Case report: First-in-human combined low-dose whole-heart irradiation and high-dose stereotactic arrhythmia radioablation for immunosuppressive refractory cardiac sarcoidosis and ventricular tachycardia

Martijn H van der Ree; Claudia Herrera Siklody; Mathieu Le Bloa; Patrizio Pascale; Alessandra P Porretta; Cheryl C Teres; Jorge Solana Munoz; Adrian Luca; Giulia Domenichini; Mahmut Ozasahin; Raphael Jumeau; Pieter G Postema; Camillo Ribi; Jean Bourhis; Luis Schiappacasse; Etienne Pruvot

doi:10.3389/fcvm.2023.1213165

Case report: First-in-human combined low-dose whole-heart irradiation and high-dose stereotactic arrhythmia radioablation for immunosuppressive refractory cardiac sarcoidosis and ventricular tachycardia

Front Cardiovasc Med. 2023 Jul 21:10:1213165. doi: 10.3389/fcvm.2023.1213165. eCollection 2023.

Authors

Martijn H van der Ree^{1

2

3}, Claudia Herrera Siklody¹, Mathieu Le Bloa¹, Patrizio Pascale¹, Alessandra P Porretta¹, Cheryl C Teres¹, Jorge Solana Munoz¹, Adrian Luca¹, Giulia Domenichini¹, Mahmut Ozasahin⁴, Raphael Jumeau⁴, Pieter G Postema^{2

3}, Camillo Ribi⁵, Jean Bourhis⁴, Luis Schiappacasse⁴, Etienne Pruvot¹

Affiliations

¹ Department of Cardiology, Lausanne University Hospital (CHUV), Lausanne, Switzerland.
² Department of Cardiology, Amsterdam UMC Location University of Amsterdam, Amsterdam, Netherlands.
³ Heart Failure and Arrhythmias, Amsterdam Cardiovascular Sciences, Amsterdam, Netherlands.
⁴ Department of Radiation Oncology, Lausanne University Hospital (CHUV), Lausanne, Switzerland.
⁵ Division of Immunology and Allergy, Department of Medicine, Lausanne University Hospital (CHUV), Lausanne, Switzerland.

Abstract

Background: Cardiac sarcoidosis is associated with heart failure, conduction abnormalities, and life-threatening arrhythmias including ventricular tachycardia (VT). Radiotherapy has been suggested as a treatment for extra-cardiac sarcoidosis in patients refractory to immunomodulatory treatment.

Methods: The effectiveness and safety of low-dose whole-heart radiotherapy for therapy refractory cardiac sarcoidosis were evaluated in a pre- and post-intervention case report comparing the 54 months before and after treatment. Immunomodulatory low-dose whole-heart irradiation as sarcoidosis treatment consisted of a 2 × 2 Gy scheme. Additionally, high-dose single-fraction stereotactic arrhythmia radioablation of 1 × 20 Gy was applied to the pro-arrhythmic region to manage the ventricular tachycardia episodes. Cardiac sarcoidosis disease activity was measured by hypermetabolic areas on repeated fluorodeoxyglucose ([¹⁸F]FDG)-PET/computed tomography (CT) scans and by evaluating changes in ventricular tachycardia episodes before and after treatment.

Results: One patient with therapy refractory progressive cardiac sarcoidosis and recurrent ventricular tachycardia was treated. The cardiac sarcoidosis disease activity showed a durable regression of inflammatory disease activity from 3 months onwards. The [¹⁸F]FDG-PET/CT scan at 54 months did not show any signs of active cardiac sarcoidosis, and a state of remission was achieved. The number of sustained VT episodes was reduced by 95%. We observed that the development of moderate aortic valve regurgitation was likely irradiation-related. No other irradiation-related adverse events occurred, and the left ventricular ejection fraction remained stable.

Conclusion: We report here for the first time on the beneficial and lasting effects of combined immunomodulatory low-dose whole-heart radiotherapy and high-dose stereotactic arrhythmia radioablation in a patient with therapy refractory cardiac sarcoidosis and recurrent VT.

Keywords: cardiac radioablation; cardiac sarcoidosis; heart failure; radiotherapy; stereotactic arrhythmia radioablation (STAR); ventricular tachycardia.

Publication types

Case Reports

Grants and funding

MR has been supported by the Foundation “De Drie Lichten” in The Netherlands. PGP was supported by the Netherlands Heart Foundation (Grant 03-003-2021-T061). The project is part of the STOPSTORM.eu consortium and has also partially received funding from the European Union’s Horizon 2020 Research and Innovation Program under Grant Agreement No 945119.