nodgoРоссийский журнал детской гематологии и онкологии (РЖДГиО)Russian Journal of Pediatric Hematology and Oncology2311-12672413-5496LTD “Graphica”10.21682/2311-1267-2020-7-2-64-77nodgo-605Research ArticleОБЗОРЫ ЛИТЕРАТУРЫLITERATURE REVIEWSВоспалительные миофибробластические опухоли у детей: обзор литературыInflammatory myofibroblastic tumors in children: literature reviewhttps://orcid.org/0000-0002-5489-1879СулеймановаА. М.SuleymanovaA. M.

Амина Магомедовна Сулейманова, аспирант очной формы обучения; врач-детский онколог

Россия, 117997, Москва, ул. Саморы Машела, 1

115478, Москва, Каширское шоссе, 23

A.M. Suleymanova: Postdoctoral Fellowship; Pediatric Oncologist Research Institute of Pediatric Oncology and Hematology

1 Samory Mashela St., Moscow, 117997, Russia

23 Kashirskoe Shosse, Moscow, 115478, Russia

a.sulejmanova@ronc.ruhttps://orcid.org/0000-0002-3704-8783КачановД. Ю.KachanovD. Yu.

д.м.н., заведующий отделением клинической онкологии, заместитель директора Института онкологии, радиологии и ядерной медицины

Россия, 117997, Москва, ул. Саморы Машела, 1

SPIN-код: 9878-5540

Dr. of Sci. (Med.), Head of the Department of Clinical Oncology, Deputy Director of the Institute of Oncology, Radiology and Nuclear Medicine

1 Samory Mashela St., Moscow, 117997, Russia

Denis.Kachanov@fccho-moscow.ruhttps://orcid.org/0000-0003-4529-7891ИмянитовЕ. Н.ImyanitovE. N.

член-корреспондент РАН, д.м.н., профессор, руководитель научного отдела биологии опухолевого роста и лаборатории молекулярной онкологии

Россия, 197758, Санкт-Петербург, пос. Песочный, ул. Ленинградская, 68

SPIN-код: 1909-7323

Corresponding Member of RAS, Dr. of Sci. (Med.), Professor, Head of the Research Department of Biology of Tumor Growth and the Laboratory of Molecular Oncology

68 Leningradskaya St., Pesochny, Saint Petersburg, 197758, Russia

https://orcid.org/0000-0002-9375-7517РощинВ. Ю.RoshchinV. Yu.

врач-патологоанатом патологоанатомического отделения

Россия, 117997, Москва, ул. Саморы Машела, 1

Pathologist of the Pathology Department

1 Samory Mashela St., Moscow, 117997, Russia

vit1982@list.ruhttps://orcid.org/0000-0002-3767-4477ШаманскаяТ. В.ShamanskayaT. V.

к.м.н., руководитель отдела изучения эмбриональных опухолей Института онкологии, радиологии и ядерной медицины

Россия, 117997, Москва, ул. Саморы Машела, 1

Cand. of Sci. (Med.), Head of the Department of Embryonic Tumors Research of the Institute of Oncology, Radiology and Nuclear Medicine

1 Samory Mashela St., Moscow, 117997, Russia

shamanskaya.tatyana@gmail.comhttps://orcid.org/0000-0001-6131-1783ВарфоломееваС. Р.VarfolomeevaS. R.

д.м.н., профессор, заместитель директора по научной и лечебной работе – директор НИИ детской онкологии и гематологии аппарата управления

115478, Москва, Каширское шоссе, 23

Dr. of Sci. (Med.), Professor, Deputy Director for Research and Clinical Work – Director of the Research Institute of Pediatric Oncology and Hematology

23 Kashirskoe Shosse, Moscow, 115478, Russia

s.varfolomeeva@ronc.ruФГБУ «Национальный медицинский исследовательский центр детской гематологии, онкологии и иммунологии им. Дмитрия Рогачева» Минздрава России; ФГБУ «НМИЦ онкологии им. Н.Н. Блохина» Минздрава РоссииРоссияDmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology, Ministry of Health of Russia; N.N. Blokhin National Medical Research Centre of Oncology, Ministry of Health of RussiaRussian FederationФГБУ «Национальный медицинский исследовательский центр детской гематологии, онкологии и иммунологии им. Дмитрия Рогачева» Минздрава РоссииРоссияDmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology, Ministry of Health of RussiaRussian FederationФГБУ «Национальный медицинский исследовательский центр онкологии им. Н.Н. Петрова» Минздрава РоссииРоссияN.N. Petrov National Medical Research Center of Oncology, Ministry of Health of RussiaRussian FederationФГБУ «НМИЦ онкологии им. Н.Н. Блохина» Минздрава РоссииРоссияN.N. Blokhin National Medical Research Centre of Oncology, Ministry of Health of RussiaRussian Federation202004072020726477Copyright © Сулейманова А.М., Качанов Д.Ю., Имянитов Е.Н., Рощин В.Ю., Шаманская Т.В., Варфоломеева С.Р., 20202020Сулейманова А.М., Качанов Д.Ю., Имянитов Е.Н., Рощин В.Ю., Шаманская Т.В., Варфоломеева С.Р.Suleymanova A.M., Kachanov D.Y., Imyanitov E.N., Roshchin V.Y., Shamanskaya T.V., Varfolomeeva S.R.Данная работа распространяется под лицензией Creative Commons Attribution 4.0.This work is licensed under a Creative Commons Attribution 4.0 License.https://journal.nodgo.org/jour/article/view/605

Воспалительная миофибробластическая опухоль (ВМО) – редкий вид новообразования с неопределенным биологическим потенциалом. ВМО могут встречаться как в детском, так и во взрослом возрасте. Стандартом терапии является радикальное хирургическое лечение, однако для пациентов с неоперабельными/рецидивирующими или метастатическими формами ВМО терапевтические опции весьма ограничены. В данном обзоре литературы описаны специфические клинические, морфологические и биологические характеристики данного новообразования, приведены современные подходы диагностики и лечения ВМО.

Inflammatory myofibroblastic tumor (IMT) is a rare type of neoplasm with undetermined biological potential. IMT can occur in both childhood and adulthood. The standard of therapy is radical surgical treatment, but for patients with inoperable/recurrent or metastatic forms of IMT, therapeutic options are very limited. This literature review describes specific clinical, morphological and biological characteristics of this neoplasm, provides modern approaches to the diagnosis and treatment of IMT.

воспалительная миофибробластическая опухольдетиредкие опухолиген ALKinflammatory myofibroblastic tumorschildrenrare tumorsgene ALKReferencesBrunn H. Two interesting benign lung tumours of contradictory histopathology: remarks on the necessity for maintaining the chest tumour registry. J Thorac Surg 1939;9:119–31.Brunn H. Two interesting benign lung tumours of contradictory histopathology: remarks on the necessity for maintaining the chest tumour registry. J Thorac Surg 1939;9:119–31.Lane J.D., Krohn S., Kolozzi W., Whitehead R.E. Plasma cell granuloma of the lung. Dis Chest 1955;27:216–21. doi: 10.1378/chest.27.2.216.Lane J.D., Krohn S., Kolozzi W., Whitehead R.E. Plasma cell granuloma of the lung. Dis Chest 1955;27:216–21. doi: 10.1378/chest.27.2.216.Meis J.M., Enzinger F.M. Infl ammatory fi brosarcoma of the mesentery and retroperitoneum. A tumor closely simulating inflammatory pseudotumor. Am J Surg Pathol 1991;15(12):1146–56. doi: 10.1097/00000478-199112000-00005.Meis J.M., Enzinger F.M. Infl ammatory fi brosarcoma of the mesentery and retroperitoneum. A tumor closely simulating inflammatory pseudotumor. Am J Surg Pathol 1991;15(12):1146–56. doi: 10.1097/00000478-199112000-00005.Coffi n C.M., Watterson J., Priest J.R., Dehner L.P. 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.Coffi n C.M., Watterson J., Priest J.R., Dehner L.P. 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.Alaggio R., Cecchetto G., Bisogno G., Gambini C., Calabrò M.L., Inserra A., Boldrini R., De Salvo G.L., G d’Amore E.S., Dall’igna P. Inflammatory myofibroblastic tumors in childhood: a report from the Italian Cooperative Group Studies. Cancer 2010;116(1):216–26. doi: 10.1002/cncr.24684.Alaggio R., Cecchetto G., Bisogno G., Gambini C., Calabrò M.L., Inserra A., Boldrini R., De Salvo G.L., G d’Amore E.S., Dall’igna P. Inflammatory myofibroblastic tumors in childhood: a report from the Italian Cooperative Group Studies. Cancer 2010;116(1):216–26. doi: 10.1002/cncr.24684.Kube S., Vokuhl C., Dantonello T., Scheer M., Hallmen E., Feuchtgruber S., Escherich G., Niggli F., Kuehnle I., von Kalle T., Bielack S., Klingebiel T., Koscielniak E. Inflammatory myofibroblastic tumors – a retrospective analysis of the Cooperative Weichteilsarkom Studiengruppe. Pediatr Blood Cancer 2018;65(6):e27012. doi: 10.1002/pbc.27012.Kube S., Vokuhl C., Dantonello T., Scheer M., Hallmen E., Feuchtgruber S., Escherich G., Niggli F., Kuehnle I., von Kalle T., Bielack S., Klingebiel T., Koscielniak E. Inflammatory myofibroblastic tumors – a retrospective analysis of the Cooperative Weichteilsarkom Studiengruppe. Pediatr Blood Cancer 2018;65(6):e27012. doi: 10.1002/pbc.27012.Casanova M., Brennan B., Alaggio R., Kelsey A., Orbach D., van Noesel M.M., Corradini N., Minard-Colin V., Zanetti I., Bisogno G., Gallego S., Merks J.H.M., De Salvo G.L., Ferrari A. 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.Casanova M., Brennan B., Alaggio R., Kelsey A., Orbach D., van Noesel M.M., Corradini N., Minard-Colin V., Zanetti I., Bisogno G., Gallego S., Merks J.H.M., De Salvo G.L., Ferrari A. 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.Goldblum J.R., Golpe A.L., Weiss S.W. Benign fibroblastic/myofibroblastic proliferations, including superficial fibromatoses. In: Enzinger and Weiss’s soft tissue tumors. 6th ed. Elsevier Saunders, 2014.Goldblum J.R., Golpe A.L., Weiss S.W. Benign fibroblastic/myofibroblastic proliferations, including superficial fibromatoses. In: Enzinger and Weiss’s soft tissue tumors. 6th ed. Elsevier Saunders, 2014.Coffi n C.M., Dehner L.P., Meis-Kindblom J.M. Inflammatory myofi broblastic tumor, inflammatory fibrosarcoma, and related lesions: an historical review with differential diagnostic considerations. Semin Diagn Pathol 1998;15(2):102–10. PMID: 9606802.Coffi n C.M., Dehner L.P., Meis-Kindblom J.M. Inflammatory myofi broblastic tumor, inflammatory fibrosarcoma, and related lesions: an historical review with differential diagnostic considerations. Semin Diagn Pathol 1998;15(2):102–10. PMID: 9606802.Matsubara O., Tan-Liu N.S., Kenney R.M., Mark E.J. Inflammatory pseudotumors of the lung: progression from organizing pneumonia to fi brous histiocytoma or to plasma cell granuloma in 32 cases. Hum Pathol 1988;19(7):807–14. doi: 10.1016/s0046-8177(88)80264-8.Matsubara O., Tan-Liu N.S., Kenney R.M., Mark E.J. Inflammatory pseudotumors of the lung: progression from organizing pneumonia to fi brous histiocytoma or to plasma cell granuloma in 32 cases. Hum Pathol 1988;19(7):807–14. doi: 10.1016/s0046-8177(88)80264-8.Coffi n C.M., Alaggio R. Fibroblastic and myofi broblastic tumors in children and adolescents. Pediatr Dev Pathol 2012;15(1 Suppl):127–80. doi: 10.2350/10-12-0944-PB.1.Coffi n C.M., Alaggio R. Fibroblastic and myofi broblastic tumors in children and adolescents. Pediatr Dev Pathol 2012;15(1 Suppl):127–80. doi: 10.2350/10-12-0944-PB.1.Lopez-Nunez O., John I., Panasiti R.N., Ranganathan S., Santoro L., Grélaud D., Wu T., Buccoliero A.M., Casanova M., Alaggio R., Surrey L.F. Infantile inflammatory myofibroblastic tumors: clinicopathological and molecular characterization of 12 cases. Mod Pathol 2020;33(4):576–90. doi: 10.1038/s41379-019-0406-6.Lopez-Nunez O., John I., Panasiti R.N., Ranganathan S., Santoro L., Grélaud D., Wu T., Buccoliero A.M., Casanova M., Alaggio R., Surrey L.F. Infantile inflammatory myofibroblastic tumors: clinicopathological and molecular characterization of 12 cases. Mod Pathol 2020;33(4):576–90. doi: 10.1038/s41379-019-0406-6.Haimes J.D., Stewart C.J.R., Kudlow B.A., Culver B.P., Meng B., Koay E., Whitehouse A., Cope N., Lee J.C., Ng T., McCluggage W.G., Lee C.H. Uterine inflammatory myofibroblastic tumors frequently harbor ALK fusions with IGFBP5 and THBS1. Am J Surg Pathol 2017;41(6):773–80. doi: 10.1097/PAS.0000000000000801.Haimes J.D., Stewart C.J.R., Kudlow B.A., Culver B.P., Meng B., Koay E., Whitehouse A., Cope N., Lee J.C., Ng T., McCluggage W.G., Lee C.H. Uterine inflammatory myofibroblastic tumors frequently harbor ALK fusions with IGFBP5 and THBS1. Am J Surg Pathol 2017;41(6):773–80. doi: 10.1097/PAS.0000000000000801.Kling E., Kutys R., Virmani R., Miettinen M. Cardiac infl ammatory myofi broblastic tumor: a “benign” neoplasm that may result in syncope, myocardial infarction, and sudden death. Am J Surg Pathol 2007;31(7):1115–22. doi: 10.1097/PAS.0b013e31802d68ff .Kling E., Kutys R., Virmani R., Miettinen M. Cardiac infl ammatory myofi broblastic tumor: a “benign” neoplasm that may result in syncope, myocardial infarction, and sudden death. Am J Surg Pathol 2007;31(7):1115–22. doi: 10.1097/PAS.0b013e31802d68ff .Liu H.K., Lin Y.C., Yeh M.L., Chen Y.S., Su Y.T., Tsai C.C. Infl ammatory myofi broblastic tumors of the pancreas in children. A case report and literature review. Medicine (Baltimore) 2017;96(2):e5870. doi: 10.1097/MD.0000000000005870.Liu H.K., Lin Y.C., Yeh M.L., Chen Y.S., Su Y.T., Tsai C.C. Infl ammatory myofi broblastic tumors of the pancreas in children. A case report and literature review. Medicine (Baltimore) 2017;96(2):e5870. doi: 10.1097/MD.0000000000005870.Bumber Z., Jurlina M., Manojlović S., Jakić-Razumović J. Infl ammatory pseudotumor of the trachea. J Pediatr Surg 2001;36(4):631–4. doi: 10.1053/jpsu.2001.22306.Bumber Z., Jurlina M., Manojlović S., Jakić-Razumović J. Infl ammatory pseudotumor of the trachea. J Pediatr Surg 2001;36(4):631–4. doi: 10.1053/jpsu.2001.22306.Al-Humidi A., Al-Khamiss A. Infl ammatory myofi broblastic tumor arising in the external ear: unexpected location (case report). Int J Health Sci (Qassim) 2015;9(2):201–5. PMID: 26309441.Al-Humidi A., Al-Khamiss A. Infl ammatory myofi broblastic tumor arising in the external ear: unexpected location (case report). Int J Health Sci (Qassim) 2015;9(2):201–5. PMID: 26309441.Lourenço S.V., Boggio P., Simonsen Nico M.M. Infl ammatory myofi broblastic tumor of the tongue: report of an unusual case in a teenage patient. Dermatol Online J 2012;18(5):6. PMID: 22630576.Lourenço S.V., Boggio P., Simonsen Nico M.M. Infl ammatory myofi broblastic tumor of the tongue: report of an unusual case in a teenage patient. Dermatol Online J 2012;18(5):6. PMID: 22630576.Качанов Д.Ю., Шаманская Т.В., Сулейманова А.М., Меркулов Н.Н., Феоктистова Е.В., Талыпов С.Р., Рощин В.Ю., Щербаков А.П., Терещенко Г.В., Казакова А.Н., Варфоломеева С.Р. Воспалительная миофибробластическая опухоль брыжейки тонкой кишки, осложнившаяся острой кишечной непроходимостью. Вопросы гематологии/онкологии и иммунопатологии в педиатрии 2017;16(1):54–61. doi: 10.24287/1726-1708-2017-16-1-54-61.Kachanov D.Y., Shamanskaya T.V., Suleymanova A.M., Merkulov N.N., Feoktistova E.V., Talypov S.R., Roschin V.Y., Shcherbakov A.P., Tereschenko G.V., Kazakova A.N., Varfolomeeva S.R. Infl ammatory myofi broblastic tumor of the small bowel mesentery complicated by the intestinal obstruction. Voprosy gematologii/ onkologii i immunopatologii v pediatrii = Pediatric Hematology/Oncology and Immunopathology. 2017;16(1):54–61. (In Russ.)Souid A.K., Ziemba M.C., Dubansky A.S., Mazur M., Oliphant M., Thomas F.D., Ratner M., Sadowitz P.D. Inflammatory myofibroblastic tumor in children. Cancer 1993;72(6):2042–8. doi: 10.1002/10970142(19930915)72:6<2042::aid-cncr2820720641>3.0.co;2-i.Souid A.K., Ziemba M.C., Dubansky A.S., Mazur M., Oliphant M., Thomas F.D., Ratner M., Sadowitz P.D. Inflammatory myofibroblastic tumor in children. Cancer 1993;72(6):2042–8. doi: 10.1002/10970142(19930915)72:6<2042::aid-cncr2820720641>3.0.co;2-i.Coffin C.M., Fletcher J.A. Inflammatory myofibroblastic tumour. WHO Classification of Tumours of Soft Tissue and Bone. WHO/IARC Classification of Tumours, 4th ed., vol. 5. Fletcher C.D.M., Bridge J.A., Hogendoorn P.C.W., Mertens F. (eds.) International Agency for Research on Cancer. Lyon, 2013. Pр. 83–86.Coffin C.M., Fletcher J.A. Inflammatory myofibroblastic tumour. WHO Classification of Tumours of Soft Tissue and Bone. WHO/IARC Classification of Tumours, 4th ed., vol. 5. Fletcher C.D.M., Bridge J.A., Hogendoorn P.C.W., Mertens F. (eds.) International Agency for Research on Cancer. Lyon, 2013. Pр. 83–86.Hagenstad C.T., Kilpatrick S.E., Pettenati M.J., Savage P.D. Inflammatory myofibroblastic tumor with bone marrow involvement. A case report and review of the literature. Arch Pathol Lab Med 2003;127(7):865–7. doi: 10.1043/1543-2165(2003)127<865:IMTWBM>2.0.CO;2.Hagenstad C.T., Kilpatrick S.E., Pettenati M.J., Savage P.D. Inflammatory myofibroblastic tumor with bone marrow involvement. A case report and review of the literature. Arch Pathol Lab Med 2003;127(7):865–7. doi: 10.1043/1543-2165(2003)127<865:IMTWBM>2.0.CO;2.Debelenko L.V., Arthur D.C., Pack S.D., Helman L.J., Schrump D.S., Tsokos M. Identification of CARS-ALK fusion in primary and metastatic lesions of an inflammatory myofibroblastic tumor. Lab Invest 2003;83(9):1255–65. doi: 10.1097/01.lab.0000088856.49388.ea.Debelenko L.V., Arthur D.C., Pack S.D., Helman L.J., Schrump D.S., Tsokos M. Identification of CARS-ALK fusion in primary and metastatic lesions of an inflammatory myofibroblastic tumor. Lab Invest 2003;83(9):1255–65. doi: 10.1097/01.lab.0000088856.49388.ea.Fangusaro J., Klopfenstein K., Groner J., Hammond S., Altura R.A. Inflammatory myofi broblastic tumor following hematopoietic stem cell transplantation: report of two pediatric cases. Bone Marrow Transplant 2004;33(1):103–7. doi: 10.1038/sj.bmt.1704292.Fangusaro J., Klopfenstein K., Groner J., Hammond S., Altura R.A. Inflammatory myofi broblastic tumor following hematopoietic stem cell transplantation: report of two pediatric cases. Bone Marrow Transplant 2004;33(1):103–7. doi: 10.1038/sj.bmt.1704292.Cook J.R., Dehner L.P., Collins M.H., Ma Z., Morris S.W., Coffi n C.M., Hill D.A. Anaplastic lymphoma kinase (ALK) expression in the inflammatory myofibroblastic tumor: a comparative immunohistochemical study. Am J Surg Pathol 2001;25(11):1364–71. doi: 10.1097/00000478-200111000-00003.Cook J.R., Dehner L.P., Collins M.H., Ma Z., Morris S.W., Coffi n C.M., Hill D.A. Anaplastic lymphoma kinase (ALK) expression in the inflammatory myofibroblastic tumor: a comparative immunohistochemical study. Am J Surg Pathol 2001;25(11):1364–71. doi: 10.1097/00000478-200111000-00003.Mariño-Enríquez A., Wang W.L., Roy A., Lopez-Terrada D., Lazar A.J., Fletcher C.D., Coffin C.M., Hornick J.L. Epithelioid inflammatory myofibroblastic sarcoma: an aggressive intra-abdominal variant of infl ammatory myofi broblastic tumor with nuclear membrane or perinuclear ALK. Am J Surg Pathol 2011;35(1):135–44. doi: 10.1097/PAS.0b013e318200cfd5.Mariño-Enríquez A., Wang W.L., Roy A., Lopez-Terrada D., Lazar A.J., Fletcher C.D., Coffin C.M., Hornick J.L. Epithelioid inflammatory myofibroblastic sarcoma: an aggressive intra-abdominal variant of infl ammatory myofi broblastic tumor with nuclear membrane or perinuclear ALK. Am J Surg Pathol 2011;35(1):135–44. doi: 10.1097/PAS.0b013e318200cfd5.Coffi n C.M., Hornick J.L., Fletcher C.D. Infl ammatory myofi broblastic 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.Coffi n C.M., Hornick J.L., Fletcher C.D. Infl ammatory myofi broblastic 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.Gleason B.C., Hornick J.L. Inflammatory myofibroblastic tumours: where are we now? J Clin Pathol 2008;61(4):428–37. doi: 10.1136/jcp.2007.049387.Gleason B.C., Hornick J.L. Inflammatory myofibroblastic tumours: where are we now? J Clin Pathol 2008;61(4):428–37. doi: 10.1136/jcp.2007.049387.Chen C.H., Lin R.L., Liu H.C., Chen C.H., Hung T.T., Huang W.C. Inflammatory myofibroblastic tumor mimicking anterior mediastinal malignancy. Ann Thorac Surg 2008;86(4):1362–4. doi: 10.1016/j.athoracsur.2008.03.031.Chen C.H., Lin R.L., Liu H.C., Chen C.H., Hung T.T., Huang W.C. Inflammatory myofibroblastic tumor mimicking anterior mediastinal malignancy. Ann Thorac Surg 2008;86(4):1362–4. doi: 10.1016/j.athoracsur.2008.03.031.Hornick J.L., Sholl L.M., Dal Cin P., Childress M.A., Lovly C.M. Expression of ROS1 predicts ROS1 gene rearrangement in inflammatory myofi broblastic tumors. Mod Pathol 2015;28(5):732–9. doi: 10.1038/modpathol.2014.165.Hornick J.L., Sholl L.M., Dal Cin P., Childress M.A., Lovly C.M. Expression of ROS1 predicts ROS1 gene rearrangement in inflammatory myofi broblastic tumors. Mod Pathol 2015;28(5):732–9. doi: 10.1038/modpathol.2014.165.Gatalica Z., Xiu J., Swensen J., Vranic S. Molecular characterization of cancers with NTRK gene fusions. Mod Pathol 2019;32(1):147–53. doi: 10.1038/s41379-018-0118-3.Gatalica Z., Xiu J., Swensen J., Vranic S. Molecular characterization of cancers with NTRK gene fusions. Mod Pathol 2019;32(1):147–53. doi: 10.1038/s41379-018-0118-3.Cottrell T.R., Duong A.T., Gocke C.D., Xu H., Ogurtsova A., Taube J.M., Belchis D.A. PD-L1 expression in inflammatory myofibroblastic tumors. Mod Pathol 2018;31(7):1155–63. doi: 10.1038/s41379-018-0034-6.Cottrell T.R., Duong A.T., Gocke C.D., Xu H., Ogurtsova A., Taube J.M., Belchis D.A. PD-L1 expression in inflammatory myofibroblastic tumors. Mod Pathol 2018;31(7):1155–63. doi: 10.1038/s41379-018-0034-6.Zen Y., Kitagawa S., Minato H., Kurumaya H., Katayanagi K., Masuda S., Niwa H., Fujimura M., Nakanuma Y. IgG4-positive plasma cells in inflammatory pseudotumor (plasma cell granuloma) of the lung. Hum Pathol 2005;36(7):710–7. doi: 10.1016/j.humpath.2005.05.011.Zen Y., Kitagawa S., Minato H., Kurumaya H., Katayanagi K., Masuda S., Niwa H., Fujimura M., Nakanuma Y. IgG4-positive plasma cells in inflammatory pseudotumor (plasma cell granuloma) of the lung. Hum Pathol 2005;36(7):710–7. doi: 10.1016/j.humpath.2005.05.011.Griffi n C.A., Hawkins A.L., Dvorak C., Henkle C., Ellingham T., Perlman E.J. Recurrent involvement of 2p23 in infl ammatory myofi broblastic tumors. Cancer Res 1999;59(12):2776–80. PMID: 10383129.Griffi n C.A., Hawkins A.L., Dvorak C., Henkle C., Ellingham T., Perlman E.J. Recurrent involvement of 2p23 in infl ammatory myofi broblastic tumors. Cancer Res 1999;59(12):2776–80. PMID: 10383129.Sokai A., Enaka M., Sokai R., Mori S., Gunji M., Fujino M., Ito M. Pulmonary inflammatory myofibroblastic tumor harboring EML4-ALK fusion gene. Jpn J Clin Oncol 2014;44(1):93–6. doi: 10.1093/jjco/hyt173.Sokai A., Enaka M., Sokai R., Mori S., Gunji M., Fujino M., Ito M. Pulmonary inflammatory myofibroblastic tumor harboring EML4-ALK fusion gene. Jpn J Clin Oncol 2014;44(1):93–6. doi: 10.1093/jjco/hyt173.Antonescu C.R., Suurmeijer A.J., Zhang L., Sung Y.S., Jungbluth A.A., Travis W.D., Al-Ahmadie H., Fletcher C.D., Alaggio R. Molecular characterization of infl ammatory myofi broblastic tumors with frequent ALK and ROS1 fusions and rare novel RET gene rearrangement. Am J Surg Pathol 2015;39(7):957–67. doi: 10.1097/PAS.0000000000000404.Antonescu C.R., Suurmeijer A.J., Zhang L., Sung Y.S., Jungbluth A.A., Travis W.D., Al-Ahmadie H., Fletcher C.D., Alaggio R. Molecular characterization of infl ammatory myofi broblastic tumors with frequent ALK and ROS1 fusions and rare novel RET gene rearrangement. Am J Surg Pathol 2015;39(7):957–67. doi: 10.1097/PAS.0000000000000404.Coffi n C.M., Patel A., Perkins S., Elenitoba-Johnson K.S., Perlman E., Griffi n C.A. ALK1 and p80 expression and chromosomal rearrangements involving 2p23 in inflammatory myofibroblastic tumor. Mod Pathol 2001;14(6):569–76. doi: 10.1038/modpathol.3880352.Coffi n C.M., Patel A., Perkins S., Elenitoba-Johnson K.S., Perlman E., Griffi n C.A. ALK1 and p80 expression and chromosomal rearrangements involving 2p23 in inflammatory myofibroblastic tumor. Mod Pathol 2001;14(6):569–76. doi: 10.1038/modpathol.3880352.Lawrence B., Perez-Atayde A., Hibbard M.K., Rubin B.P., Dal Cin P., Pinkus J.L., Pinkus G.S., Xiao S., Yi E.S., Fletcher C.D., Fletcher J.A. TPM3-ALK and TPM4-ALK oncogenes in inflammatory myofibroblastic tumors. Am J Pathol 2000;157(2):377–84. doi: 10.1016/S0002-9440(10)64550-6.Lawrence B., Perez-Atayde A., Hibbard M.K., Rubin B.P., Dal Cin P., Pinkus J.L., Pinkus G.S., Xiao S., Yi E.S., Fletcher C.D., Fletcher J.A. TPM3-ALK and TPM4-ALK oncogenes in inflammatory myofibroblastic tumors. Am J Pathol 2000;157(2):377–84. doi: 10.1016/S0002-9440(10)64550-6.Lovly C.M., Gupta A., Lipson D., Otto G., Brennan T., Chung C.T., Borinstein S.C., Ross J.S., Stephens P.J., Miller V.A., Coffi n C.M. Inflammatory myofibroblastic tumors harbor multiple potentially actionable kinase fusions. Cancer Discov 2014;4(8):889–95. doi: 10.1158/2159-8290.CD-14-0377.Lovly C.M., Gupta A., Lipson D., Otto G., Brennan T., Chung C.T., Borinstein S.C., Ross J.S., Stephens P.J., Miller V.A., Coffi n C.M. Inflammatory myofibroblastic tumors harbor multiple potentially actionable kinase fusions. Cancer Discov 2014;4(8):889–95. doi: 10.1158/2159-8290.CD-14-0377.Chang J.C., Zhang L., Drilon A.E., Chi P., Alaggio R., Borsu L., Benayed R., Travis W.D., Ladanyi M., Antonescu C.R. Thoracic infl ammatory myofi broblastic tumors beyond ALK gene rearrangements. J Thorac Oncol 2019;14(5):825–34. doi: 10.1016/j.jtho.2018.12.003.Chang J.C., Zhang L., Drilon A.E., Chi P., Alaggio R., Borsu L., Benayed R., Travis W.D., Ladanyi M., Antonescu C.R. Thoracic infl ammatory myofi broblastic tumors beyond ALK gene rearrangements. J Thorac Oncol 2019;14(5):825–34. doi: 10.1016/j.jtho.2018.12.003.Baldi G.G., Gronchi A., Vincenzi B., Martino De Pas T., Pantaleo M.A., D’Ambrosio L., Grignani G., Casanova M., Ferrari A., Simeone N., Provenzano S., Dei Tos A.P., Sbaraglia M., Collini P., Dagrada G., Morosi C., Casali P.G., Stacchiotti S. Activity of chemotherapy in infl ammatory myofi broblastic tumor (IMT): A retrospective analysis within the Italian Rare Tumours Network (RTR). J Clin Oncol 2019;37(15 suppl). doi: 10.1200/JCO.2019.37.15_suppl.e22545.Baldi G.G., Gronchi A., Vincenzi B., Martino De Pas T., Pantaleo M.A., D’Ambrosio L., Grignani G., Casanova M., Ferrari A., Simeone N., Provenzano S., Dei Tos A.P., Sbaraglia M., Collini P., Dagrada G., Morosi C., Casali P.G., Stacchiotti S. Activity of chemotherapy in infl ammatory myofi broblastic tumor (IMT): A retrospective analysis within the Italian Rare Tumours Network (RTR). J Clin Oncol 2019;37(15 suppl). doi: 10.1200/JCO.2019.37.15_suppl.e22545.Applebaum H., Kieran M.W., Cripe T.P., Coffi n C.M., Collins M.H., Kaipainen A., Laforme A., Shamberger R.C. The rationale for nonsteroidal anti-infl ammatory drug therapy for infl ammatory myofi broblastic tumors: a Children’s Oncology Group Study. J Pediatr Surg 2005;40(6):999–1003;discussion 1003. doi: 10.1016/j.jpedsurg.2005.03.016.Applebaum H., Kieran M.W., Cripe T.P., Coffi n C.M., Collins M.H., Kaipainen A., Laforme A., Shamberger R.C. The rationale for nonsteroidal anti-infl ammatory drug therapy for infl ammatory myofi broblastic tumors: a Children’s Oncology Group Study. J Pediatr Surg 2005;40(6):999–1003;discussion 1003. doi: 10.1016/j.jpedsurg.2005.03.016.Maire J.P., Eimer S., San Galli F., Franco-Vidal V., Galland-Girodet S., Huchet A., Darrouzet V. Infl ammatory myofi broblastic tumour of the skull base. Case Rep Otolaryngol 2013;2013:103646. doi: 10.1155/2013/103646.Maire J.P., Eimer S., San Galli F., Franco-Vidal V., Galland-Girodet S., Huchet A., Darrouzet V. Infl ammatory myofi broblastic tumour of the skull base. Case Rep Otolaryngol 2013;2013:103646. doi: 10.1155/2013/103646.Sasagawa Y., Akai T., Itou S., Iizuka H. Multiple intraosseous infl ammatory myofi broblastic tumors presenting with an aggressive clinical course: case report. Neurosurgery 2011;69(4):E1010–5;discussion E1015-6. doi: 10.1227/NEU.0b013e318223b651.Sasagawa Y., Akai T., Itou S., Iizuka H. Multiple intraosseous infl ammatory myofi broblastic tumors presenting with an aggressive clinical course: case report. Neurosurgery 2011;69(4):E1010–5;discussion E1015-6. doi: 10.1227/NEU.0b013e318223b651.Butrynski J.E., D’Adamo D.R., Hornick J.L., Dal Cin P., Antonescu C.R., Jhanwar S.C., Ladanyi M., Capelletti M., Rodig S.J., Ramaiya N., Kwak E.L., Clark J.W., Wilner K.D., Christensen J.G., Jänne P.A., Maki R.G., Demetri G.D., Shapiro G.I. Crizotinib in ALK-rearranged infl ammatory myofi broblastic tumor. N Engl J Med 2010;363(18):1727–33. doi: 10.1056/NEJMoa1007056.Butrynski J.E., D’Adamo D.R., Hornick J.L., Dal Cin P., Antonescu C.R., Jhanwar S.C., Ladanyi M., Capelletti M., Rodig S.J., Ramaiya N., Kwak E.L., Clark J.W., Wilner K.D., Christensen J.G., Jänne P.A., Maki R.G., Demetri G.D., Shapiro G.I. Crizotinib in ALK-rearranged infl ammatory myofi broblastic tumor. N Engl J Med 2010;363(18):1727–33. doi: 10.1056/NEJMoa1007056.Mossé Y.P., Lim M.S., Voss S.D., Wilner K., Ruff ner K., Laliberte J., Rolland D., Balis F.M., Maris J.M., Weigel B.J., Ingle A.M., Ahern C., Adamson P.C., Blaney S.M. Safety and activity of crizotinib for paediatric patients with refractory solid tumours or anaplastic large-cell lymphoma: a Children’s Oncology Group phase 1 consortium study. Lancet Oncol 2013;14(6):472–80. doi: 10.1016/S1470-2045(13)70095-0.Mossé Y.P., Lim M.S., Voss S.D., Wilner K., Ruff ner K., Laliberte J., Rolland D., Balis F.M., Maris J.M., Weigel B.J., Ingle A.M., Ahern C., Adamson P.C., Blaney S.M. Safety and activity of crizotinib for paediatric patients with refractory solid tumours or anaplastic large-cell lymphoma: a Children’s Oncology Group phase 1 consortium study. Lancet Oncol 2013;14(6):472–80. doi: 10.1016/S1470-2045(13)70095-0.Mossé Y.P., Voss S.D., Lim M.S., Rolland D., Minard C.G., Fox E., Adamson P., Wilner K., Blaney S.M., Weigel B.J. Targeting ALK with crizotinib in pediatric anaplastic large cell lymphoma and infl ammatory myofi broblastic tumor: a Children’s Oncology Group Study. J Clin Oncol 2017;35(28):3215–21. doi: 10.1200/JCO.2017.73.4830.Mossé Y.P., Voss S.D., Lim M.S., Rolland D., Minard C.G., Fox E., Adamson P., Wilner K., Blaney S.M., Weigel B.J. Targeting ALK with crizotinib in pediatric anaplastic large cell lymphoma and infl ammatory myofi broblastic tumor: a Children’s Oncology Group Study. J Clin Oncol 2017;35(28):3215–21. doi: 10.1200/JCO.2017.73.4830.Schöff ski P., Sufl iarsky J., Gelderblom H., Blay J.Y., Strauss S.J., Stacchiotti S., Rutkowski P., Lindner L.H., Leahy M.G., Italiano A., Isambert N., Debiec-Rychter M., Sciot R., Van Cann T., Marréaud S., Nzokirantevye A., Collette S., Wozniak A. Crizotinib in patients with advanced, inoperable infl ammatory myofi broblastic 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.Schöff ski P., Sufl iarsky J., Gelderblom H., Blay J.Y., Strauss S.J., Stacchiotti S., Rutkowski P., Lindner L.H., Leahy M.G., Italiano A., Isambert N., Debiec-Rychter M., Sciot R., Van Cann T., Marréaud S., Nzokirantevye A., Collette S., Wozniak A. Crizotinib in patients with advanced, inoperable infl ammatory myofi broblastic 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.Brivio E., Zwaan C.M. ALK inhibition in two emblematic cases of pediatric infl ammatory myofi broblastic tumor: Effi cacy and side eff ects. Pediatr Blood Cancer 2019;66(5):e27645. doi: 10.1002/pbc.27645.Brivio E., Zwaan C.M. ALK inhibition in two emblematic cases of pediatric infl ammatory myofi broblastic tumor: Effi cacy and side eff ects. Pediatr Blood Cancer 2019;66(5):e27645. doi: 10.1002/pbc.27645.Ambati S.R., Slotkin E.K., Chow-Maneval E., Basu E.M. Entrectinib in two pediatric patients with infl ammatory myofi broblastic tumors harboring ROS1 or ALK gene fusions. JCO Precis Oncol 2018;2018. doi:/10.1200/PO.18.00095.Ambati S.R., Slotkin E.K., Chow-Maneval E., Basu E.M. Entrectinib in two pediatric patients with infl ammatory myofi broblastic tumors harboring ROS1 or ALK gene fusions. JCO Precis Oncol 2018;2018. doi:/10.1200/PO.18.00095.Parker B.M., Parker J.V., Lymperopoulos A., Konda V. A case report: pharmacology and resistance patterns of three generations of ALK inhibitors in metastatic infl ammatory myofi broblastic sarcoma. J Oncol Pharm Pract 2019;25(5):1226–30. doi: 10.1177/1078155218781944.Parker B.M., Parker J.V., Lymperopoulos A., Konda V. A case report: pharmacology and resistance patterns of three generations of ALK inhibitors in metastatic infl ammatory myofi broblastic sarcoma. J Oncol Pharm Pract 2019;25(5):1226–30. doi: 10.1177/1078155218781944.Alassiri A.H., Ali R.H., Shen Y., Lum A., Strahlendorf C., Deyell R., Rassekh R., Sorensen P.H., Laskin J., Marra M., Yip S., Lee C.H., Ng T.L. ETV6-NTRK3 is expressed in a subset of ALK-negative infl ammatory myofi broblastic tumors. Am J Surg Pathol 2016;40(8):1051–61. doi: 10.1097/PAS.0000000000000677.Alassiri A.H., Ali R.H., Shen Y., Lum A., Strahlendorf C., Deyell R., Rassekh R., Sorensen P.H., Laskin J., Marra M., Yip S., Lee C.H., Ng T.L. ETV6-NTRK3 is expressed in a subset of ALK-negative infl ammatory myofi broblastic tumors. Am J Surg Pathol 2016;40(8):1051–61. doi: 10.1097/PAS.0000000000000677.Schram A.M., Chang M.T., Jonsson P., Drilon A. Fusions in solid tumours: diagnostic strategies, targeted therapy, and acquired resistance. Nat Rev Clin Oncol 2017;14(12):735–48. doi: 10.1038/nrclinonc.2017.127.Schram A.M., Chang M.T., Jonsson P., Drilon A. Fusions in solid tumours: diagnostic strategies, targeted therapy, and acquired resistance. Nat Rev Clin Oncol 2017;14(12):735–48. doi: 10.1038/nrclinonc.2017.127.Shaw A.T., Friboulet L., Leshchiner I., Gainor J.F., Bergqvist S., Brooun A., Burke B.J., Deng Y.L., Liu W., Dardaei L., Frias R.L., Schultz K.R., Logan J., James L.P., Smeal T., Timofeevski S., Katayama R., Iafrate A.J., Le L., McTigue M., Getz G., Johnson T.W., Engelman J.A. Resensitization to crizotinib by the lorlatinib ALK resistance mutation L1198F. N Engl J Med 2016;374(1):54–61. doi: 10.1056/NEJMoa1508887.Shaw A.T., Friboulet L., Leshchiner I., Gainor J.F., Bergqvist S., Brooun A., Burke B.J., Deng Y.L., Liu W., Dardaei L., Frias R.L., Schultz K.R., Logan J., James L.P., Smeal T., Timofeevski S., Katayama R., Iafrate A.J., Le L., McTigue M., Getz G., Johnson T.W., Engelman J.A. Resensitization to crizotinib by the lorlatinib ALK resistance mutation L1198F. N Engl J Med 2016;374(1):54–61. doi: 10.1056/NEJMoa1508887.Cerfolio R.J., Allen M.S., Nascimento A.G., Deschamps C., Trastek V.F., Miller D.L., Pairolero P.C. Infl ammatory pseudotumors of the lung. Ann Thorac Surg 1999;67(4):933–6. doi: 10.1016/s0003-4975(99)00155-1.Cerfolio R.J., Allen M.S., Nascimento A.G., Deschamps C., Trastek V.F., Miller D.L., Pairolero P.C. Infl ammatory pseudotumors of the lung. Ann Thorac Surg 1999;67(4):933–6. doi: 10.1016/s0003-4975(99)00155-1.Chun Y.S., Wang L., Nascimento A.G., Moir C.R., Rodeberg D.A. Pediatric infl ammatory myofi broblastic tumor: anaplastic lymphoma kinase (ALK) expression and prognosis. Pediatr Blood Cancer 2005;45(6):796–801. doi: 10.1002/pbc.20294.Chun Y.S., Wang L., Nascimento A.G., Moir C.R., Rodeberg D.A. Pediatric infl ammatory myofi broblastic tumor: anaplastic lymphoma kinase (ALK) expression and prognosis. Pediatr Blood Cancer 2005;45(6):796–801. doi: 10.1002/pbc.20294.Morotti R.A., Legman M.D., Kerkar N., Pawel B.R., Sanger W.G., Coffi n C.M. Pediatric infl ammatory myofi broblastic tumor with late metastasis to the lung: case report and review of the literature. Pediatr Dev Pathol 2005;8(2):224–9. doi: 10.1007/s10024-004-8088-5.Morotti R.A., Legman M.D., Kerkar N., Pawel B.R., Sanger W.G., Coffi n C.M. Pediatric infl ammatory myofi broblastic tumor with late metastasis to the lung: case report and review of the literature. Pediatr Dev Pathol 2005;8(2):224–9. doi: 10.1007/s10024-004-8088-5.Hussong J.W., Brown M., Perkins S.L., Dehner L.P., Coffi n C.M. Comparison of DNA ploidy, histologic, and immunohistochemical fi ndings with clinical outcome in infl ammatory myofi broblastic tumors. Mod Pathol 1999;12(3):279–86. PMID: 10102613.Hussong J.W., Brown M., Perkins S.L., Dehner L.P., Coffi n C.M. Comparison of DNA ploidy, histologic, and immunohistochemical fi ndings with clinical outcome in infl ammatory myofi broblastic tumors. Mod Pathol 1999;12(3):279–86. PMID: 10102613.Yamamoto H., Yamaguchi H., Aishima S., Oda Y., Kohashi K., Oshiro Y., Tsuneyoshi M. Infl ammatory myofi broblastic tumor versus IgG4-related sclerosing disease and infl ammatory pseudotumor: a comparative clinicopathologic study. Am J Surg Pathol 2009;33(9):1330–40. doi: 10.1097/pas.0b013e3181a5a207.Yamamoto H., Yamaguchi H., Aishima S., Oda Y., Kohashi K., Oshiro Y., Tsuneyoshi M. Infl ammatory myofi broblastic tumor versus IgG4-related sclerosing disease and infl ammatory pseudotumor: a comparative clinicopathologic study. Am J Surg Pathol 2009;33(9):1330–40. doi: 10.1097/pas.0b013e3181a5a207.Biselli R., Ferlini C., Fattorossi A., Boldrini R., Bosman C. Infl ammatory myofi broblastic tumor (infl ammatory pseudotumor): DNA fl ow cytometric analysis of nine pediatric cases. Cancer 1996;77(4):778–84. doi: 10.1002/(sici)1097-0142(19960215)77:4<778::aid-cncr25>3.0.co;2-x.Biselli R., Ferlini C., Fattorossi A., Boldrini R., Bosman C. Infl ammatory myofi broblastic tumor (infl ammatory pseudotumor): DNA fl ow cytometric analysis of nine pediatric cases. Cancer 1996;77(4):778–84. doi: 10.1002/(sici)1097-0142(19960215)77:4<778::aid-cncr25>3.0.co;2-x.Donner L.R., Trompler R.A., White R.R. Progression of infl ammatory myofi broblastic tumor (infl ammatory pseudotumor) of soft tissue into sarcoma after several recurrences. Hum Pathol 1996;27(10):1095–8. doi: 10.1016/s0046-8177(96)90291-9.Donner L.R., Trompler R.A., White R.R. Progression of infl ammatory myofi broblastic tumor (infl ammatory pseudotumor) of soft tissue into sarcoma after several recurrences. Hum Pathol 1996;27(10):1095–8. doi: 10.1016/s0046-8177(96)90291-9.

The authors declare that there are no conflicts of interest present.