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bioRxiv · 10.1101/2025.10.23.684089

Exploring light chain cardiotoxicity in AL amyloidosis: Impact on hiPSC-derived Cardiomyocyte Activity

Abstract

AimsImmunoglobulin light chain (AL) amyloidosis is a protein misfolding disease characterized by the systemic deposition of amyloid fibrils derived from monoclonal light chains (LCs). Cardiac involvement is the major determinant of prognosis and mortality, and beyond fibril accumulation, soluble cardiotoxic LCs play a critical role in disease progression. While current in vivo models like C. elegans and murine systems have demonstrated LC toxicity, they lack human relevance or fail to capture soluble LC-induced cardiotoxicity. This study aimed to characterize the electrophysiological effects of cardiotoxic LCs on a human-relevant model using human-induced pluripotent stem cell-derived cardiomyocytes (hiPSC-CMs). Methods and ResultsTwo amyloidogenic cardiotoxic LCs (H3 and H6) from AL patients and one non-cardiotoxic LC (M10) from a multiple myeloma patient were biophysically characterized and tested in hiPSC-CMs at clinically relevant concentrations. Electrophysiological recordings revealed that H3 and H6 significantly reduced spontaneous action potential (AP) firing frequency and maximal upstroke velocity (dV/dt) in hiPSC-CMs, indicating impaired excitability. H6 also shortened AP duration. H3 exposure led to a [~]40% reduction in peak sodium current density and altered inactivation kinetics of the L-type calcium current, without affecting major pacemaker or repolarizing potassium (IKr or IKs) currents. In contrast, M10 had no effect on any measured parameter, validating the models ability to discriminate toxic from non-toxic LCs. ConclusionThis study demonstrates that hiPSC-CMs provide a clinically relevant human model to investigate LC-induced cardiotoxicity. Cardiotoxic LCs exert distinct but converging electrophysiological impairments, including disruption of sodium and L-type calcium currents, contributing to reduced excitability and altered AP morphology. These findings provide mechanistic insights into AL amyloidosis-related cardiac dysfunction and establish a foundation for future therapeutic screening targeting soluble LC toxicity in a human context.

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BibTeXRIS

Calamaio, S., Frosio, A., Melgari, D., Broggini, L., Sonzini, F., Prevostini, R., Anastasia, L., Pappone, C., Nuvolone, M., Palladini, G., Ricagno, S., Rivolta, I.. 2025-10-24. Exploring light chain cardiotoxicity in AL amyloidosis: Impact on hiPSC-derived Cardiomyocyte Activity. https://doi.org/10.1101/2025.10.23.684089

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