The rare co-occurrence of cholangiocarcinoma and chronic eosinophilic leukemia
, 1, 1, 1, 2, 3, 4, 4
Abstract
Introduction. Cholangiocarcinoma (CCA) is a rare and highly aggressive malignancy of the bile ducts with a poor prognosis and a 5-year survival rate of approximately 5%. Chronic eosinophilic leukemia (CEL), characterized by clonal eosinophil proliferation, is a rare hematologic malignancy often progressing to acute leukemia. The simultaneous occurrence of these two malignancies is exceedingly rare, according to available research, this is the first case reported in the literature.
Case report. This is the case of a 44-year-old female presenting with fatigue, diagnosed with metastatic CCA following imaging and biopsy. During chemotherapy with gemcitabine and cisplatin, the patient developed unexplained leukocytosis and eosinophilia, leading to a subsequent diagnosis of CEL. Despite aggressive treatment, the patient experienced complications, including cholangitis and renal failure, and succumbed to her illness four months post-diagnosis.
Conclusions. This case highlights the rare co-occurrence of CCA and CEL, emphasizing the importance of vigilant monitoring for secondary hematologic malignancies in oncology patients. Early recognition and treatment of concurrent disorders can improve clinical outcomes and inform future research on such rare malignancy combinations.
Keywords: cholangiocarcinomachronic eosinophilic leukemiachemotherapyconcurrent malignancies
References
- Clements O, Eliahoo J, Kim JUn, et al. Risk factors for intrahepatic and extrahepatic cholangiocarcinoma: A systematic review and meta-analysis. J Hepatol. 2020; 72(1): 95–103.
- Palmer WC, Patel T. Are common factors involved in the pathogenesis of primary liver cancers? A meta-analysis of risk factors for intrahepatic cholangiocarcinoma. J Hepatol. 2012; 57(1): 69–76.
- Mosconi S, Beretta GD, Labianca R, et al. Cholangiocarcinoma. Crit Rev Oncol Hematol. 2009; 69(3): 259–270.
- Ying J, Chen J. Combination versus mono-therapy as salvage treatment for advanced biliary tract cancer: A comprehensive meta-analysis of published data. Crit Rev Oncol Hematol. 2019; 139: 134–142.
- Razumilava N, Gores GJ. Cholangiocarcinoma. Lancet. 2014; 383(9935): 2168–2179.
- Olgum A, Ceylan C. Chronic Neutrophilic Leukemia and Chronic Eosinophilic Leukemia-Not Otherwise Specified. Turkiye Klinikleri Hematology-Special Topics. 2021; 14(4): 81–85.
- Abu-Hamda EM, Baron TH. Endoscopic management of cholangiocarcinoma. Semin Liver Dis. 2004; 24(2): 165–175.
- Laing FC. The gallbladder and bile ducts. Diagnostic ultrasound. 1998; 1: 175–223.
- Farges O, Fuks D, Boleslawski E, et al. Influence of surgical margins on outcome in patients with intrahepatic cholangiocarcinoma: a multicenter study by the AFC-IHCC-2009 study group. Ann Surg. 2011; 254(5): 824–29; discussion 830.
- Mavros MN, Economopoulos KP, Alexiou VG, et al. Treatment and Prognosis for Patients With Intrahepatic Cholangiocarcinoma: Systematic Review and Meta-analysis. JAMA Surg. 2014; 149(6): 565–574.
- Rizzo A, Ricci AD, Brandi G. IDH inhibitors in advanced cholangiocarcinoma: Another arrow in the quiver? Cancer Treat Res Commun. 2021; 27: 100356.
- Fruchtman H, Avigan ZM, Waksal JA, et al. Management of isocitrate dehydrogenase 1/2 mutated acute myeloid leukemia. Leukemia. 2024; 38(5): 927–935.
- Waitkus MS, Diplas BH, Yan H. Biological Role and Therapeutic Potential of IDH Mutations in Cancer. Cancer Cell. 2018; 34(2): 186–195.
- Fujii T, Khawaja MR, DiNardo CD, et al. Targeting isocitrate dehydrogenase (IDH) in cancer. Discov Med. 2016; 21(117): 373–380.
- Krell D, Mulholland P, Frampton AE, et al. IDH mutations in tumorigenesis and their potential role as novel therapeutic targets. Future Oncol. 2013; 9(12): 1923–1935.
- Rimini M, Fabregat-Franco C, Burgio V, et al. Molecular profile and its clinical impact of IDH1 mutated versus IDH1 wild type intrahepatic cholangiocarcinoma. Sci Rep. 2022; 12(1): 18775.
- Byun JaM, Yoo SJ, Kim HJ, et al. IDH1/2 mutations in acute myeloid leukemia. Blood Res. 2022; 57(1): 13–19.
- Feng JH, Guo XP, Chen YY, et al. Prognostic significance of IDH1 mutations in acute myeloid leukemia: a meta-analysis. Am J Blood Res. 2012; 2(4): 254–264.
- Uson Junior PL, Borad MJ. Clinical Utility of Ivosidenib in the Treatment of IDH1-Mutant Cholangiocarcinoma: Evidence To Date. Cancer Manag Res. 2023; 15: 1025–1031.
- Tefferi A, Gotlib J, Pardanani A. Hypereosinophilic syndrome and clonal eosinophilia: point-of-care diagnostic algorithm and treatment update. Mayo Clin Proc. 2010; 85(2): 158–164.
- Fauci AS, Harley JB, Roberts WC, et al. NIH conference. The idiopathic hypereosinophilic syndrome. Clinical, pathophysiologic, and therapeutic considerations. Ann Intern Med. 1982; 97(1): 78–92.
- Gotlib J, Cools J, Malone JM, et al. The FIP1L1-PDGFRalpha fusion tyrosine kinase in hypereosinophilic syndrome and chronic eosinophilic leukemia: implications for diagnosis, classification, and management. Blood. 2004; 103(8): 2879–2891.
- Helbig G, Soja A, Bartkowska-Chrobok A, et al. Chronic eosinophilic leukemia-not otherwise specified has a poor prognosis with unresponsiveness to conventional treatment and high risk of acute transformation. Am J Hematol. 2012; 87(6): 643–645.
- Morsia E, Reichard K, Pardanani A, et al.
WHO defined chronic eosinophilic leukemia, not otherwise specified (CEL ,NOS ): A contemporary series from the Mayo Clinic. Am J Hematol. 2020; 95(7): E172–E174.