The Clinicopathologic Features of Uterine Inflammatory Myofibroblastic Tumor: A Case Report

Overview

Journal Medicine (Baltimore)

Date 2025 Jan 20

PMID 39833069

Authors

Xiaoming Pan

Jian Zou

Shizhou Yang

Yuanhe Wang

Jie Luo

Xiaojun Zhu

Affiliations

Soon will be listed here.

Abstract

Rationale: Inflammatory myofibroblastic tumor (IMT) is a rare soft tissue neoplasm with low malignant potential. These patients present with a certain probability of malignant potential. The management of IMT has not been standardized, especially for the patients with fertility needs.

Patient Concerns And Diagnoses: Thirteen patients with IMT who attended in the Department of Gynecology, Women's Hospital, Zhejiang University School of Medicine were enrolled between 2019 and 2023. The data of the clinical and pathological features was analyzed.

Interventions And Outcomes: The mean age of the patients was 45.31 ± 12.80 years. Seven of the 13 cases (53.85%) had abnormal uterine bleeding. Five of them (38.46%) had a rapidly growing mass, and 2 of them (15.38%) had no obvious symptoms. With regard to surgical strategies, 7 patients underwent hysterectomy, and 6 patients underwent mass resection only. All the patients were alive with no evidence of disease at an average of 9.58 months of follow-up. One of them gave birth to a full-term male infant at 40 weeks of gestation after hysteroscopic resection, without recurrence.

Lessons: Uterine inflammatory myofibroblastic tumor can be diagnosed by the anaplastic lymphoma kinase overexpression. Complete excision under hysteroscopy or laparoscopy seems to be effective and safe. Because there is a certain risk of recurrence and metastasis, conservative surgery should be performed carefully to preserve fertility in patients who can undergo close follow-up.

References

Brivio E, Zwaan C . 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. View

Zhang Y, Dong W, Li S, Zhao J, DU B . [Clinical and molecular pathological features of uterine inflammatory myofibroblastic tumor]. Zhonghua Bing Li Xue Za Zhi. 2021; 50(9):1020-1023. DOI: 10.3760/cma.j.cn112151-20210110-00024. View

Sanaya S, Matsneva I, Redkina N, Telezhnikova I, Magnaeva A, Tregubova A . [Immunohistochemical and molecular diagnosis of inflammatory myofibroblastic tumor of the uterus: a literature review and a clinical case]. Arkh Patol. 2021; 83(5):43-48. DOI: 10.17116/patol20218305143. View

Collins K, Ramalingam P, Euscher E, Reques Llanos A, Garcia A, Malpica A . Uterine Inflammatory Myofibroblastic Neoplasms With Aggressive Behavior, Including an Epithelioid Inflammatory Myofibroblastic Sarcoma: A Clinicopathologic Study of 9 Cases. Am J Surg Pathol. 2021; 46(1):105-117. DOI: 10.1097/PAS.0000000000001756. View

Stikova Z, Ptakova N, Horakova M, Kostun J, Ondic O . Inflammatory myofibroblastic tumor of the uterus - case report. Cesk Patol. 2019; 55(4):239-243. View

Bowman C, Medeiros F, Fadare O, Sangoi A, Horvai A, Devine W . ALK Immunoexpression is Specific for Inflammatory Myofibroblastic Tumor Among Vulvovaginal Mesenchymal Neoplasms. Int J Gynecol Pathol. 2022; 42(1):1-10. DOI: 10.1097/PGP.0000000000000858. View

Gilks C, Taylor G, Clement P . Inflammatory pseudotumor of the uterus. Int J Gynecol Pathol. 1987; 6(3):275-86. DOI: 10.1097/00004347-198709000-00008. View

Bennett J, Wang P, Wanjari P, Diaz L, Oliva E . Uterine inflammatory myofibroblastic tumor: First report of a ROS1 fusion. Genes Chromosomes Cancer. 2021; 60(12):822-826. DOI: 10.1002/gcc.22986. View

Lopez-Nunez O, John I, Panasiti R, Ranganathan S, Santoro L, Grelaud D . Infantile inflammatory myofibroblastic tumors: clinicopathological and molecular characterization of 12 cases. Mod Pathol. 2019; 33(4):576-590. DOI: 10.1038/s41379-019-0406-6. View

10.

Takahashi A, Kurosawa M, Uemura M, Kitazawa J, Hayashi Y . Anaplastic lymphoma kinase-negative uterine inflammatory myofibroblastic tumor containing the ETV6-NTRK3 fusion gene: a case report. J Int Med Res. 2018; 46(8):3498-3503. PMC: 6134678. DOI: 10.1177/0300060518780873. View

11.

Preobrazhenskaya E, Iyevleva A, Suleymanova A, Tiurin V, Mitiushkina N, Bizin I . Gene rearrangements in consecutive series of pediatric inflammatory myofibroblastic tumors. Pediatr Blood Cancer. 2020; 67(5):e28220. DOI: 10.1002/pbc.28220. View

12.

Zarei S, Abdul-Karim F, Chase D, Astbury C, Policarpio-Nicolas M . Uterine Inflammatory Myofibroblastic Tumor Showing an Atypical ALK Signal Pattern by FISH and DES-ALK Fusion by RNA Sequencing: A Case Report. Int J Gynecol Pathol. 2019; 39(2):152-156. DOI: 10.1097/PGP.0000000000000588. View

13.

Liu X, Duan Y, Wang G, Zhu P . A novel TPD52L2-ROS1 gene fusion expanding the molecular alterations in inflammatory myofibroblastic tumor: case report and literature review. Diagn Pathol. 2023; 18(1):105. PMC: 10512592. DOI: 10.1186/s13000-023-01382-0. View

14.

Mohammad N, Haimes J, Mishkin S, Kudlow B, Leong M, Chew S . ALK Is a Specific Diagnostic Marker for Inflammatory Myofibroblastic Tumor of the Uterus. Am J Surg Pathol. 2018; 42(10):1353-1359. DOI: 10.1097/PAS.0000000000001120. View

15.

Kuisma H, Jokinen V, Pasanen A, Heikinheimo O, Karhu A, Valimaki N . Histopathologic and Molecular Characterization of Uterine Leiomyoma-like Inflammatory Myofibroblastic Tumor: Comparison to Molecular Subtypes of Uterine Leiomyoma. Am J Surg Pathol. 2022; 46(8):1126-1136. DOI: 10.1097/PAS.0000000000001904. View

16.

Shukla P, Mittal K . Inflammatory Myofibroblastic Tumor in Female Genital Tract. Arch Pathol Lab Med. 2018; 143(1):122-129. DOI: 10.5858/arpa.2017-0575-RA. View

17.

von Mehren M, Kane J, Agulnik M, Bui M, Carr-Ascher J, Choy E . Soft Tissue Sarcoma, Version 2.2022, NCCN Clinical Practice Guidelines in Oncology. J Natl Compr Canc Netw. 2022; 20(7):815-833. PMC: 10186762. DOI: 10.6004/jnccn.2022.0035. View

18.

Carballo E, Pham T, Turashvili G, Hanley K, Starbuck K, Meisel J . Recurrent uterine inflammatory myofibroblastic tumor previously managed as leiomyosarcoma has sustained response to alectinib. Gynecol Oncol Rep. 2022; 43:101062. PMC: 9403501. DOI: 10.1016/j.gore.2022.101062. View

19.

Preobrazhenskaya E, Suleymanova A, Bizin I, Zagrebin F, Romanko A, Saitova E . Spectrum of kinase gene rearrangements in a large series of paediatric inflammatory myofibroblastic tumours. Histopathology. 2023; 83(1):109-115. DOI: 10.1111/his.14912. View

20.

Regaieg C, Triki M, Cheikrouhou T, Thabet A, Charfi S, Dhaou M . ALK-Negative Inflammatory Myofibroblastic Tumor: A Challenging Case in a Premature Newborn. J Neonatal Perinatal Med. 2023; 16(2):355-359. DOI: 10.3233/NPM-230007. View