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
- Philippe Lory (ORCID: https://orcid.org/0000-0002-1638-7604)
- Jeremy A. Tanner (ORCID: https://orcid.org/0000-0001-9828-1776)
- Nattaya Thongsepee (ORCID: https://orcid.org/0000-0001-7034-8756)
- Arnaud Monteil (ORCID: https://orcid.org/0000-0003-1632-7318)
- Donard S. Dwyer (ORCID: https://orcid.org/0000-0002-4201-0958)
- Isabel del Pino (ORCID: https://orcid.org/0000-0002-8672-926X)
- Alexandra Typou
- Romain Baudat
Institutions
- 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)
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
- Fondation Maladies Rares
- Agence Nationale de la Recherche
- National Institute on Aging