Fatal Fulminant Myocarditis Following a Single Dose of Pembrolizumab in a Patient with Recurrent Thymoma: A Case Report
References
- Morgenthaler, T.I., et al., Thymoma. Mayo Clin Proc, 1993. 68(11): p. 1110–23.
- Detterbeck, F.C. and A. Zeeshan, Thymoma: current diagnosis and treatment. Chin Med J (Engl), 2013. 126(11): p. 2186–91.
- Gripp, S., et al., Thymoma: prognostic factors and treatment outcomes. Cancer, 1998. 83(8): p. 1495–503.
- Alothaimeen, H.S. and M.A. Memon, Treatment Outcome and Prognostic Factors of Malignant Thymoma - A Single Institution Experience. Asian Pac J Cancer Prev, 2020. 21(3): p. 653–661.
- Robinson, S.P. and H. Akhondi, Thymoma, in StatPearls. 2025, StatPearls Publishing: Treasure Island (FL).
- Shelly, S., et al., Thymoma and autoimmunity. Cell Mol Immunol, 2011. 8(3): p. 199–202.
- von der Thüsen, J., Thymic epithelial tumours: histopathological classification and differential diagnosis. Histopathology, 2024. 84(1): p. 196–215.
- Marx, A., et al., The 2015 World Health Organization Classification of Tumors of the Thymus: Continuity and Changes. J Thorac Oncol, 2015. 10(10): p. 1383–95.
- Suster, D.I. and S. Suster, Histologic features, growth patterns and classification of atypical thymomas. Mediastinum, 2025. 9: p. 20.
- Drevet, G., et al., Optimal management of thymic malignancies: current perspectives. Cancer Manag Res, 2019. 11: p. 6803–6814.
- Roden, A.C., Non-surgical treatment of locally advanced thymic epithelial tumors-a need for multicenter trials. J Thorac Dis, 2016. 8(8): p. 1859–62.
- Song, Z., X. Yu, and Y. Zhang, Chemotherapy and prognosis in advanced thymic carcinoma patients. Clinics (Sao Paulo), 2015. 70(12): p. 775–80.
- Jakopovic, M., et al., Immunotherapy for thymoma. J Thorac Dis, 2020. 12(12): p. 7635–7641.
- Meng, L., et al., Mechanisms of immune checkpoint inhibitors: insights into the regulation of circular RNAS involved in cancer hallmarks. Cell Death Dis, 2024. 15(1): p. 3.
- Rajan, A., A.K. Sivapiromrat, and M.J. McAdams, Immunotherapy for Thymomas and Thymic Carcinomas: Current Status and Future Directions. Cancers (Basel), 2024. 16(7).
- Sekine, I., Y. Aida, and H. Suzuki, Expression patterns and prognostic value of programmed death ligand-1 and programmed death 1 in thymoma and thymic carcinoma. Mediastinum, 2018. 2.
- Hao, Y., et al., Analysis of the efficacy and safety of immunotherapy in advanced thymoma patients. Cancer Med, 2023. 12(5): p. 5649–5655.
- Liu, S., et al., Severe cardiotoxicity in 2 patients with thymoma receiving immune checkpoint inhibitor therapy: A case report. Medicine (Baltimore), 2022. 101(46): p. e31873.
- Moslehi, J.J., et al., Increased reporting of fatal immune checkpoint inhibitor-associated myocarditis. Lancet, 2018. 391(10124): p. 933.
- Johnson, D.B., et al., Fulminant Myocarditis with Combination Immune Checkpoint Blockade. N Engl J Med, 2016. 375(18): p. 1749–1755.
- Yokoyama, S. and H. Miyoshi, Thymic tumors and immune checkpoint inhibitors. J Thorac Dis, 2018. 10(Suppl 13): p. S1509–s1515.
- Herbst, R.S., et al., Predictive correlates of response to the anti-PD-L1 antibody MPDL3280A in cancer patients. Nature, 2014. 515(7528): p. 563–7.
- Radovich, M., et al., The Integrated Genomic Landscape of Thymic Epithelial Tumors. Cancer Cell, 2018. 33(2): p. 244–258.e10.
- Giaccone, G., et al., Pembrolizumab in patients with thymic carcinoma: a single-arm, single-centre, phase 2 study. Lancet Oncol, 2018. 19(3): p. 347–355.
- Rajan, A., et al., Efficacy and tolerability of anti-programmed death-ligand 1 (PD-L1) antibody (Avelumab) treatment in advanced thymoma. J Immunother Cancer, 2019. 7(1): p. 269.
- Cho, J., et al., Pembrolizumab for Patients With Refractory or Relapsed Thymic Epithelial Tumor: An Open-Label Phase II Trial. J Clin Oncol, 2019. 37(24): p. 2162–2170.
- Jang, S.Y., et al., Early development of pembrolizumab-induced fulminant myositis and cardiotoxicity in a patient with metastatic thymoma. Respirol Case Rep, 2022. 10(9): p. e01025.
- Feng, Y., et al., Immune checkpoint inhibitor myocarditis in thymic epithelial tumors: a case report and literature review. Transl Cancer Res, 2024. 13(2): p. 1208–1218.
- Wong-Chong, J., et al., Pembrolizumab as a novel therapeutic option for patients with refractory thymic epithelial tumor: A case report. World J Clin Cases, 2021. 9(5): p. 1139–1147.
- Cooper, L.T., Jr., et al., Myocarditis Associated With Immune Checkpoint Inhibitors in Patients With Thymoma. JACC Adv, 2023. 2(6): p. 100428.
- Huang D, S.F., Zhang W, Ma H, Lin X,, Fulminant Myocarditis Complicated with Immune Checkpoint Inhibitor in Patients with Thymoma: Report of Three Cases. Int J Cardiovasc Pract., 2023. 7(1):e130990.
- Mahmood, S.S., et al., Myocarditis in Patients Treated With Immune Checkpoint Inhibitors. J Am Coll Cardiol, 2018. 71(16): p. 1755–1764.
- Demet Seker, A.Y., Fatal triad of checkpoint inhibitors: Pembrolizumab induced myasthenia gravis with myositis and myocarditis in a patient with thymoma. Neurology Asia, 2023. 28(2): p. 439 – 443.
- Matsumori, A., Myocarditis and autoimmunity. Expert Review of Cardiovascular Therapy, 2023. 21(6): p. 437–451.
- Nicolas, M.-C., et al., Anti-PD1 associated fulminant myocarditis after a single pembrolizumab dose: the role of occult pre-existing autoimmunity. Haematologica, 2018. 103(7): p. e318–e321.
- Giovannini, E., et al., Pembrolizumab-Induced Fatal Myasthenia, Myocarditis, and Myositis in a Patient with Metastatic Melanoma: Autopsy, Histological, and Immunohistochemical Findings—A Case Report and Literature Review. International Journal of Molecular Sciences, 2023. 24(13): p. 10919.
Language: English
Submitted on: Aug 17, 2025
Accepted on: Nov 3, 2025
Published on: May 21, 2026
Published by: Helenic Society of Medical Oncology
In partnership with: Paradigm Publishing Services
Publication frequency: 2 issues per year
Keywords:
Related subjects:
© 2026 Marwa Haitham Al-Najafi, Rasha Raed Al-Mallah, Nazar Mohammed Ther Jawhar, published by Helenic Society of Medical Oncology
This work is licensed under the Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 License.