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

TITLE=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

JOURNAL=Frontiers in Cardiovascular Medicine

VOLUME=Volume 10 - 2023

YEAR=2023

URL=https://www.frontiersin.org/journals/cardiovascular-medicine/articles/10.3389/fcvm.2023.1213165

DOI=10.3389/fcvm.2023.1213165

ISSN=2297-055X

ABSTRACT=Cardiac sarcoidosis is associated with heart failure, conduction abnormalities and life-threatening arrhythmias including ventricular tachycardia. Radiotherapy has been suggested as treatment for extra-cardiac sarcoidosis in patients refractory to immunomodulatory treatment.The effectiveness and safety of low-dose whole-heart radiotherapy for therapy-refractory cardiac sarcoidosis was evaluated in a pre-post intervention case report comparing the 54 months before and after treatment. Immunomodulatory low-dose whole-heart irradiation as sarcoidosis treatment consisted of a 2x2 Gy scheme. Additionally, high-dose single fraction Stereotactic Arrhythmia Radioablation (STAR) of 1x20 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 [ 18 F]FDG-PET/CT scans and by evaluating changes in ventricular tachycardia episodes before and after treatment.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 [ 18 F]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 the development of moderate aortic valve regurgitation likely irradiation-related. No other irradiation-related adverse events occurred and the left ventricular ejection fraction remained stable.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.