Literature DB >> 26594844

Treatment during a vulnerable developmental period rescues a genetic epilepsy.

Stephan Lawrence Marguet1,2,3, Vu Thao Quyen Le-Schulte3, Andrea Merseburg1,2,3, Axel Neu3, Ronny Eichler3, Igor Jakovcevski1,2, Anton Ivanov4, Ileana Livia Hanganu-Opatz5, Christophe Bernard4, Fabio Morellini3,6, Dirk Isbrandt1,2,3.   

Abstract

The nervous system is vulnerable to perturbations during specific developmental periods. Insults during such susceptible time windows can have long-term consequences, including the development of neurological diseases such as epilepsy. Here we report that a pharmacological intervention timed during a vulnerable neonatal period of cortical development prevents pathology in a genetic epilepsy model. By using mice with dysfunctional Kv7 voltage-gated K(+) channels, which are mutated in human neonatal epilepsy syndromes, we demonstrate the safety and efficacy of the sodium-potassium-chloride cotransporter NKCC1 antagonist bumetanide, which was administered during the first two postnatal weeks. In Kv7 current-deficient mice, which normally display epilepsy, hyperactivity and stereotypies as adults, transient bumetanide treatment normalized neonatal in vivo cortical network and hippocampal neuronal activity, prevented structural damage in the hippocampus and restored wild-type adult behavioral phenotypes. Furthermore, bumetanide treatment did not adversely affect control mice. These results suggest that in individuals with disease susceptibility, timing prophylactically safe interventions to specific windows during development may prevent or arrest disease progression.

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Year:  2015        PMID: 26594844     DOI: 10.1038/nm.3987

Source DB:  PubMed          Journal:  Nat Med        ISSN: 1078-8956            Impact factor:   53.440


  38 in total

1.  Neurodevelopmental disorders: Preventative treatment for infantile epilepsy.

Authors:  Megan Cully
Journal:  Nat Rev Drug Discov       Date:  2016-01       Impact factor: 84.694

2.  A limited role of NKCC1 in telencephalic glutamatergic neurons for developing hippocampal network dynamics and behavior.

Authors:  Jürgen Graf; Chuanqiang Zhang; Stephan Lawrence Marguet; Tanja Herrmann; Tom Flossmann; Robin Hinsch; Vahid Rahmati; Madlen Guenther; Christiane Frahm; Anja Urbach; Ricardo Melo Neves; Otto W Witte; Stefan J Kiebel; Dirk Isbrandt; Christian A Hübner; Knut Holthoff; Knut Kirmse
Journal:  Proc Natl Acad Sci U S A       Date:  2021-04-06       Impact factor: 11.205

3.  Developmental timing and critical windows for the treatment of psychiatric disorders.

Authors:  Oscar Marín
Journal:  Nat Med       Date:  2016-10-26       Impact factor: 53.440

4.  Nurture Trumps Nature in a Model of Heritable Epilepsy: Will Bumetanide Be the First Antiepileptogenic Drug?

Authors:  Robert J Heuermann; Dane Chetkovich
Journal:  Epilepsy Curr       Date:  2016 May-Jun       Impact factor: 7.500

5.  Disturbed Prefrontal Cortex Activity in the Absence of Schizophrenia-Like Behavioral Dysfunction in Arc/Arg3.1 Deficient Mice.

Authors:  Xiaoyan Gao; Jasper Grendel; Mary Muhia; Sergio Castro-Gomez; Ute Süsens; Dirk Isbrandt; Matthias Kneussel; Dietmar Kuhl; Ora Ohana
Journal:  J Neurosci       Date:  2019-09-05       Impact factor: 6.167

6.  Epilepsy. Early treatment could prevent genetic epilepsy in mice.

Authors:  Hemi Malkki
Journal:  Nat Rev Neurol       Date:  2015-12-16       Impact factor: 42.937

7.  Cannabinoid Exposure via Lactation in Rats Disrupts Perinatal Programming of the Gamma-Aminobutyric Acid Trajectory and Select Early-Life Behaviors.

Authors:  Andrew F Scheyer; Milene Borsoi; Jim Wager-Miller; Anne-Laure Pelissier-Alicot; Michelle N Murphy; Ken Mackie; Olivier J J Manzoni
Journal:  Biol Psychiatry       Date:  2019-09-05       Impact factor: 13.382

8.  Developmental HCN channelopathy results in decreased neural progenitor proliferation and microcephaly in mice.

Authors:  Anna Katharina Schlusche; Sabine Ulrike Vay; Niklas Kleinenkuhnen; Steffi Sandke; Rafael Campos-Martín; Marta Florio; Wieland Huttner; Achim Tresch; Jochen Roeper; Maria Adele Rueger; Igor Jakovcevski; Malte Stockebrand; Dirk Isbrandt
Journal:  Proc Natl Acad Sci U S A       Date:  2021-08-31       Impact factor: 11.205

Review 9.  Commonalities in epileptogenic processes from different acute brain insults: Do they translate?

Authors:  Pavel Klein; Raymond Dingledine; Eleonora Aronica; Christophe Bernard; Ingmar Blümcke; Detlev Boison; Martin J Brodie; Amy R Brooks-Kayal; Jerome Engel; Patrick A Forcelli; Lawrence J Hirsch; Rafal M Kaminski; Henrik Klitgaard; Katja Kobow; Daniel H Lowenstein; Phillip L Pearl; Asla Pitkänen; Noora Puhakka; Michael A Rogawski; Dieter Schmidt; Matti Sillanpää; Robert S Sloviter; Christian Steinhäuser; Annamaria Vezzani; Matthew C Walker; Wolfgang Löscher
Journal:  Epilepsia       Date:  2017-12-15       Impact factor: 5.864

Review 10.  The Drosophila Larval Locomotor Circuit Provides a Model to Understand Neural Circuit Development and Function.

Authors:  Iain Hunter; Bramwell Coulson; Aref Arzan Zarin; Richard A Baines
Journal:  Front Neural Circuits       Date:  2021-07-01       Impact factor: 3.492

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