Bumetanide Effects on Resting-State EEG in Tuberous Sclerosis Complex in Relation to Clinical Outcome: An Open-Label Study.
EEG
bumetanide
excitation-inhibition balance
irritability
open-label study
repetitive behavior
tuberous sclerosis complex (TSC)
Journal
Frontiers in neuroscience
ISSN: 1662-4548
Titre abrégé: Front Neurosci
Pays: Switzerland
ID NLM: 101478481
Informations de publication
Date de publication:
2022
2022
Historique:
received:
19
02
2022
accepted:
15
04
2022
entrez:
1
6
2022
pubmed:
2
6
2022
medline:
2
6
2022
Statut:
epublish
Résumé
Neuronal excitation-inhibition (E/I) imbalances are considered an important pathophysiological mechanism in neurodevelopmental disorders. Preclinical studies on tuberous sclerosis complex (TSC), suggest that altered chloride homeostasis may impair GABAergic inhibition and thereby E/I-balance regulation. Correction of chloride homeostasis may thus constitute a treatment target to alleviate behavioral symptoms. Recently, we showed that bumetanide-a chloride-regulating agent-improved behavioral symptoms in the open-label study Bumetanide to Ameliorate Tuberous Sclerosis Complex Hyperexcitable Behaviors trial (BATSCH trial; Eudra-CT: 2016-002408-13). Here, we present resting-state EEG as secondary analysis of BATSCH to investigate associations between EEG measures sensitive to network-level changes in E/I balance and clinical response to bumetanide. EEGs of 10 participants with TSC (aged 8-21 years) were available. Spectral power, long-range temporal correlations (LRTC), and functional E/I ratio ( EU Clinical Trial Register, EudraCT 2016-002408-13 (www.clinicaltrialsregister.eu/ctr-search/trial/2016-002408-13/NL). Registered 25 July 2016.
Identifiants
pubmed: 35645706
doi: 10.3389/fnins.2022.879451
pmc: PMC9134117
doi:
Types de publication
Journal Article
Langues
eng
Pagination
879451Informations de copyright
Copyright © 2022 Juarez-Martinez, van Andel, Sprengers, Avramiea, Oranje, Scheepers, Jansen, Mansvelder, Linkenkaer-Hansen and Bruining.
Déclaration de conflit d'intérêts
KL-H is shareholder of NBT Analytics BV, which provides EEG-analysis services for clinical trials. HB and KL-H are shareholders of Aspect Neuroprofiles BV, which develops physiology-informed prognostic measures for neurodevelopmental disorders. KL-H has filed the patent claim (PCT/NL2019/050167) “Method of determining brain activity”; with priority date 16 March 2018. The remaining authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.
Références
Curr Opin Psychiatry. 2015 Mar;28(2):113-20
pubmed: 25602245
Transl Psychiatry. 2012 Dec 11;2:e202
pubmed: 23233021
Ann Neurol. 2001 Jan;49(1):67-78
pubmed: 11198298
Neuroimage. 2004 Sep;23(1):364-9
pubmed: 15325384
Epilepsia. 2010 Dec;51(12):2440-8
pubmed: 20887366
Biol Psychiatry Cogn Neurosci Neuroimaging. 2023 Mar;8(3):251-261
pubmed: 34506972
Nat Med. 2015 Dec;21(12):1436-44
pubmed: 26594844
Neurotherapeutics. 2014 Apr;11(2):251-68
pubmed: 24481729
Curr Opin Neurobiol. 2012 Oct;22(5):895-901
pubmed: 22560338
Front Neurol. 2017 Sep 08;8:469
pubmed: 28943860
Brain. 2000 Sep;123 ( Pt 9):1838-44
pubmed: 10960047
Neuropharmacology. 2020 Jan 1;162:107754
pubmed: 31476353
Front Physiol. 2021 Dec 23;12:775172
pubmed: 35002760
Ann Neurol. 2013 Nov;74(5):679-87
pubmed: 23798472
Front Neurosci. 2019 Apr 24;13:310
pubmed: 31068771
Brain Dev. 2012 Jun;34(6):478-86
pubmed: 21959128
Neuron. 2015 Aug 19;87(4):684-98
pubmed: 26291155
Am J Ment Defic. 1985 Mar;89(5):485-91
pubmed: 3993694
Mol Autism. 2020 May 6;11(1):29
pubmed: 32375878
Lancet. 2008 Aug 23;372(9639):657-68
pubmed: 18722871
Transl Psychiatry. 2020 Jan 27;10(1):9
pubmed: 32066666
Elife. 2020 Apr 27;9:
pubmed: 32324137
Dev Med Child Neurol. 2022 Apr;64(4):495-501
pubmed: 34601720
Trends Neurosci. 2017 Sep;40(9):536-554
pubmed: 28818303
Nat Neurosci. 2020 Dec;23(12):1655-1665
pubmed: 33230329
Neurotherapeutics. 2010 Jul;7(3):275-82
pubmed: 20643380
Sci Rep. 2020 Jun 8;10(1):9195
pubmed: 32513931
J Autism Dev Disord. 2003 Aug;33(4):427-33
pubmed: 12959421
Epilepsia. 2014 Jun;55(6):806-18
pubmed: 24802699
Epilepsia. 2013 Jan;54(1):e9-12
pubmed: 23061490
JAMA Netw Open. 2020 Jan 3;3(1):e1918377
pubmed: 31899530
Autism Res. 2019 Dec;12(12):1758-1773
pubmed: 31419043
J Neurodev Disord. 2013 Sep 16;5(1):24
pubmed: 24040879
J Child Neurol. 2009 May;24(5):572-6
pubmed: 19406757
PLoS One. 2012;7(6):e39127
pubmed: 22745707
Brain Topogr. 2017 Nov;30(6):810-821
pubmed: 28905146
Pediatr Neurol. 2013 Oct;49(4):243-54
pubmed: 24053982
J Am Acad Child Adolesc Psychiatry. 2021 Jul;60(7):865-876
pubmed: 32730977
N Engl J Med. 2014 Mar 27;370(13):1209-1219
pubmed: 24670167
Science. 2014 Feb 7;343(6171):675-9
pubmed: 24503856
Pediatr Neurol. 2015 Jan;52(1):25-35
pubmed: 25532776
Stem Cell Reports. 2019 Jan 8;12(1):42-56
pubmed: 30581017
Nat Biotechnol. 2020 Apr;38(4):439-447
pubmed: 32042166
Ann Neurol. 2005 Sep;58(3):466-9
pubmed: 16130096
Neurobiol Dis. 2016 Nov;95:93-101
pubmed: 27425893
J Child Neurol. 2004 Sep;19(9):666-74
pubmed: 15563012
Transl Psychiatry. 2017 Mar 14;7(3):e1056
pubmed: 28291262
Ann Neurol. 2012 Apr;71(4):539-51
pubmed: 22447678
Neuron. 2013 May 8;78(3):510-22
pubmed: 23664616
Epilepsy Behav. 2011 Apr;20(4):700-5
pubmed: 21439912
Clin Neurophysiol. 2021 Jan;132(1):25-35
pubmed: 33248432
Neurology. 2019 Jul 9;93(2):e200-e209
pubmed: 31217257
Epilepsia. 2011 Mar;52(3):467-76
pubmed: 21204828
Mol Autism. 2020 Feb 19;11(1):16
pubmed: 32075691
J Neurosci. 2001 Feb 15;21(4):1370-7
pubmed: 11160408
Epilepsy Res. 2010 Aug;90(3):228-33
pubmed: 20554157
J Neurosci. 2012 Jul 18;32(29):9817-23
pubmed: 22815496
Front Comput Neurosci. 2018 Apr 04;12:23
pubmed: 29670518
J Autism Dev Disord. 2007 May;37(5):855-66
pubmed: 17048092
Front Neurol. 2020 Oct 16;11:582891
pubmed: 33178126
JAMA Netw Open. 2020 Jun 1;3(6):e206653
pubmed: 32568399
Cereb Cortex. 2007 Jun;17(6):1386-93
pubmed: 16908492
Front Physiol. 2012 Nov 30;3:450
pubmed: 23226132
Chaos. 1995;5(1):82-7
pubmed: 11538314
Trends Neurosci. 2007 Apr;30(4):150-8
pubmed: 17307258
Mol Autism. 2020 May 7;11(1):30
pubmed: 32381101
Pediatr Neurol. 2014 Apr;50(4):290-6
pubmed: 24486221
Ann Neurol. 2008 Feb;63(2):222-35
pubmed: 17918265
Neuropharmacology. 2013 Jun;69:62-74
pubmed: 22705273
Ann Neurol. 2007 Feb;61(2):139-52
pubmed: 17279540
Curr Opin Neurol. 2015 Apr;28(2):110-6
pubmed: 25710286
Ann Clin Transl Neurol. 2017 Nov 12;4(12):877-887
pubmed: 29296616
Neuroimage. 2009 Oct 1;47(4):1207-14
pubmed: 19481612
J Int Neuropsychol Soc. 2009 May;15(3):331-43
pubmed: 19402919
Cereb Cortex. 2012 Sep;22(9):2111-9
pubmed: 22021912
Neuroimage Clin. 2020;25:102145
pubmed: 31911342
CNS Drugs. 2019 Mar;33(3):283-291
pubmed: 30784026
Curr Biol. 2014 Dec 15;24(24):2878-87
pubmed: 25454585
Pediatr Neurol. 2015 Mar;52(3):281-9
pubmed: 25591831
Pediatr Neurol. 2016 Aug;61:28-37
pubmed: 27255413
Clin Neurophysiol. 2012 May;123(5):883-91
pubmed: 22000706
Nat Rev Neurosci. 2004 Feb;5(2):97-107
pubmed: 14735113