Coexistence of acute severe leukocytosis and anaplastic lymphoma kinase‑positive histiocytic sarcoma, a rare entity with an unusual presentation: A case report

Bayram,Ertugrul; Pehlivan,Umur Anil; Erdogan,Kivilcim Eren; Turker,Mehmet; Yalniz,Hafize; Paydas,Semra

doi:10.3892/ol.2024.14649

Coexistence of acute severe leukocytosis and anaplastic lymphoma kinase‑positive histiocytic sarcoma, a rare entity with an unusual presentation: A case report

Authors:
- Ertugrul Bayram
- Umur Anil Pehlivan
- Kivilcim Eren Erdogan
- Mehmet Turker
- Hafize Yalniz
- Semra Paydas
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Affiliations: Department of Medical Oncology, Cukurova University Faculty of Medicine, Adana 01330, Turkey, Department of Radiology, Baskent University Adana Dr. Turgut Noyan Application and Research Center, Adana 01240, Turkey, Department of Pathology, Cukurova University Faculty of Medicine, Adana 01330, Turkey, Department of Medical Oncology, Adana City Training and Research Hospital, Adana 01330, Turkey, Department of Cardiovascular Surgery, Cukurova University Faculty of Medicine, Adana 01330, Turkey
Published online on: August 29, 2024 https://doi.org/10.3892/ol.2024.14649
Article Number: 516
Copyright: © Bayram et al. This is an open access article distributed under the terms of Creative Commons Attribution License.

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Abstract

Soft tissue sarcomas are rare cancers and most cases are metastatic at the time of diagnosis. Although the chances of survival are good with surgical treatment in the early stages, systemic treatment in the advanced stages is only associated with a survival duration of ~12 months. Alterations in the anaplastic lymphoma kinase (ALK) gene are becoming increasingly recognized as pan‑cancer indicators in solid tumors. However, little is known regarding the molecular spectrum of ALK‑positive histiocytosis. Molecular treatments, including ALK inhibitors, are potential treatment options. The present case report describes an aggressive ALK‑positive soft tissue sarcoma with intracardiac metastases and severe leukocytosis responding to ALK inhibitors. The patient initially responded to crizotinib but required alectinib due to central nervous system progression. The patient has shown a near‑complete response and remained stable for 2 years; however, there has been recent lymph node progression.

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1	Ansari J, Naqash AR, Munker R, El-Osta H, Master S, Cotelingam JD, Griffiths E, Greer AH, Yin H, Peddi P and Shackelford RE: Histiocytic sarcoma as a secondary malignancy: Pathobiology, diagnosis, and treatment. Eur J Haematol. 97:9–16. 2016. View Article : Google Scholar : PubMed/NCBI
2	Hung YP and Qian X: Histiocytic sarcoma. Arch Pathol Lab Med. 144:650–654. 2020. View Article : Google Scholar : PubMed/NCBI
3	Egan C, Lack J, Skarshaug S, Pham TA, Abdullaev Z, Xi L, Pack S, Pittaluga S, Jaffe ES and Raffeld M: The mutational landscape of histiocytic sarcoma associated with lymphoid malignancy. Mod Pathol. 34:336–347. 2021. View Article : Google Scholar : PubMed/NCBI
4	Butrynski JE, D'Adamo DR, Hornick JL, Dal Cin P, Antonescu CR, Jhanwar SC, Ladanyi M, Capelletti M, Rodig SJ, Ramaiya N, et al: Crizotinib in ALK-rearranged inflammatory myofibroblastic tumor. N Engl J Med. 363:1727–1733. 2010. View Article : Google Scholar : PubMed/NCBI
5	Gambacorti-Passerini C, Orlov S, Zhang L, Braiteh F, Huang H, Esaki T, Horibe K, Ahn JS, Beck JT, Edenfield WJ, et al: Long-term effects of crizotinib in ALK-positive tumors (excluding NSCLC): A phase 1b open-label study. Am J Hematol. 93:607–614. 2018. View Article : Google Scholar : PubMed/NCBI
6	International Agency for Research on Cancer (IARC), . Soft tissue and bone tumours. Vol 3. IARC; Lyon, France: 2020
7	Sethi B, Pai T, Allam A and Epari S: Anaplastic lymphoma kinase-positive pulmonary inflammatory myofibroblastic tumor with sarcomatous morphology and distant metastases: An unusual histomorphology and behavior. Indian J Pathol Microbiol. 58:509–512. 2015. View Article : Google Scholar : PubMed/NCBI
8	Wang L and Wang W: Safety and efficacy of anaplastic lymphoma kinase tyrosine kinase inhibitors in non-small cell lung cancer (Review). Oncol Rep. 45:13–28. 2021.PubMed/NCBI
9	Zou Z, Xing P, Hao X, Wang Y, Song X, Shan L, Zhang C, Liu Z, Ma K, Dong G and Li J: Intracranial efficacy of alectinib in ALK-positive NSCLC patients with CNS metastases-a multicenter retrospective study. BMC Med. 20:122022. View Article : Google Scholar : PubMed/NCBI
10	WHO, . Classification of tumours editorial board. Soft tissue and bone tumours. Lyon (France): International agency for research on cancer. Journal. 3:1042020.
11	Go RS, Jacobsen E, Baiocchi R, Buhtoiarov I, Butler EB, Campbell PK, Coulter DW, Diamond E, Flagg A, Goodman AM, et al: Histiocytic neoplasms, version 2.2021, NCCN clinical practice guidelines in oncology. J Natl Compr Cancer Netw. 19:1277–1303. 2021. View Article : Google Scholar : PubMed/NCBI
12	Swerdlow SH, Campo E, Harris NL, Jaffe ES, Pileri SA, Stein H and Thiele J: WHO classification of tumours of haematopoietic and lymphoid tissues. International agency for research on cancer Lyon; 2008
13	Picarsic J and Jaffe R: Pathology of histiocytic disorders and neoplasms and related disorders. Histiocytic disorders. Abla O and Janka G: Springer International Publishing; Cham: pp. 3–50. 2018, View Article : Google Scholar
14	Yogarajah M and Tefferi A: Leukemic transformation in myeloproliferative neoplasms: A literature review on risk, characteristics, and outcome. Mayo Clin Proc. 92:1118–1128. 2017. View Article : Google Scholar : PubMed/NCBI
15	Kemps PG, Picarsic J, Durham BH, Hélias-Rodzewicz Z, Hiemcke-Jiwa L, van den Bos C, van de Wetering MD, van Noesel CJM, van Laar JAM, Verdijk RM, et al: ALK-positive histiocytosis: A new clinicopathologic spectrum highlighting neurologic involvement and responses to ALK inhibition. Blood. 139:256–280. 2022. View Article : Google Scholar : PubMed/NCBI
16	Reddivari AKR, Mehta P and Janapala US: Rare presentation of a rare tumor: Histiocytic sarcoma. Cureus. 12:e77702020.PubMed/NCBI
17	Solomon BJ, Mok T, Kim DW, Wu YL, Nakagawa K, Mekhail T, Felip E, Cappuzzo F, Paolini J, Usari T, et al: First-line crizotinib versus chemotherapy in ALK-positive lung cancer. N Engl J Med. 371:2167–2177. 2014. View Article : Google Scholar : PubMed/NCBI
18	Xu X, Li H, Peng K, Yu Y, Chen L, Fang Y, Sun Y, Hou Y and Liu T: ALK-G1269A mutation in epithelioid inflammatory myofibroblastic sarcoma after progression on crizotinib: A case report. Oncol Lett. 17:2370–2376. 2019.PubMed/NCBI
19	Shon W and Billings SD: Epithelioid vascular tumors: A review. Adv Anat Pathol. 26:186–197. 2019. View Article : Google Scholar : PubMed/NCBI
20	Luzar B and Calonje E: Update on cutaneous epithelioid vascular tumours. Diagnostic Histopathology. 24:273–287. 2018. View Article : Google Scholar
21	Folpe AL: Vascular tumors of intermediate malignancy: An update. Hum Pathol. 147:114–128. 2024. View Article : Google Scholar : PubMed/NCBI
22	Magdy M, Karim NA, Eldessouki I, Gaber O, Rahouma M and Ghareeb M: Myeloid sarcoma. Oncol Res Treat. 42:224–229. 2019. View Article : Google Scholar : PubMed/NCBI
23	Thalhammer-Scherrer R, Mitterbauer G, Simonitsch I, Jaeger U, Lechner K, Schneider B, Fonatsch C and Schwarzinger I: The immunophenotype of 325 adult acute leukemias: Relationship to morphologic and molecular classification and proposal for a minimal screening program highly predictive for lineage discrimination. Am J Clin Pathol. 117:380–389. 2002. View Article : Google Scholar : PubMed/NCBI
24	Xue T, Jiang XN, Wang WG, Zhou XY and Li XQ: Interdigitating dendritic cell sarcoma: Clinicopathologic study of 8 cases with review of the literature. Ann Diagn Pathol. 34:155–160. 2018. View Article : Google Scholar : PubMed/NCBI
25	Szumera-Ciećkiewicz A, Bosisio F, Teterycz P, Antoranz A, Delogu F, Koljenović S, van de Wiel BA, Blokx W, van Kempen LC, Rutkowski P, et al: SOX10 is as specific as S100 protein in detecting metastases of melanoma in lymph nodes and is recommended for sentinel lymph node assessment. Eur J Cancer. 137:175–182. 2020. View Article : Google Scholar : PubMed/NCBI
26	Tuffaha MSA, Guski H and Kristiansen G: Markers and immunoprofile of lymphoid neoplasms. Immunohistochemistry in tumor diagnostics. Springer International Publishing; Cham: pp. 207–250. 2023, View Article : Google Scholar
27	Facchetti F, Simbeni M and Lorenzi L: Follicular dendritic cell sarcoma. Pathologica. 113:316–329. 2021. View Article : Google Scholar : PubMed/NCBI
28	Russo A, Franchina T, Ricciardi GRR, Ferraro G, Scimone A, Bronte G, Russo A, Rolfo C and Adamo V: Central nervous system involvement in ALK-rearranged NSCLC: promising strategies to overcome crizotinib resistance. Expert Rev Anticancer Ther. 16:615–623. 2016. View Article : Google Scholar : PubMed/NCBI
29	Nelson TA and Wang N: Targeting lung cancer brain metastases: A narrative review of emerging insights for anaplastic lymphoma kinase (ALK)-positive disease. Transl Lung Cancer Res. 12:379–392. 2023. View Article : Google Scholar : PubMed/NCBI