An improved electrophysiological cellular assay to unlock the pharmacological modulation of the NALCN channelosome

The sodium leak channel non-selective (NALCN) is a key regulator of resting membrane potential and cellular excitability in neurons and endocrine cells. Gain-of-function de novo pathogenic variants in NALCN cause severe neurodevelopmental disorders with a broad and heterogeneous clinical spectrum. Partial inhibition of NALCN has been proposed as a therapeutic strategy; however, progress has been limited by the absence of selective pharmacological modulators. This gap largely reflects the lack of a robust heterologous expression system suitable for high-throughput screening, as functional NALCN requires multiple ancillary subunits and its constitutive expression is toxic in commonly used cell lines such as HEK293. To address these challenges, we developed a multitransposon-based approach to generate inducible HEK293 cell lines that stably express the complete NALCN channelosome, including wild-type and disease-associated variants. We further demonstrate that NALCN current expression is cell cycle-dependent, enabling the definition of optimized conditions for consistent and reproducible electrophysiological recordings. Using these cell lines, we conducted a systematic pharmacological characterization of the NALCN channelosome by patch-clamp electrophysiology and identified several candidate modulators that are currently under evaluation. Notably, we revisited NALCN modulation by N-benzhydryl quinuclidine compounds and found that these compounds can restore locomotor phenotypes in an animal model of NALCN gain-of-function. Together, this work establishes a foundational platform for the discovery of NALCN-targeting compounds and opens new therapeutic avenues not only for NALCN-associated neurodevelopmental diseases, but also potentially for psychiatric disorders, chronic pain, and cancer.

Authors

Institutions

Publication Details

Journal
Biomedicine & Pharmacotherapy
Published
2026-05-01
DOI
https://doi.org/10.1016/j.biopha.2026.119458
Primary Topic
Ion channel regulation and function
Type
article
Field-Weighted Citation Impact
0.00

Funders

Controls
|||
ALL TIME
JAN
FEB
MAR
APR
MAY
JUN
JUL
AUG
SEP
OCT
article

An improved electrophysiological cellular assay to unlock the pharmacological modulation of the NALCN channelosome

Philippe Lory, Jeremy A. Tanner, Nattaya Thongsepee, Arnaud Monteil et al.
Biomedicine & Pharmacotherapy
Ion channel regulation and function
article

An improved electrophysiological cellular assay to unlock the pharmacological modulation of the NALCN channelosome

Philippe Lory, Jeremy A. Tanner, Nattaya Thongsepee, Arnaud Monteil, Donard S. Dwyer, Isabel del Pino, Alexandra Typou, Romain Baudat
article en

Abstract

The sodium leak channel non-selective (NALCN) is a key regulator of resting membrane potential and cellular excitability in neurons and endocrine cells. Gain-of-function de novo pathogenic variants in NALCN cause severe neurodevelopmental disorders with a broad and heterogeneous clinical spectrum. Partial inhibition of NALCN has been proposed as a therapeutic strategy; however, progress has been limited by the absence of selective pharmacological modulators. This gap largely reflects the lack of a robust heterologous expression system suitable for high-throughput screening, as functional NALCN requires multiple ancillary subunits and its constitutive expression is toxic in commonly used cell lines such as HEK293. To address these challenges, we developed a multitransposon-based approach to generate inducible HEK293 cell lines that stably express the complete NALCN channelosome, including wild-type and disease-associated variants. We further demonstrate that NALCN current expression is cell cycle-dependent, enabling the definition of optimized conditions for consistent and reproducible electrophysiological recordings. Using these cell lines, we conducted a systematic pharmacological characterization of the NALCN channelosome by patch-clamp electrophysiology and identified several candidate modulators that are currently under evaluation. Notably, we revisited NALCN modulation by N-benzhydryl quinuclidine compounds and found that these compounds can restore locomotor phenotypes in an animal model of NALCN gain-of-function. Together, this work establishes a foundational platform for the discovery of NALCN-targeting compounds and opens new therapeutic avenues not only for NALCN-associated neurodevelopmental diseases, but also potentially for psychiatric disorders, chronic pain, and cancer.

Biomedicine & PharmacotherapyVol. 199
Thammasat University (TH), Universitat de Miguel Hernández d'Elx (ES), Centre National de la Recherche Scientifique (FR), Siriraj Hospital (TH), Inserm (FR), The University of Texas at San Antonio Health Science Center (US), Institute for Neurodegenerative Disorders (US), Institut de Génomique Fonctionnelle (FR), Louisiana State University Health Sciences Center Shreveport (US)
Fondation Maladies Rares, Agence Nationale de la Recherche, National Institute on Aging
Good health and well-being
Openalex Percentile: Top 13%
Ion channel regulation and function
AI Navigator

Ask Laika to Summarize, Analyze, and Connect papers live on the map.

Summarize Papers & Methodologies

Extract key findings, datasets, and comparative methods across publications.

Benchmark Rankings & Visual Analytics

Rank top research institutions, authors, funders, topics, and journals by Field-Weighted Citation Impact (FWCI) and paper volume with instant charts.

Connect Distant Disciplines

Bridge topological clusters on the map to find hidden collaborative intersections.