The efficacy of immune checkpoint inhibitors in rare tumors: A systematic review of published clinical trials

Güven, Deniz Can; Stephen, Bettzy; Şahin, Taha Koray; Cakir, Ibrahim Yahya; Erul, Enes; Aksoy, Sercan

doi:10.1016/j.critrevonc.2022.103700

Cited by 9 publications

(9 citation statements)

References 115 publications

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“…In recent years, ICIs have been a promising therapy for malignancies. [ 8 ] Anti-PD-1/PD-L1 monoclonal antibodies have been extensively studied in different type of cancers. [ 3 ] The common side effects are reported as pneumonitis, hepatitis, endocrinopathies, dermatologic toxicity, gastrointestinal toxicity, and myositis.…”

Section: Discussionmentioning

confidence: 99%

Immune checkpoint inhibitor sintilimab-induced lethal myocarditis overlapping with myasthenia gravis in thymoma patient: A case report

Wang

Bing-di

et al. 2023

Medicine

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Rationale: Immune checkpoint inhibitors have been extensively used and significantly improved the clinical outcomes in multiple types of cancer. But the immune-related adverse events occur frequently, particularly in thymoma. The cardiac immune-related adverse, which is relatively rare but fatal, have been increasing reported. Patient concerns: A 45-year-old thymoma patient was admitted to our hospital after receiving anti-programmed cell death-1 treatment with sintilimab 14 days later, accompanied by abdominal pain, intermittent chest tightness and dizziness. Diagnoses: The laboratory tests revealed elevated serum troponin I. Electrocardiogram reported the prolongation of QTc interval. Echocardiography showed small amount of pericardial effusion, a left ventricular ejection fraction of 71%. Coronary artery computed tomography angiography revealed localized noncalcified plaque in the middle of the left anterior descending artery and mild stenosis of the lumen. Enhanced computed tomography scanning of the whole abdomen showed no abnormal signs in the parenchyma organs. Combining the results of the examinations, the Immune checkpoint inhibitor induced myocarditis was diagnosed. Interventions: The patient was treated with glucocorticoids (120 mg/day, IV, methylprednisolone) within 24 hours of admission. Seven days later, the patient experienced tachy ventricular arrhythmia and cardiogenic shock and was transferred to intensive care unit after electrical cardioversion, tracheal intubation and cardiopulmonary resuscitation. Intravenous immunoglobulin therapy at 25 g/day was given and methylprednisolone was reduced to 40 mg/day for the next 3 days. Intravenous esmolol and lidocaine were used for correcting arrhythmias. Ventilator positive pressure ventilation was used for respiratory support. She was administrated with plasmapheresis when the electrocardiogram monitoring showed ventricular arrhythmia storms. Outcome: The patient progressed to ventricular arrhythmia storms and cardiac failure, which eventually resulted in death. Lessons: The case aims to raise awareness of immune-mediated cardiotoxicity and bring thoughts to the prospects of immunotherapy in thymoma.

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Section: Discussionmentioning

confidence: 99%

Immune checkpoint inhibitor sintilimab-induced lethal myocarditis overlapping with myasthenia gravis in thymoma patient: A case report

Wang

Bing-di

et al. 2023

Medicine

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“…By assisting the immune system in recognizing cancer cells, immunotherapy has proven to be a promising anti-tumor strategy. Particularly, immune checkpoint inhibitors have benefited numerous tumor patients [ 2 , 3 ]. However, other than the high cost, immunotherapy also has many other deficiencies such as unique toxicity profiles [ 4 ].…”

Section: Introductionmentioning

confidence: 99%

Pan-cancer analysis of super-enhancer-induced LINC00862 and validation as a SIRT1-promoting factor in cervical cancer and gastric cancer

Liu,

Wang,

et al. 2024

Translational Oncology

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“…While individually rare, the rare cancers constitute almost over 20% of newly diagnosed cancers 14 . However, the developments in rare cancers were slow due to problems with case definition and diagnosis, limitations with clinical trial involvement, and lesser support from the industry due to a smaller target sample size 11,14,15 . Additionally, the relative inefficacy of ICIs in earlier trials of sarcoma 16 and neuroendocrine tumors 17 further slowed the interest in ICI use in rare tumors.…”

Section: Introductionmentioning

confidence: 99%

“…The available trials mainly focused on tumors with an active immune milieu 10 or tumors with a higher incidence or prevalence, while the interest and advances in the ICI field were relatively slow in most rare tumors. 11 Rare tumors are a significant but understudied problem. 12 While definitions vary across organizations, the NCI defines rare tumors as tumors with an incidence of 15 or fewer cases for 100,000 people per year.…”

Section: Introductionmentioning

confidence: 99%

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The efficacy of immunotherapy and chemoimmunotherapy in patients with advanced rare tumors: A Turkish oncology group (TOG) study

Guven,

Aykan,

Muglu

et al. 2023

Cancer Medicine

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IntroductionThe advances in immune checkpoint inhibitors (ICIs) were relatively slow in rare tumors. Therefore, we conducted a multi‐center study evaluating the efficacy of ICI monotherapy and the combination of ICIs with chemotherapy (CT) in patients with advanced rare tumors.MethodsIn this retrospective cohort study, we included 93 patients treated with ICIs for NCI‐defined rare tumors from the 12 cancer centers in Turkey. The primary endpoints were the overall response (ORR) and disease control rate (DCR).ResultsThe cohort's median age was 56, and 53.8% of the patients were male. The most frequent diagnosis was sarcoma (29%), and 81.7% of the patients were previously treated with at least one line of systemic therapy in the advanced stage.The ORR and DCR were 36.8% and 63.2%, respectively. The germ cell tumors had the lowest ORR (0%), while the Merkel cell carcinoma had the highest ORR to ICIs (57.1%). Patients treated with ICI + ICI or ICI plus chemotherapy combinations had higher ORR (55.2% vs. 27.6%, p = 0.012) and DCR (82.8% vs. 53.4%, p = 0.008).The median OS was 13.47 (95% CI: 7.79–19.15) months, and the six and 12‐month survival rates were 71% and 52%. The median duration of response was 16.59 months, and the 12‐month progression‐free survival rate was 66% in responders. The median time‐to‐treatment failure was 5.06 months (95% CI: 3.42–6.71). Three patients had high‐grade irAEs with ICIs (grade 3 colitis, grade 3 gastritis, and grade 3 encephalitis in one patient each).ConclusionWe observed over 30% ORR and a 13‐month median OS in patients with rare cancers treated with ICI monotherapy or ICI plus CT combinations. The response rates to ICIs or ICIs plus CT significantly varied across different tumor types. Responding patients had over 2 years of survival, highlighting a need for further trials with ICIs for patients with rare tumors.

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The efficacy of immune checkpoint inhibitors in rare tumors: A systematic review of published clinical trials

Cited by 9 publications

References 115 publications

Immune checkpoint inhibitor sintilimab-induced lethal myocarditis overlapping with myasthenia gravis in thymoma patient: A case report

Immune checkpoint inhibitor sintilimab-induced lethal myocarditis overlapping with myasthenia gravis in thymoma patient: A case report

Pan-cancer analysis of super-enhancer-induced LINC00862 and validation as a SIRT1-promoting factor in cervical cancer and gastric cancer

The efficacy of immunotherapy and chemoimmunotherapy in patients with advanced rare tumors: A Turkish oncology group (TOG) study

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