Редкий случай воспалительной миофибробластической опухоли легкого с мультиорганным метастазированием (клиническое наблюдение)

Будурова М. Д., Трифанов В. С., Багров В. А., Ложкин М. В., Черниченко М. А., Суркова В. С., Полуэктов С. И., Любченко Л. Н. Редкий случай воспалительной миофибробластической опухоли легкого с мультиорганным метастазированием (клиническое наблюдение). Современная Онкология. 2023;25(4):540–546. DOI: 10.26442/18151434.2023.4.202438

© ООО «КОНСИЛИУМ МЕДИКУМ», 2023 г.

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Budurova MD, Trifanov VS, Bagrov VА, Lozhkin MV, Chernichenko MA, Surkova VS, Poluektov SI, Lyubchenko LN. Rare Inflammatory myofibroblastic lung tumor with multi-organ metastasis: A case report. Journal of Modern Oncology. 2023;25(4):540–546. DOI: 10.26442/18151434.2023.4.202438

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Аннотация

Воспалительные миофибробластические опухоли (ВМФО; Inflammatory myofibroblastic tumor – IMT) являются чрезвычайно редкими новообразованиями с распространенностью 0,04–0,7%. Очень мало данных об их клинической картине, патогенезе и биологическом поведении. Они могут возникать в любом возрасте и имеют соотношение 1:1 у мужчин и женщин. ВМФО описаны как доброкачественные новообразования с промежуточным злокачественным потенциалом, поскольку возможно развитие локального рецидива и отдаленного метастазирования. ВМФО характеризуются солидным типом роста, а мультифокальный тип встречается редко. Радикальное хирургическое вмешательство является методом выбора. В статье приведен пример клинического наблюдения ВМФО со злокачественным течением в виде мультиорганного метастазирования через 6 лет после радикально выполненной операции на первичной опухоли. Не существует стандарта оказания медицинской помощи при такой клинической ситуации, особенно учитывая отсутствие мутаций в первичной и метастатической опухолях.

Ключевые слова: воспалительная миофибробластическая опухоль, метастаз воспалительной миофибробластической опухоли, лечение воспалительной миофибробластической опухоли

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Inflammatory myofibroblastic tumors (IMTs) are extremely rare neoplasms, accounting for 0.04–0.7% of all tumors. Data on their clinical presentation, pathogenesis, and biological behavior are scarce. They can occur at any age, involving equally males and females. IMTs have been described as benign neoplasms with intermediate malignant potential, as local recurrence and distant metastasis may occur. IMTs are characterized by a solid growth type, and a multifocal type is uncommon. Definitive surgery is the treatment of choice. We describe a clinical case of a malignant IMT with multi-organ metastasis 6 years after definitive surgery on the primary tumor. There is no standard of care in such a clinical situation, especially given the absence of mutations in the primary and metastatic tumors.

Keywords: inflammatory myofibroblastic tumor, inflammatory myofibroblastic tumor metastasis, treatment of inflammatory myofibroblastic tumor

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Список литературы

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26. Baldi GG, Brahmi M, Lo Vullo S, et al. The Activity of Chemotherapy in Inflammatory Myofibroblastic Tumors: A Multicenter, European Retrospective Case Series Analysis. Oncologist. 2020;25(11):e1777‑84. DOI:10.1634/theoncologist.2020‑0352
27. Butrynski JE, D’Adamo DR, Hornick JL, et al. Crizotinib in ALK-rearranged inflammatory myofibroblastic tumor. N Engl J Med. 2010;363(18):1727‑33. DOI:10.1056/NEJMoa1007056
28. Schöffski P, Sufliarsky J, Gelderblom H, et al. Crizotinib in patients with advanced, inoperable inflammatory myofibroblastic tumours with and without anaplastic lymphoma kinase gene alterations (European Organisation for Research and Treatment of Cancer 90101 CREATE): a multicentre, single-drug, prospective, non-randomised phase 2 trial. Lancet Respir Med. 2018;6(6):431‑41. DOI:10.1016/S2213‑2600(18)30116‑4
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38. Fabre D, Fadel E, Singhal S, et al. Complete resection of pulmonary inflammatory pseudotumors has excellent long-term prognosis. J Thorac Cardiovasc Surg. 2009;137(2):435‑40. DOI:10.1016/j.jtcvs.2008.07.009
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1. Coffin CM, Hornick JL, Fletcher CDM. Inflammatory myofibroblastic tumor: comparison of clinicopathologic, histologic, and immunohistochemical features including ALK expression in atypical and aggressive cases. Am J Surg Pathol. 2007;31(4):509‑20. DOI:10.1097/01.pas.0000213393.57322.c7
2. Pikin OV, Ryabov AB, Volchenko NN, et al. Myofibroblastic tumors of the lung. P. A. Herzen Journal of Oncology. 2017;6(1):57‑61 (in Russian). DOI:10.17116/onkolog20176157‑61
3. Filips A, Maurer MH, Montani M, et al. Inflammatory myofibroblastic tumor of the liver: A case report and review of literature. World J Hepatol. 2020;12(4):170‑83. DOI:10.4254/wjh.v12.i4.170
4. Mahajan P, Casanova M, Ferrari A, et al. Inflammatory myofibroblastic tumor: molecular landscape, targeted therapeutics, and remaining challenges. Curr Probl Cancer. 2021;45(4):100768. DOI:10.1016/j.currproblcancer.2021.100768
5. Ognerubov NA, Ulitina ED. Miofibroblastom of breast: clinical case. Tambov University Review. 2013;18(6‑2):3303‑5 (in Russian).
6. Mariño-Enríquez A, Wang WL, Roy A, et al. Epithelioid inflammatory myofibroblastic sarcoma: An aggressive intra-abdominal variant of inflammatory myofibroblastic tumor with nuclear membrane or perinuclear ALK. Am J Surg Pathol. 2011;35(1):135‑44. DOI:10.1097/PAS.0b013e318200cfd5
7. Casanova M, Brennan B, Alaggio R, et al. Inflammatory myofibroblastic tumor: The experience of the European pediatric Soft Tissue Sarcoma Study Group (EpSSG). Eur J Cancer. 2020;127:123‑9. DOI:10.1016/j.ejca.2019.12.021
8. WHO Classification of Tumours of Soft Tissue and Bone. Edited by Fletcher CDM, Bridge JA, Hogendoorn P, Mertens F. IARC WHO Classification of Tumours. 4th edition. V. 5. 2013. Available at: https://publications.iarc.fr/Book-And-Report-Series/Who-Classification-Of-Tumours/WHO-Classifi.... Accessed: 26.05.2023.
9. Alaggio R, Cecchetto G, Bisogno G, et al. Inflammatory myofibroblastic tumors in childhood: a report from the Italian Cooperative Group studies. Cancer. 2010;116(1):216‑26. DOI:10.1002/cncr.24684
10. Coffin CM, Watterson J, Priest JR, Dehner LP. Extrapulmonary inflammatory myofibroblastic tumor (inflammatory pseudotumor). A clinicopathologic and immunohistochemical study of 84 cases. Am J Surg Pathol. 1995;19(8):859‑72. DOI:10.1097/00000478‑199508000‑00001
11. Dong A, Wang Y, Dong H, et al. Inflammatory myofibroblastic tumor: FDG PET/CT findings with pathologic correlation. Clin Nucl Med. 2014;39(2):113‑21. DOI:10.1097/RLU.0b013e3182952caa
12. Lovly CM, Gupta A, Lipson D, et al. Inflammatory myofibroblastic tumors harbor multiple potentially actionable kinase fusions. Cancer Discov. 2014;4(8):889‑95. DOI:10.1158/2159‑8290.CD‑14‑0377
13. Antonescu CR, Suurmeijer AJH, Zhang L, et al. Molecular characterization of inflammatory myofibroblastic tumors with frequent ALK and ROS1 gene fusions and rare novel RET rearrangement. Am J Surg Pathol. 2015;39(7):957‑67. DOI:10.1097/PAS.0000000000000404
14. Alassiri AH, Ali RH, Shen Y, et al. ETV6-NTRK3 Is Expressed in a Subset of ALK-Negative Inflammatory Myofibroblastic Tumors. Am J Surg Pathol. 2016;40(8): 1051‑61. DOI:10.1097/PAS.0000000000000677
15. Pavlick D, Schrock AB, Malicki D, et al. Identification of NTRK fusions in pediatric mesenchymal tumors. Pediatr Blood Cancer. 2017;64(8):e26433. DOI:10.1002/pbc.26433
16. Preobrazhenskaya EV, Iyevleva AG, Suleymanova AM, et al. Gene rearrangements in consecutive series of pediatric inflammatory myofibroblastic tumors. Pediatr Blood Cancer. 2020;67(5):e28220. DOI:10.1002/pbc.28220
17. Chang JC, Zhang L, Drilon AE, et al. Expanding the Molecular Characterization of Thoracic Inflammatory Myofibroblastic Tumors beyond ALK Gene Rearrangements. J Thorac Oncol. 2019;14(5):825‑34. DOI:10.1016/j.jtho.2018.12.003
18. Mittal A, Gupta A, Dhamija E, et al. Sustained complete response on crizotinib in primary lung inflammatory myofibroblastic tumor – Case report and literature review. Monaldi Arch Chest Dis. 2021;91(3). DOI:10.4081/monaldi.2021.1586
19. Cerfolio RJ, Allen MS, Nascimento AG, et al. Inflammatory pseudotumors of the lung. Ann Thorac Surg. 1999;67(4):933‑6. DOI:10.1016/s0003‑4975(99)00155‑1
20. Debonis SA, Bongiovanni A, Pieri F, et al. ALK-negative lung inflammatory myofibroblastic tumor in a young adult: A case report and literature review of molecular alterations. Medicine (Baltimore). 2021;100(20): e25972. DOI:10.1097/MD.0000000000025972
21. Jiang JY, Comsa M, Wong VCK, Mansberg R. Steroid responsive inflammatory myofibroblastic tumor of the lung evaluated by FDG PET/CT imaging. Radiol Case Rep. 2022;17(3):907‑10. DOI:10.1016/j.radcr.2021.11.049
22. Applebaum H, Kieran MW, Cripe TP, et al. The rationale for nonsteroidal anti-inflammatory drug therapy for inflammatory myofibroblastic tumors: a Children’s Oncology Group study. J Pediatr Surg. 2005;40(6):999‑1003. DOI:10.1016/j.jpedsurg.2005.03.016
23. Grünholz D, Appiani F, Abarca C, et al. Peritoneal myofibroblastic tumor successfully treated with infliximab: Report of one case. Rev Med Chil. 2015;143(7):943‑7 (in Spanish). DOI:10.4067/S0034‑98872015000700017
24. Schaeffer CJ, Minai OA, Sharma N, et al. Inflammatory myofibroblastic tumor of the lung: recurrence after steroid treatment. J Thorac Imaging. 2008;23(3):191‑3. DOI:10.1097/RTI.0b013e31816591a4
25. Panigada S, Sacco O, Girosi D, et al. Corticosteroids may favor proliferation of thoracic inflammatory myofibroblastic tumors. Pediatr Pulmonol. 2014;49(3):E109‑11. DOI:10.1002/ppul.22977
26. Baldi GG, Brahmi M, Lo Vullo S, et al. The Activity of Chemotherapy in Inflammatory Myofibroblastic Tumors: A Multicenter, European Retrospective Case Series Analysis. Oncologist. 2020;25(11):e1777‑84. DOI:10.1634/theoncologist.2020‑0352
27. Butrynski JE, D’Adamo DR, Hornick JL, et al. Crizotinib in ALK-rearranged inflammatory myofibroblastic tumor. N Engl J Med. 2010;363(18):1727‑33. DOI:10.1056/NEJMoa1007056
28. Schöffski P, Sufliarsky J, Gelderblom H, et al. Crizotinib in patients with advanced, inoperable inflammatory myofibroblastic tumours with and without anaplastic lymphoma kinase gene alterations (European Organisation for Research and Treatment of Cancer 90101 CREATE): a multicentre, single-drug, prospective, non-randomised phase 2 trial. Lancet Respir Med. 2018;6(6):431‑41. DOI:10.1016/S2213‑2600(18)30116‑4
29. Trahair T, Gifford AJ, Fordham A, et al. Crizotinib and Surgery for Long-Term Disease Control in Children and Adolescents With ALK-Positive Inflammatory Myofibroblastic Tumors. JCO Precis Oncol. 2019;3:PO.18.00297. DOI:10.1200/PO.18.00297
30. Theilen TM, Soerensen J, Bochennek K, et al. Crizotinib in ALK+ inflammatory myofibroblastic tumors – Current experience and future perspectives. Pediatr Blood Cancer. 2018;65(4):e26920. DOI:10.1002/pbc.26920
31. Alan O, Kuzhan O, Koca S, et al. How long should we continue crizotinib in ALK translocation-positive inflammatory myofibroblastic tumors? Long-term complete response with crizotinib and review of the literature. J Oncol Pharm Pract. 2020;26(4):1011‑8. DOI:10.1177/1078155219879757
32. Mai S, Xiong G, Diao D, et al. Case report: Crizotinib is effective in a patient with ROS1‑rearranged pulmonary inflammatory myofibroblastic tumor. Lung Cancer. 2019;128:101‑4. DOI:10.1016/j.lungcan.2018.12.016
33. Mossé YP, Voss SD, Lim MS, et al. Targeting ALK With Crizotinib in Pediatric Anaplastic Large Cell Lymphoma and Inflammatory Myofibroblastic Tumor: A Children’s Oncology Group Study. J Clin Oncol. 2017;35(28):3215‑21. DOI:10.1200/JCO.2017.73.4830
34. Peters S, Camidge DR, Shaw AT, et al; ALEX Trial Investigators. Alectinib versus Crizotinib in Untreated ALK-Positive Non-Small-Cell Lung Cancer. N Engl J Med. 2017;377(9):829‑38. DOI:10.1056/NEJMoa1704795
35. Gambacorti-Passerini C, Mussolin L, Brugieres L. Abrupt relapse of ALK-positive lymphoma after discontinuation of crizotinib. N Engl J Med. 2016;374(1):95‑6. DOI:10.1056/NEJMc1511045
36. Du X, Gao Y, Zhao H, et al. Clinicopathological analysis of epithelioid inflammatory myofibroblastic sarcoma. Oncol Lett. 2018;15(6):9317‑26. DOI:10.3892/ol.2018.8530
37. Panagiotopoulos N, Patrini D, Gvinianidze L, et al. Inflammatory myofibroblastic tumour of the lung: a reactive lesion or a true neoplasm? J Thorac Dis. 2015;7(5):908‑11. DOI:10.3978/j.issn.2072‑1439.2015.04.60
38. Fabre D, Fadel E, Singhal S, et al. Complete resection of pulmonary inflammatory pseudotumors has excellent long-term prognosis. J Thorac Cardiovasc Surg. 2009;137(2):435‑40. DOI:10.1016/j.jtcvs.2008.07.009
39. Fordham AM, Xie J, Gifford AJ, et al. CD30 and ALK combination therapy has high therapeutic potency in RANBP2-ALK-rearranged epithelioid inflammatory myofibroblastic sarcoma. Br J Cancer. 2020;123(7):1101‑13. DOI:10.1038/s41416‑020‑0996‑2
40. Schulte JH, Moreno L, Ziegler DS, et al. Final analysis of phase I study of ceritinib in pediatric patients with malignancies harboring activated anaplastic lymphoma kinase (ALK). JCO. 2020;38(15_suppl.):10505. DOI:10.1200/JCO.2020.38.15_suppl.10505
41. Brivio E, Zwaan CM. ALK inhibition in two emblematic cases of pediatric inflammatory myofibroblastic tumor: Efficacy and side effects. Pediatr Blood Cancer. 2019;66(5):e27645. DOI:10.1002/pbc.27645
42. Drilon A, Laetsch TW, Kummar S, et al. Efficacy of Larotrectinib in TRK Fusion-Positive Cancers in Adults and Children. N Engl J Med. 2018;378(8):731‑9. DOI:10.1056/NEJMoa1714448
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Авторы

М. Д. Будурова*^1,2, В. С. Трифанов^1,3, В. А. Багров¹, М. В. Ложкин⁴, М. А. Черниченко¹, В. С. Суркова1, С. И. Полуэктов¹, Л. Н. Любченко¹

¹Московский научно-исследовательский онкологический институт им. П. А. Герцена – филиал ФГБУ «Национальный медицинский исследовательский центр радиологии» Минздрава России, Москва, Россия;
²ФГБОУ ВО «Российский университет медицины» Минздрава России, Москва, Россия;
³ФГАОУ ВО «Российский университет дружбы народов», Москва, Россия;
⁴ГБУЗ «Городская клиническая онкологическая больница №1» Департамента здравоохранения г. Москвы, Москва, Россия
*m.budurova@rambler.ru

________________________________________________

Marina D. Budurova*^1,2, Vladimir S. Trifanov^1,3, Vladimir А. Bagrov¹, Mikhail V. Lozhkin⁴, Maria A. Chernichenko¹, Viktoria S. Surkova¹, Sergey I. Poluektov¹, Liudmila N. Lyubchenko¹

¹Hertsen Moscow Oncology Research Institute – branche of the National Medical Research Radiological Centre, Moscow, Russia;
²Russian University of Medicine, Moscow, Russia;
³People’s Friendship University of Russia (RUDN University), Moscow, Russia;
⁴Moscow State Budgetary Healthcare Institution “Moscow City Oncological Hospital No. 1” of the Moscow Department of Healthcare, Moscow, Russia
*m.budurova@rambler.ru

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