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Checkpoint inhibitor-induced myocarditis and myasthenia gravis in a recurrent/metastatic thymic carcinoma patient: a case report.

ABSTRACT: Background:Pembrolizumab, an immune checkpoint inhibitor (ICI), is an IgG4 antibody that blocks interaction between programmed cell death protein 1 and programmed death-ligand 1. Myocarditis, an immune-related adverse event, has been reported in thymic epithelial tumours. Pembrolizumab has also been associated with development/exacerbation of myasthenia gravis (MG). Case summary:A 70-year-old woman with metastatic thymic cancer presented to the hospital with shortness of breath, 21?days after initiation of pembrolizumab. She was diagnosed with ICI-related myocarditis and was subsequently intubated due to respiratory failure. A dual-chamber pacemaker was placed due to complete heart block with asystole. Her troponin levels were elevated, an electrocardiogram was suspicious for myocardial infarction, but coronary angiogram revealed normal coronary arteries and endomyocardial biopsy confirmed the presence of myocarditis. Treatment was started with high-dose intravenous methylprednisolone and cardiovascular status improved. However, the patient was unable to be weaned from mechanical ventilation and tested positive for acetylcholine receptor binding/blocking antibodies due to de novo MG. After 50?days of hospitalization, she was discharged home in stable condition. A computed tomography scan was performed 6?weeks after pembrolizumab; results showed significant decrease/resolution of all measurable sites of metastatic disease in the lungs. Discussion:This is the first reported case of a patient developing single-agent pembrolizumab-induced myocarditis concomitant with new-onset MG after treatment for advanced thymic malignancy. Additional studies are needed to explore the association between myocarditis, MG, and ICI therapy.

SUBMITTER: Szuchan C

PROVIDER: S-EPMC7319805 | biostudies-literature | 2020 Jun

REPOSITORIES: biostudies-literature

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Checkpoint inhibitor-induced myocarditis and myasthenia gravis in a recurrent/metastatic thymic carcinoma patient: a case report.

Szuchan Charles C Elson Leah L Alley Evan E Leung Kevin K Camargo Antonio Lewis AL Elimimian Elizabeth E Nahleh Zeina Z Sadler Diego D

European heart journal. Case reports 20200407 3

<h4>Background</h4>Pembrolizumab, an immune checkpoint inhibitor (ICI), is an IgG4 antibody that blocks interaction between programmed cell death protein 1 and programmed death-ligand 1. Myocarditis, an immune-related adverse event, has been reported in thymic epithelial tumours. Pembrolizumab has also been associated with development/exacerbation of myasthenia gravis (MG).<h4>Case summary</h4>A 70-year-old woman with metastatic thymic cancer presented to the hospital with shortness of breath, 2 ...[more]

PMID: 32617460

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Project description:BackgroundThe development of immune checkpoint inhibitors (ICIs) represents a paradigm shift in the treatment of cancers. Despite showing remarkable efficacy, these agents can be associated with life-threatening immune-related adverse events. In recent years, several cases of myocarditis with myositis and/or myasthenia gravis overlap syndrome (IM3OS) have been reported. However, given the rarity, the clinical features and outcomes of these cases remain poorly understood. We, therefore, attempted to systematically review and summarize all cases of IM3OS reported in the literature.Materials and methodsStudies reporting IM3OS were identified in Embase and MEDLINE. Only case reports and case series published in journals or presented at conferences were included. We conducted a systematic review according to the PRISMA Harms guidelines.ResultsA total of 60 cases were eligible. The patients' median age was 71 years, and the majority (67%) were males; melanoma was the most common indication for ICIs (38%). The most-reported symptoms were fatigue (80%) and muscle weakness (78%). The median number of doses to the development of IM3OS was one. The average creatine kinase level was 9,645 IU/L. Cardiac arrhythmias occurred in 67% of patients, and 18% had depressed ejection fraction. Initial treatment consisted of immunosuppression with high-dose steroids and supportive therapies. Sixty percent of the patients died in hospital because of acute complications.ConclusionIM3OS can be associated with significant mortality and morbidity. Prospective studies are needed to understand the optimal approach to diagnose and manage these patients and to develop biomarkers to predict the occurrence and severity of this rare but serious condition.Implications for practiceClinicians should suspect coexisting myositis and/or myasthenia gravis in all patients with immune checkpoint inhibitor-induced myocarditis, given their propensity to occur together. Early recognition and prompt treatment with the help of a multidisciplinary team might help improve the outcomes of this life-threatening condition.

Project description:BackgroundImmunotherapy offers new hope for cancer patients but presents new medical challenges for healthcare workers in terms of the management of immune-related adverse events (irAEs). The clinical data of two patients with advanced thymoma (T) admitted to the Fujian Cancer Hospital who developed fulminant myocarditis after undergoing immunosuppressant therapy were analyzed retrospectively, and the relevant literature was reviewed. This study aims to examine treatment of thymic epithelial tumors (TETs)-associated immune myocarditis.Case descriptionAn online search was conducted to retrieve relevant full-text articles, and the selected articles were assessed. In total, 13 articles, comprising the data of 113 patients, were included in an analysis to evaluate the efficacy of immunotherapy. Of the 113 patients, 19 had T and 94 had thymic carcinoma (TC). Of the 19 patients with T, 10 (52.6%) achieved a partial response (PR), 8 (42.1%) had stable disease (SD), and 1 (5.3%) had progressive disease (PD). Of the 94 patients with TC, 1 (1.1%) achieved a complete response (CR), 20 (21.3%) achieved a PR, 51 (54.3%) had SD, and 22 (23.4%) had PD. Five articles reported that fulminant myocarditis developed in nine thymic epithelioma patients who were treated with immunosuppressive agents. Two TC patients who presented with fulminant myocarditis were treated with high-dose corticosteroid therapy and underwent pacemaker insertion; none of the patients died of immune myocardial toxicity. However, of the seven T patients who received high-dose corticosteroid therapy and immunoglobulin therapy, and underwent pacemaker implantation, three survived and four died.ConclusionsImmunotherapy has shown promising results in the treatment of patients with refractory or relapsed TETs. Due to their susceptibility to paraneoplastic autoimmunity, TET patients are at a higher risk of developing severe irAEs than patients with other types of cancer. Given the relatively high morbidity and mortality of irAEs, the administration of immune checkpoint inhibitors (ICIs) to treat TETs should be balanced against the clinical response and the precipitation of potentially severe irAEs.

Dataset Information

Checkpoint inhibitor-induced myocarditis and myasthenia gravis in a recurrent/metastatic thymic carcinoma patient: a case report.

Publications

Checkpoint inhibitor-induced myocarditis and myasthenia gravis in a recurrent/metastatic thymic carcinoma patient: a case report.

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