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Open Access 23-12-2023 | Checkpoint Inhibitors | Original Communication

Immune checkpoint inhibitor-related myositis and myocarditis: diagnostic pitfalls and imaging contribution in a real-world, institutional case series

Authors: Alex Vicino, Andreas F. Hottinger, Sofiya Latifyan, Sarah Boughdad, Fabio Becce, John O. Prior, Thierry Kuntzer, Jean-Philippe Brouland, Vincent Dunet, Michel Obeid, Marie Théaudin

Published in: Journal of Neurology | Issue 4/2024

Abstract

Background

Immune checkpoint inhibitors (ICIs) are reshaping the prognosis of many cancers, but often cause immune-related adverse events (irAEs). Among neurological irAEs, myositis is the most frequently reported. Our aim is to describe clinical and non-clinical characteristics, treatment and outcome of all irMyositis (skeletal limb-girdle and/or ocular myositis) and irMyocarditis cases in our reference center.

Methods

We retrospectively enrolled all irMyositis/irMyocarditis patients seen between 2018 and 2022. We reviewed demographics, clinical characteristics, biological, neurophysiological, imaging workup, treatment and outcome.

Results

We included 14 consecutive patients. The most frequent treatments were pembrolizumab (35%) or ipilimumab–nivolumab combination (35%). Limb-girdle, ocular (non-fluctuating palpebral ptosis and/or diplopia with or without ophthalmoparesis) and cardiac phenotypes were equally distributed, overlapping in 40% of cases. Ocular involvement was frequently misdiagnosed; review of brain MRIs disclosed initially missed signs of skeletal myositis in one patient and ocular myositis in 3. Seven patients had other co-existing irAEs. When performed, myography showed a myogenic pattern. CK was elevated in 8/15 patients, troponin-T in 12/12 and troponin-I in 7/9 tested patients. ICI were discontinued in all cases, with further immunosuppressive treatment in nine patients. In most cases, neurological and cardiological outcome was good at last follow-up.

Conclusion

Myositis is a potentially severe irAE. Despite its heterogeneous presentation, some highly suggestive clinical symptoms, such as ocular involvement, or radiological signs should raise physicians’ attention to avoid misdiagnosis. We thus recommend a multidisciplinary assessment (including complete neuromuscular evaluation) even in case of isolated myocarditis. Our series underlines the importance of an early diagnosis, since suspension of ICI and adequate treatment are usually associated with good functional outcome.

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Title: Immune checkpoint inhibitor-related myositis and myocarditis: diagnostic pitfalls and imaging contribution in a real-world, institutional case series
Authors: Alex Vicino
Andreas F. Hottinger
Sofiya Latifyan
Sarah Boughdad
Fabio Becce
John O. Prior
Thierry Kuntzer
Jean-Philippe Brouland
Vincent Dunet
Michel Obeid
Marie Théaudin
Publication date: 23-12-2023
Publisher: Springer Berlin Heidelberg
Keywords: Checkpoint Inhibitors
Myocarditis
Diplopia
Published in: Journal of Neurology / Issue 4/2024
Print ISSN: 0340-5354
Electronic ISSN: 1432-1459
DOI: https://doi.org/10.1007/s00415-023-12134-x

Keynote webinar | Spotlight on medication adherence

Springer Medicine

Immune checkpoint inhibitor-related myositis and myocarditis: diagnostic pitfalls and imaging contribution in a real-world, institutional case series

Abstract

Background

Methods

Results

Conclusion

Keynote webinar | Spotlight on medication adherence

Springer Medicine

Abstract

Background

Methods

Results

Conclusion

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