Case report: A severe myositis mimicking bulbar palsy after administration of immune checkpoint inhibitors

ObjectivesImmune Checkpoint Inhibitors (ICI) are nowadays a cornerstone of anti-cancer treatments. However, the wide spectrum of immune-related adverse events (irAEs) represents a challenge in the oncological practice. Our objective is to document rare complications of ICI to help the community of o...

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Main Authors: Philippe Rochigneux, Alexandre Bertucci, Elika Loir, Alexia Mattei, Danielle Robert, Michael Dassa, Brice Chanez, Mikael Ebbo, Lea Gaigne, Anne Sophie Chretien, Giovanni Corazza, Nicolas Schleinitz
Format: Article
Language:English
Published: Frontiers Media S.A. 2025-02-01
Series:Frontiers in Immunology
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Online Access:https://www.frontiersin.org/articles/10.3389/fimmu.2025.1496427/full
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author Philippe Rochigneux
Philippe Rochigneux
Alexandre Bertucci
Elika Loir
Alexia Mattei
Danielle Robert
Michael Dassa
Brice Chanez
Mikael Ebbo
Lea Gaigne
Anne Sophie Chretien
Giovanni Corazza
Giovanni Corazza
Nicolas Schleinitz
author_facet Philippe Rochigneux
Philippe Rochigneux
Alexandre Bertucci
Elika Loir
Alexia Mattei
Danielle Robert
Michael Dassa
Brice Chanez
Mikael Ebbo
Lea Gaigne
Anne Sophie Chretien
Giovanni Corazza
Giovanni Corazza
Nicolas Schleinitz
author_sort Philippe Rochigneux
collection DOAJ
description ObjectivesImmune Checkpoint Inhibitors (ICI) are nowadays a cornerstone of anti-cancer treatments. However, the wide spectrum of immune-related adverse events (irAEs) represents a challenge in the oncological practice. Our objective is to document rare complications of ICI to help the community of onco-immunologists.MethodsWe reported the case of a severe myositis mimicking bulbar palsy treated in our Medical Oncology Department together with Internal Medicine Department. We present the clinical work-up (neurological exam, capillaroscopy) and the diagnostic tests (myositis specific and associated antibodies, nerve conduction study, electromyography) leading to this diagnosis. We also discussed the elimination of differential diagnoses (notably with normal MRI and cerebrospinal fluid analysis) and finally the clinical management of this severe irAE.ResultsA 57 years woman presented multiple sub-diaphragmatic adenopathies related with an advanced melanoma of unknown primary. She started a treatment with Ipilimumab (Ipi, anti CTLA-4) and Nivolumab (Nivo, anti PD-1) and presented at day 10 a grade IV myositis mimicking bulbar palsy with dysphonia, dysarthria and aphagia. In a multidisciplinary setting, she was treated with IV corticosteroids (methylprednisolone 1 mg/kg started at day 10, with a progressive decrease until 1 mg of prednisone in March 2024), IV immunoglobulins started at day 18 (1.5 g/kg in 2 days, administered monthly, with a progressive decrease and a cessation in June 2022), enteral nutrition, speech therapy and physical therapy, with noticeable improvement. After 4 years of follow-up, and only one infusion of Ipi/Nivo, the melanoma is still in complete response.ConclusionWe report an ICI-induced severe myositis mimicking bulbar palsy after the administration of Ipi/Nivo. The diagnosis and clinical care management of this rare complication requires a multi-disciplinary work-up.
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spelling doaj-art-a5ff7ccb04b1483a983afd51301d211f2025-02-10T06:48:31ZengFrontiers Media S.A.Frontiers in Immunology1664-32242025-02-011610.3389/fimmu.2025.14964271496427Case report: A severe myositis mimicking bulbar palsy after administration of immune checkpoint inhibitorsPhilippe Rochigneux0Philippe Rochigneux1Alexandre Bertucci2Elika Loir3Alexia Mattei4Danielle Robert5Michael Dassa6Brice Chanez7Mikael Ebbo8Lea Gaigne9Anne Sophie Chretien10Giovanni Corazza11Giovanni Corazza12Nicolas Schleinitz13Medical Oncology Department, Paoli-Calmettes Institute, Marseille, FranceTeam Immunity and Cancer, Centre de Recherche en Cancérologie de Marseille (CRCM), Inserm, U1068, Centre National de la Recherche Scientifique (CNRS), UMR7258, Paoli-Calmettes Institute, Aix-Marseille University, Marseille, FranceMedical Oncology Department, Paoli-Calmettes Institute, Marseille, FranceMedical Oncology Department, Paoli-Calmettes Institute, Marseille, FranceOtolaryngology Department, Assistance Publique Hôpitaux de Marseille, Aix-Marseille University, Marseille, FranceOtolaryngology Department, Assistance Publique Hôpitaux de Marseille, Aix-Marseille University, Marseille, FranceRadiology Department, Paoli-Calmettes Institute, Marseille, FranceMedical Oncology Department, Paoli-Calmettes Institute, Marseille, FranceInternal Medicine Department, Assistance Publique Hôpitaux de Marseille, Aix-Marseille University, Marseille, FranceInternal Medicine Department, Assistance Publique Hôpitaux de Marseille, Aix-Marseille University, Marseille, FranceTeam Immunity and Cancer, Centre de Recherche en Cancérologie de Marseille (CRCM), Inserm, U1068, Centre National de la Recherche Scientifique (CNRS), UMR7258, Paoli-Calmettes Institute, Aix-Marseille University, Marseille, FranceNeurology Department, Assistance Publique Hôpitaux de Marseille, Aix-Marseille University, Marseille, FranceReferral Centre for Neuromuscular Diseases and Amyotrophic Lateral Sclerosis (ALS), Hôpital La Timone, Marseille, FranceInternal Medicine Department, Assistance Publique Hôpitaux de Marseille, Aix-Marseille University, Marseille, FranceObjectivesImmune Checkpoint Inhibitors (ICI) are nowadays a cornerstone of anti-cancer treatments. However, the wide spectrum of immune-related adverse events (irAEs) represents a challenge in the oncological practice. Our objective is to document rare complications of ICI to help the community of onco-immunologists.MethodsWe reported the case of a severe myositis mimicking bulbar palsy treated in our Medical Oncology Department together with Internal Medicine Department. We present the clinical work-up (neurological exam, capillaroscopy) and the diagnostic tests (myositis specific and associated antibodies, nerve conduction study, electromyography) leading to this diagnosis. We also discussed the elimination of differential diagnoses (notably with normal MRI and cerebrospinal fluid analysis) and finally the clinical management of this severe irAE.ResultsA 57 years woman presented multiple sub-diaphragmatic adenopathies related with an advanced melanoma of unknown primary. She started a treatment with Ipilimumab (Ipi, anti CTLA-4) and Nivolumab (Nivo, anti PD-1) and presented at day 10 a grade IV myositis mimicking bulbar palsy with dysphonia, dysarthria and aphagia. In a multidisciplinary setting, she was treated with IV corticosteroids (methylprednisolone 1 mg/kg started at day 10, with a progressive decrease until 1 mg of prednisone in March 2024), IV immunoglobulins started at day 18 (1.5 g/kg in 2 days, administered monthly, with a progressive decrease and a cessation in June 2022), enteral nutrition, speech therapy and physical therapy, with noticeable improvement. After 4 years of follow-up, and only one infusion of Ipi/Nivo, the melanoma is still in complete response.ConclusionWe report an ICI-induced severe myositis mimicking bulbar palsy after the administration of Ipi/Nivo. The diagnosis and clinical care management of this rare complication requires a multi-disciplinary work-up.https://www.frontiersin.org/articles/10.3389/fimmu.2025.1496427/fullimmune-related myositisbulbar palsyimmune checkpoint inhibitorsimmune toxicityipilimumab/nivolumab
spellingShingle Philippe Rochigneux
Philippe Rochigneux
Alexandre Bertucci
Elika Loir
Alexia Mattei
Danielle Robert
Michael Dassa
Brice Chanez
Mikael Ebbo
Lea Gaigne
Anne Sophie Chretien
Giovanni Corazza
Giovanni Corazza
Nicolas Schleinitz
Case report: A severe myositis mimicking bulbar palsy after administration of immune checkpoint inhibitors
Frontiers in Immunology
immune-related myositis
bulbar palsy
immune checkpoint inhibitors
immune toxicity
ipilimumab/nivolumab
title Case report: A severe myositis mimicking bulbar palsy after administration of immune checkpoint inhibitors
title_full Case report: A severe myositis mimicking bulbar palsy after administration of immune checkpoint inhibitors
title_fullStr Case report: A severe myositis mimicking bulbar palsy after administration of immune checkpoint inhibitors
title_full_unstemmed Case report: A severe myositis mimicking bulbar palsy after administration of immune checkpoint inhibitors
title_short Case report: A severe myositis mimicking bulbar palsy after administration of immune checkpoint inhibitors
title_sort case report a severe myositis mimicking bulbar palsy after administration of immune checkpoint inhibitors
topic immune-related myositis
bulbar palsy
immune checkpoint inhibitors
immune toxicity
ipilimumab/nivolumab
url https://www.frontiersin.org/articles/10.3389/fimmu.2025.1496427/full
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