A Rare Case of Pulmonary Neuroendocrine Carcinoma in Transfusion-dependent Thalassemia Patient: Clinical Presentation, Management, and Implications
Abstract
Transfusion-dependent thalassemia (TDT) is often accompanied by complications related to iron overload and the development of malignant solid tumors or hematological malignancies. The occurrence of Neuroendocrine carcinoma, specifically in the respiratory tract, is very rare, with a prevalence of approximately 25%. Therefore, this study presented a case of a 42-year-old male with a beta-thalassemia major at 28 years, complaining of shortness of breath. This case was reported due to its rarity in providing information about solid tumors in thalassemia patients. The physical examination revealed several symptoms, including tachycardia, tachypnea, anemia, icteric sclera, elevated jugular venous pressure, coarse wet Ronchi in the medial to basal areas of both lungs, hepatomegaly, and splenomegaly (Schuffner 4). The patient regularly received blood transfusions and iron chelation therapy. A thoracic CT scan showed a lung mass and a biopsy of the mass revealed Pulmonary Neuroendocrine Carcinoma with high-grade proliferation and, large cell type. The patient also passed through cisplatin-etoposide chemotherapy for 6 cycles every 21 days. There is almost no data on pulmonary neuroendocrine carcinoma in thalassemia patients, so it is hoped that this case report can provide information about malignant solid tumors that can occur in thalassemia patients.
Keywords
References
A. Subahi E. Extramedullary hematopoiesis in patients with transfusion-dependent β-thalassemia (TDT): a systematic review. Ann Med. 2022;54:764–74. https://doi.org/10.1080/07853890.2022.2048065.
Ahmad RW. Low back pain in beta-thalassemia major revealing sacral extramedullary hematopoiesis: A case report. Clin Case Rep. 2021;9.
Shie BX, Deng WX, Jin FX. Bone marrow metastatic neuroendocrine carcinoma with unknown primary site: A case report and review of the literature. World J Clin Cases. 2022;10(30):11074-81.
Ichikawa S, Inokura K, Kawamura Y, Fukuhara N, Yokoyama H, Ouchi K, Fujishima F, Harigae H. Massive bone marrow infiltration of neuroendocrine carcinoma mimicking aggressive hematological malignancy. Journal of Hematopathology. 2021;14:337-9.
Hodroj MH. Thalassemia and malignancy: An emerging concern? Blood Rev. 2019;37:100585.
Lee M-C, Aly RG, Adusumilli PS. New staging system for pulmonary neuroendocrine tumors: A clinical and pathologic necessity. J Thorac Cardiovasc Surg. 2018;155:367–8.
Sorbye H. European Neuroendocrine Tumor Society 2023 guidance paper for digestive neuroendocrine carcinoma. J Neuroendocrinol. 2023;35.
Mahayasa M, Wibowo PS. Tumor Neuroendokrin: Kasus Serial di RSUD Dr. Soetomo. JBN. 2018;2:28.
Benson AB. NCCN Guidelines Version 2. [Internet] 2022 Neuroendocrine and Adrenal Tumors NCCN Guidelines Panel Disclosures; Available from: https://www.nccn.org/home/member- (2022).
Origa R. Incidence of cancer and related deaths in hemoglobinopathies: A follow-up of 4631 patients between 1970 and 2021. Cancer. 2023;129:107–17.
Albattah A. Case report: Papillary thyroid cancer in a patient with celiac disease and thalassemia trait. Case Rep Oncol. 2020;13:1364–7.
Chung W-S, Lin C-L, Lin C-L, Kao C-H. Thalassaemia and risk of cancer: a population-based cohort study. J Epidemiol Community Health. 2015;69(1978):1066–70.
Atmakusuma TD, Girson R, Koesnoe S. Correlations between iron load and CD4 in adult transfusion-dependent beta-thalassemia. Anemia. 2021;2021:1–5.
Amrita PNA. Serum ferritin level affects T lymphocyte CD4, CD8, and CD4/CD8 ratio in transfusion-dependent beta-thalassemia. Drug Invention Today. 2020;13:887–92.
Komoto K. Iron chelation cancer therapy using hydrophilic block copolymers conjugated with deferoxamine. Cancer Sci. 2021;112:410–21.
You H. Deferoxamine inhibits acute lymphoblastic leukemia progression through repression of ROS/HIF-1α, Wnt/β-Catenin, and p38MAPK/ERK Pathways. J Oncol. 2022;2022:1–10.
Bajbouj K, Shafarin J, Hamad M. High-dose Deferoxamine treatment disrupts intracellular iron homeostasis, reduces growth, and induces apoptosis in metastatic and nonmetastatic breast cancer cell lines. Technol Cancer Res Treat. 2018;17:153303381876447.
Halawi R, Cappellini MD, Taher A. A higher prevalence of hematologic malignancies in patients with thalassemia: Background and culprits. Am J Hematol. 2017;92:414–6.
Pellegrino C. Acute promyelocytic leukemia in a woman with thalassemia intermedia: Case report and review of literature on hematological malignancies in β-thalassemia patients. Hematol Rep. 2022;14:310–21.
Refbacks
- There are currently no refbacks.