兒童罕見 <i>ROS1</i> 陽性上皮樣炎性肌纖維母細胞肉瘤診治一例_West China Medical Journal

Authors：

萬芊 ¹ , 伍家利 ² ,  徐珊玲 ³

1. ;
2. ;
3. ;

Corresponding?author：

徐珊玲, Email: xushanling_30@163.com

Keywords：

上皮樣炎性肌纖維母細胞肉瘤; 靶向治療; 基因檢測

DOI：

10.7507/1002-0179.202511300

Video：

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Citation： 萬芊, 伍家利, 徐珊玲. 兒童罕見 ROS1 陽性上皮樣炎性肌纖維母細胞肉瘤診治一例. West China Medical Journal, 2026, 41(1): 170-174. doi: 10.7507/1002-0179.202511300 Copy

1.	Mari?o-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): 135-144.
2.	Gros L, Dei Tos AP, Jones RL, et al. Inflammatory myofibroblastic tumour: state of the art. Cancers (Basel), 2022, 14(15): 3662.
3.	Zeng H, Li X, Chen S, et al. Anlotinib for the treatment of pulmonary epithelioid inflammatory myofibroblastic sarcoma: a case report. Anticancer Drugs, 2025, 36(8): 678-681.
4.	Maharjan D, Dehner C, Alani A, et al. Epithelioid inflammatory myofibroblastic sarcoma: case series with a first report of CLTC: : ALK fusion in an aggressive disease. Genes Chromosomes Cancer, 2025, 64(5): e70055.
5.	Cheng H, Lin Y, Zhu J, et al. Clinical features, treatment strategies, and prognosis of epithelioid inflammatory myofibroblastic sarcoma in children: a multicenter experience. Transl Pediatr, 2024, 13(2): 288-299.
6.	Chopra S, Maloney N, Wang WL. Epithelioid inflammatory myofibroblastic sarcoma with VCL-ALK fusion of central nervous system: case report and brief review of the literature. Brain Tumor Pathol, 2022, 39(1): 35-42.
7.	Singh P, Nambirajan A, Gaur MK, et al. Primary pulmonary epithelioid inflammatory myofibroblastic sarcoma: a rare entity and a literature review. J Pathol Transl Med, 2022, 56(4): 231-237.
8.	Collins K, Ramalingam P, Euscher ED, et al. Uterine inflammatory myofibroblastic neoplasms with aggressive behavior, including an epithelioid inflammatory myofibroblastic sarcoma: a clinicopathologic study of 9 cases. Am J Surg Pathol, 2022, 46(1): 105-117.
9.	Nayyar V, Bhutia O, Kakkar A, et al. Epithelioid inflammatory myofibroblastic sarcoma arising in the maxilla: first reported case in the jaws. Oral Surg Oral Med Oral Pathol Oral Radiol, 2023, 136(1): e15-e19.
10.	Azad M, Oye M, Torrente N, et al. Pericardial epithelioid inflammatory myofibroblastic sarcoma: an atypical presentation. Cureus, 2022, 14(7): e26827.
11.	喬一丹, 陶菁, 何樂建. 兒童上皮樣炎性肌纖維母細胞肉瘤 7 例臨床病理和分子遺傳學特征. 臨床與實驗病理學雜志, 2024, 40(7): 710-713.
12.	Okuda K, Watanabe T, Oda R, et al. Pulmonary inflammatory myofibroblastic tumor with TPM4-ALK translocation. J Thorac Dis, 2017, 9(11): E1013-E1017.
13.	PDQ Pediatric Treatment Editorial Board. Childhood soft tissue sarcoma treatment (PDQ^?): health professional version//PDQ cancer information summaries. Bethesda (MD): National Cancer Institute (US), 2002.
14.	Wachter F, Al-Ibraheemi A, Trissal MC, et al. Molecular characterization of inflammatory tumors facilitates initiation of effective therapy. Pediatrics, 2021, 148(6): e2021050990.
15.	Gambacorti-Passerini C, Orlov S, Zhang L, et al. Long-term effects of crizotinib in ALK-positive tumors (excluding NSCLC): a phase 1b open-label study. Am J Hematol, 2018, 93(5): 607-614.
16.	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): 3215-3221.
17.	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): 957-967.
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): 1586.
19.	Bai Y, Jiang M, Liang W, et al. Incomplete intestinal obstruction caused by a rare epithelioid inflammatory myofibroblastic sarcoma of the colon. Medicine, 2015, 94(51): e2342.
20.	Aminimoghaddam S, Pourali R. Epithelioid inflammatory myofibroblastic sarcoma with poor response to crizotinib: a case report. Clin Med Insights Case Rep, 2023, 16: 11795476231163954.
21.	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.
22.	孫佳凡, 韓素萍, 王聰, 等. 靶向治療盆腹腔上皮樣炎性肌纖維母細胞肉瘤一例. 國際婦產科學雜志, 2024, 51(2): 236-240.
23.	肖法嫚, 李余發, 汪斌超. 阿來替尼治療罕見 EML4-ALK 融合肺上皮樣炎性肌纖維母細胞肉瘤 1 例. 中國腫瘤臨床, 2023, 50(13): 701-702.

1. Mari?o-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): 135-144.
2. Gros L, Dei Tos AP, Jones RL, et al. Inflammatory myofibroblastic tumour: state of the art. Cancers (Basel), 2022, 14(15): 3662.
3. Zeng H, Li X, Chen S, et al. Anlotinib for the treatment of pulmonary epithelioid inflammatory myofibroblastic sarcoma: a case report. Anticancer Drugs, 2025, 36(8): 678-681.
4. Maharjan D, Dehner C, Alani A, et al. Epithelioid inflammatory myofibroblastic sarcoma: case series with a first report of CLTC: : ALK fusion in an aggressive disease. Genes Chromosomes Cancer, 2025, 64(5): e70055.
5. Cheng H, Lin Y, Zhu J, et al. Clinical features, treatment strategies, and prognosis of epithelioid inflammatory myofibroblastic sarcoma in children: a multicenter experience. Transl Pediatr, 2024, 13(2): 288-299.
6. Chopra S, Maloney N, Wang WL. Epithelioid inflammatory myofibroblastic sarcoma with VCL-ALK fusion of central nervous system: case report and brief review of the literature. Brain Tumor Pathol, 2022, 39(1): 35-42.
7. Singh P, Nambirajan A, Gaur MK, et al. Primary pulmonary epithelioid inflammatory myofibroblastic sarcoma: a rare entity and a literature review. J Pathol Transl Med, 2022, 56(4): 231-237.
8. Collins K, Ramalingam P, Euscher ED, et al. Uterine inflammatory myofibroblastic neoplasms with aggressive behavior, including an epithelioid inflammatory myofibroblastic sarcoma: a clinicopathologic study of 9 cases. Am J Surg Pathol, 2022, 46(1): 105-117.
9. Nayyar V, Bhutia O, Kakkar A, et al. Epithelioid inflammatory myofibroblastic sarcoma arising in the maxilla: first reported case in the jaws. Oral Surg Oral Med Oral Pathol Oral Radiol, 2023, 136(1): e15-e19.
10. Azad M, Oye M, Torrente N, et al. Pericardial epithelioid inflammatory myofibroblastic sarcoma: an atypical presentation. Cureus, 2022, 14(7): e26827.
11. 喬一丹, 陶菁, 何樂建. 兒童上皮樣炎性肌纖維母細胞肉瘤 7 例臨床病理和分子遺傳學特征. 臨床與實驗病理學雜志, 2024, 40(7): 710-713.
12. Okuda K, Watanabe T, Oda R, et al. Pulmonary inflammatory myofibroblastic tumor with TPM4-ALK translocation. J Thorac Dis, 2017, 9(11): E1013-E1017.
13. PDQ Pediatric Treatment Editorial Board. Childhood soft tissue sarcoma treatment (PDQ^?): health professional version//PDQ cancer information summaries. Bethesda (MD): National Cancer Institute (US), 2002.
14. Wachter F, Al-Ibraheemi A, Trissal MC, et al. Molecular characterization of inflammatory tumors facilitates initiation of effective therapy. Pediatrics, 2021, 148(6): e2021050990.
15. Gambacorti-Passerini C, Orlov S, Zhang L, et al. Long-term effects of crizotinib in ALK-positive tumors (excluding NSCLC): a phase 1b open-label study. Am J Hematol, 2018, 93(5): 607-614.
16. 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): 3215-3221.
17. 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): 957-967.
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): 1586.
19. Bai Y, Jiang M, Liang W, et al. Incomplete intestinal obstruction caused by a rare epithelioid inflammatory myofibroblastic sarcoma of the colon. Medicine, 2015, 94(51): e2342.
20. Aminimoghaddam S, Pourali R. Epithelioid inflammatory myofibroblastic sarcoma with poor response to crizotinib: a case report. Clin Med Insights Case Rep, 2023, 16: 11795476231163954.
21. 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.
22. 孫佳凡, 韓素萍, 王聰, 等. 靶向治療盆腹腔上皮樣炎性肌纖維母細胞肉瘤一例. 國際婦產科學雜志, 2024, 51(2): 236-240.
23. 肖法嫚, 李余發, 汪斌超. 阿來替尼治療罕見 EML4-ALK 融合肺上皮樣炎性肌纖維母細胞肉瘤 1 例. 中國腫瘤臨床, 2023, 50(13): 701-702.

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兒童罕見 ROS1 陽性上皮樣炎性肌纖維母細胞肉瘤診治一例

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