Dravet Syndrome Patient-Derived Neural Cells Present Altered Levels of Potassium, Copper, and Zinc

Dravet syndrome (DS), also known as severe myoclonic epilepsy of infancy (SMEI), is an intractable epilepsy syndrome. Most cases are associated with mutations in the SCN1A gene, which is responsible for the expression of the sodium voltage-gated channel alpha subunit 1, Nav1.1. These mutations lead to altered neuronal firing and a state of hyperexcitability. DS has been studied using patient samples, animal models, and more recently, iPS cells derived from DS patients. In this work, we sought to understand the impact that Nav1.1 loss-of-function has on the elementary chemical constitution of DS patient-derived neural cells. iPS cells from DS patients and controls were differentiated into neural-induced spheroids, and synchrotron X-ray radiation was used to assess alterations in their elemental concentration. We observed that DS-derived neural cells present elevated levels of potassium (K), copper (Cu), and zinc (Zn). These findings suggest that an elemental imbalance may be involved in the pathogenesis of DS, as higher levels of K, Cu, and Zn have been implicated in seizure episodes and epilepsy. We conclude that modeling DS using cell reprogramming is a relevant approach to understanding the basic mechanisms involved in this disease and perhaps provide novel treatment strategies.

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Publication Details

Journal
ACS Chemical Neuroscience
Published
2025-12-24
DOI
https://doi.org/10.1021/acschemneuro.5c00898
Primary Topic
Epilepsy research and treatment
Type
article
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article

Dravet Syndrome Patient-Derived Neural Cells Present Altered Levels of Potassium, Copper, and Zinc

Gabriela Vitória, Mariana P. Stelling, Stevens K. Rehen, Simone C. Cardoso et al.
ACS Chemical Neuroscience
Epilepsy research and treatment
article

Dravet Syndrome Patient-Derived Neural Cells Present Altered Levels of Potassium, Copper, and Zinc

Gabriela Vitória, Mariana P. Stelling, Stevens K. Rehen, Simone C. Cardoso, Sylvie Devalle, Marília Z. P. Guimarães, Rafaela C. Sartore
article en

Abstract

Dravet syndrome (DS), also known as severe myoclonic epilepsy of infancy (SMEI), is an intractable epilepsy syndrome. Most cases are associated with mutations in the SCN1A gene, which is responsible for the expression of the sodium voltage-gated channel alpha subunit 1, Nav1.1. These mutations lead to altered neuronal firing and a state of hyperexcitability. DS has been studied using patient samples, animal models, and more recently, iPS cells derived from DS patients. In this work, we sought to understand the impact that Nav1.1 loss-of-function has on the elementary chemical constitution of DS patient-derived neural cells. iPS cells from DS patients and controls were differentiated into neural-induced spheroids, and synchrotron X-ray radiation was used to assess alterations in their elemental concentration. We observed that DS-derived neural cells present elevated levels of potassium (K), copper (Cu), and zinc (Zn). These findings suggest that an elemental imbalance may be involved in the pathogenesis of DS, as higher levels of K, Cu, and Zn have been implicated in seizure episodes and epilepsy. We conclude that modeling DS using cell reprogramming is a relevant approach to understanding the basic mechanisms involved in this disease and perhaps provide novel treatment strategies.

ACS Chemical NeuroscienceVol. 17(1)
Universidade Federal do Rio de Janeiro (BR), Instituto Federal do Rio de Janeiro (BR), D’Or Institute for Research and Education (BR), National Institute of Traumatology & Orthopaedic Rehabilitation (BD)
Good health and well-being
Openalex Percentile: Top 34%
Epilepsy research and treatment
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