRENGERS, Auteur ; Marsh KÖNIGS, Auteur ; Maretha V. DE JONGE, Auteur ; Hilgo BRUINING, Auteur . - p.894-904.
Langues : Anglais (eng)
in Journal of Autism and Developmental Disorders > 54-3 (March 2024) . - p.894-904
Index. décimale : PER Périodiques Résumé : We present the secondary-analysis of neurocognitive tests in the 'Bumetanide in Autism Medication and Biomarker' (BAMBI;EUDRA-CT-2014-001560-35) study, a randomized double-blind placebo-controlled (1:1) trial testing 3-months bumetanide treatment (??1 mg twice-daily) in unmedicated children 7-15 years with ASD. Children with IQ???70 were analyzed for baseline deficits and treatment-effects on the intention-to-treat-population with generalized-linear-models, principal component analysis and network analysis. Ninety-two children were allocated to treatment and 83 eligible for analyses. Heterogeneous neurocognitive impairments were found that were unaffected by bumetanide treatment. Network analysis showed higher modularity after treatment (mean difference:-0.165, 95%CI:-0.317 to ??0.013,p = .034) and changes in the relative importance of response inhibition in the neurocognitive network (mean difference:-0.037, 95%CI:-0.073 to ??0.001,p = .042). This study offers perspectives to include neurocognitive tests in ASD trials. En ligne : https://doi.org/10.1007/s10803-022-05841-3 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=524 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 Mechanism-based interventions for ASD cannot be implemented using conventional trial designs / Jan J. SPRENGERS in Autism Research, 17-2 (February 2024)
[article]
Titre : Mechanism-based interventions for ASD cannot be implemented using conventional trial designs Type de document : Texte imprimé et/ou numérique Auteurs : Jan J. SPRENGERS, Auteur ; Lisa GEERTJENS, Auteur ; Hilgo BRUINING, Auteur Article en page(s) : p.202-203 Langues : Anglais (eng) Index. décimale : PER Périodiques En ligne : https://doi.org/10.1002/aur.3086 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=522
in Autism Research > 17-2 (February 2024) . - p.202-203[article] Mechanism-based interventions for ASD cannot be implemented using conventional trial designs [Texte imprimé et/ou numérique] / Jan J. SPRENGERS, Auteur ; Lisa GEERTJENS, Auteur ; Hilgo BRUINING, Auteur . - p.202-203.
Langues : Anglais (eng)
in Autism Research > 17-2 (February 2024) . - p.202-203
Index. décimale : PER Périodiques En ligne : https://doi.org/10.1002/aur.3086 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=522