Functional, Cohort-Level Assessment of CFTR Modulator Responses Using Biobanked Nasal Epithelial Cells from Individuals with Cystic Fibrosis

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Authors: Bente L. Aalbers, Gimano D. Amatngalim, Ellen Aarts, Lisa W. Rodenburg, Loes A. Oosterhoff, Harry G.M. Heijerman, Jeffrey Matthijn Beekman
Cited by: 0
References: 21

Abstract

Background/Objectives: Individual responses to CFTR modulators vary widely among people with cystic fibrosis (pwCF), underscoring the need for functional approaches that provide biological context alongside genotype-based therapy selection. Nasal epithelial cultures provide an individual-specific model for theratyping, but most studies rely on freshly isolated cells, restricting repeated testing and long-term sample use. In this study, we tested whether CFTR modulator responses measured in biobanked nasal cells were associated with real-world clinical outcomes. Methods: Cryopreserved nasal epithelial cells from 23 pwCF were differentiated at the air–liquid interface and assessed for CFTR modulator-responsive ion transport using Ussing chambers. In vitro responses were correlated with 6-month changes in sweat chloride concentration (SCC), FEV1, and BMI. Results: Cryopreserved cultures retained donor-specific CFTR modulator responsiveness. Modulator-induced forskolin/IBMX-stimulated currents correlated with changes in SCC (R = −0.512). CFTR inhibitor-sensitive currents correlated with FEV1 (R = 0.564). Associations between forskolin/IBMX-stimulated currents and FEV1 were positive but did not reach statistical significance using two-tailed analysis. BMI changes showed no significant association. Conclusions: Biobanked nasal epithelial cultures preserve clinically relevant CFTR modulator responses at the cohort level, supporting their use as functional assays for population-level assessment in cystic fibrosis. This cryopreservation-based strategy enables repeated testing and may expand access to theratyping beyond freshly obtained samples.

Fields of Study & Subjects

Cystic Fibrosis Research Advances Inhalation and Respiratory Drug Delivery Genetic and Kidney Cyst Diseases

Publication details

Journal / source
Journal of Personalized Medicine
Volume
16
Issue
1
Pages
51–51
Publication date
15 January 2026
Language
EN