Dataset Information

Camrelizumab-Related Myocarditis and Myositis With Myasthenia Gravis: A Case Report and Literature Review.

ABSTRACT: Immune checkpoint inhibitors (ICIs) have substantially changed the treatment of a variety of malignant tumors. With the increasing of their usage, the unique immune-mediated toxicity profile of ICIs has become apparent. We report a case of esophageal squamous cell carcinoma in a patient who received anti-programmed cell death protein 1 (PD-1) (camrelizumab) therapy and the occurrence of sequential immune-related adverse events (irAEs). Although many irAEs have been reported, severe myositis caused by camrelizumab with simultaneous involvement of multiple organs, including the myocardium, respiratory muscles, and skeletal muscles, has rarely been described in literature. This 69-year-old male patient developed a grade 4 camrelizumab-induced adverse reaction according to the Common Terminology Criteria for Adverse Events (CTCAE) and was successfully treated with methylprednisolone and immunoglobulins. The early identification of irAEs, immediate discontinuation of immunotherapy, use of steroids and/or immunosuppressants, and adjuvant supportive treatment are critical to the clinical prognosis of patients. It should be aware that autoimmune complications can occur even when ICI treatment is ceased.

SUBMITTER: Bai J

PROVIDER: S-EPMC8761813 | biostudies-literature | 2021

REPOSITORIES: biostudies-literature

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Camrelizumab-Related Myocarditis and Myositis With Myasthenia Gravis: A Case Report and Literature Review.

Bai Jing J Li Dan D Yang Peidan P Xu Kunyan K Wang Yingnan Y Li Qian Q Liu Jiang J Du Wenli W Zhang Fengbin F Feng Rui R

Frontiers in oncology 20220103

Immune checkpoint inhibitors (ICIs) have substantially changed the treatment of a variety of malignant tumors. With the increasing of their usage, the unique immune-mediated toxicity profile of ICIs has become apparent. We report a case of esophageal squamous cell carcinoma in a patient who received anti-programmed cell death protein 1 (PD-1) (camrelizumab) therapy and the occurrence of sequential immune-related adverse events (irAEs). Although many irAEs have been reported, severe myositis caus ...[more]

PMID: 35047395

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Project description:Immunotherapy with programmed death 1 (PD-1) inhibitor has shown activity as first- or second-line treatment for various metastatic human malignancies. Immune-related adverse events (irAEs) are now well-described, and most organ sites are potentially influenced, but the prevalence of myocarditis and myositis/myasthenia gravis (MG) overlap syndrome following esophageal hiatal hernia induced by immunotherapy is rarely reported. Here, we describe a 71-year-old woman with a progressed unresectable extrahepatic cholangiocarcinoma and biliary obstruction. She had no prior history of muscle weakness and neuromuscular disease with a normal body mass index. She was treated with sintilimab as a rescue regimen of immunotherapy. After the first cycle of treatment, she experienced a grade 4 myopathy including simultaneous myositis, myalgia, and myocarditis due to multiple injuries in her cardiac, skeletal, and ocular muscles. She had elevated levels of creatine kinase (CK), cardiac troponin I, and myoglobin (MYO), but MG and myositis-specific and myositis-related antibodies were negative. Immunotherapy was discontinued and pulse high-dose methylprednisolone with a slow tapering and intravenous immunoglobulin (IVIG) was initiated. Two weeks later, the patient's clinical presentation improved significantly. A subsequent cardiac magnetic resonance (MR) examination revealed an old myocardial injury that may be a result of immune-related cardiac toxicity. In the third month following the PD-1 inhibitor therapy, she restarted systemic chemotherapy in combination with an anti-angiogenic agent but without immunotherapy. Half a year later, she complained of repeated abdominal distension and radiographic examinations and endoscopy showed a clinically confirmed diagnosis of sliding hiatal hernia of the esophagus and gastroesophageal reflux disease. Due to mild symptoms associated with gastroesophageal reflux, she was suggested close monitoring with acid secretion blockade rather than immediate surgical intervention. The severity for patients with myositis and myocarditis accompanied without MG is similar to those with MG. Considering the use of PD-1 inhibitors is increasing in cancer patients, physicians should therefore pay more attention to immunotherapy-induced myocarditis with myositis/MG overlap syndrome. Since we hypothesize diaphragmatic hiatal hernia as a potential consequence of immunotherapy-induced myositis, reports on hiatal hernias subsequent to immunotherapy-induced myositis are needed.

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.

Dataset Information

Camrelizumab-Related Myocarditis and Myositis With Myasthenia Gravis: A Case Report and Literature Review.

Publications

Camrelizumab-Related Myocarditis and Myositis With Myasthenia Gravis: A Case Report and Literature Review.

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