Skip to main content

Advertisement

Log in

Analysis of clinical features and outcomes for inflammatory myofibroblastic tumors in China: 11 years of experience at a single center

  • Original Article
  • Published:
Pediatric Surgery International Aims and scope Submit manuscript

Abstract

Purpose

Inflammatory myofibroblastic tumor (IMT) is a rare benign neoplasm. The purpose of this study was to review the clinical characteristics, imaging and pathological features, and outcomes of children with IMTs from a single center in China.

Methods

A retrospective file review was conducted involving 23 cases of pathologically confirmed IMTs treated at the Children’s Hospital between April 2003 and April 2014.

Results

The tumor locations included multiple anatomic sites, as follows: abdomen or pelvis (n = 17); lungs (n = 2); head and neck (n = 1); trunk (n = 1); and extremities (n = 2). The tumors were associated with various clinical presentations. The predominant symptoms included an anemic appearance, fevers, and an asymptomatic mass. Computed tomography scanning showed solid, heterogeneous, well-demarcated masses; the appearance of enhancement was variable. MRI appeared hypointense on T1-weighted images and hypointense or hyperintense on T2-weighted images. Immunohistochemical staining revealed anaplastic lymphoma kinase was negative in 11 of 13 cases tested. One patient quit treatment for the unresectable mass after biopsy and died 2 years later, and another patient with incompletely resection is alive at 30 months following chemotherapy. The remaining 21 cases had complete resections; one patient died due to a recurrence, and the other 20 patients survived and were tumor free. The follow-up ranged from 7 to 141 months, with a mean of 56 months. The 3-year OS was 88 % (95 % CI, 57–97 %).

Conclusions

IMT is a benign neoplasm that rarely presents with malignant features. Complete resection is curative in most patients. ALK+ is variable for diagnosis. Close follow-up is necessary for patients who undergo surgical resection.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1
Fig. 2

Similar content being viewed by others

References

  1. Mehta B, Mascarenhas L, Zhou S, Wang L, Venkatramani R (2013) Inflammatory myofibroblastic tumors in childhood. Pediatr Hematol Oncol 30:640–645

    Article  PubMed  Google Scholar 

  2. Nagarajan S, Jayabose S, McBride W, Prasadh I, Tanjavur V, Marvin MR, Rodriguez-Davalos MI (2013) Inflammatory myofibroblastic tumor of the liver in children. J Pediatr Gastroenterol Nutr 57:277–280

  3. Cook JR, Dehner LP, Collins MH, Ma Z, Morris SW, Coffin CM, Hill DA (2001) Anaplastic lymphoma kinase (ALK) expression in the inflammatory myofibroblastic tumor: a comparative immunohistochemical study. Am J Surg Pathol 25:1364–1371

    Article  CAS  PubMed  Google Scholar 

  4. Diop B, Konate I, Ka S, Sall I, Fall D, Dieng M, Wone Y (2011) Mesenteric myofibroblastic tumor: NSAID therapy after incomplete resection. J Visc Surg 148:e311–e314

    Article  CAS  PubMed  Google Scholar 

  5. Kovach SJ, Fischer AC, Katzman PJ, Salloum RM, Ettinghausen SE, Madeb R, Koniaris LG (2006) Inflammatory myofibroblastic tumors. J Surg Oncol 94:385–391

    Article  PubMed  Google Scholar 

  6. Mattei P, Barnaby K (2008) Rapid regression of duodenal inflammatory myofibroblastic tumor after intravenous ketorolac: case report and review of the literature. J Pediatr Surg 43:1196–1199

    Article  PubMed  Google Scholar 

  7. Fletcher CDM, Unni KK, Mertens F (2002) World Health Organization classification of tumors. Pathology and genetics of tumors of soft tissue and bone. IARC Press, Lyon, pp 48–106

    Google Scholar 

  8. Tao YL, Wang ZJ, Han JG, Wei P (2012) Inflammatory myofibroblastic tumor successfully treated with chemotherapy and nonsteroidals: a case report. World J Gastroenterol 18:7100–7103

    Article  PubMed Central  PubMed  Google Scholar 

  9. Bertocchini A, Lo ZC, Callea F, Gennari F, Serra A, Monti L, de Ville DGJ (2011) Unresectable multifocal omental and peritoneal inflammatory myofibroblastic tumor in a child: revisiting the role of adjuvant therapy. J Pediatr Surg 46:e17–21

  10. Coffin CM, Hornick JL, Fletcher CD (2007) Inflammatory myofibroblastic tumor: comparison of clinicopathologic, histologic, and immunohistochemical features including ALK expression in atypical and aggressive cases. Am J Surg Pathol 31:509–520

    Article  PubMed  Google Scholar 

  11. Karnak I, Senocak ME, Ciftci AO, Caglar M, Bingol-Kologlu M, Tanyel FC, Buyukpamukcu N (2001) Inflammatory myofibroblastic tumor in children: diagnosis and treatment. J Pediatr Surg 36:908–912

    Article  CAS  PubMed  Google Scholar 

  12. Yang X, Miao R, Yang H, Chi T, Jiang C, Wan X, Xu Y, Xu H, Du S, Lu X, Mao Y, Zhong S, Zhao H, Sang X (2014) A retrospective and comparative study of inflammatory myofibroblastic tumor of the liver. J Gastroenterol Hepatol 30:885–890

  13. Cessna MH, Zhou H, Sanger WG, Perkins SL, Tripp S, Pickering D, Daines C, Coffin CM (2002) Expression of ALK1 and p80 in inflammatory myofibroblastic tumor and its mesenchymal mimics: a study of 135 cases. Mod Pathol 15:931–938

    Article  PubMed  Google Scholar 

  14. Dong A, Wang Y, Dong H, Gong J, Cheng C, Zuo C, Lu J (2014) Inflammatory myofibroblastic tumor: FDG PET/CT findings with pathologic correlation. Clin Nucl Med 39:113–121

    PubMed  Google Scholar 

  15. Fernandez DLPG, Garcia AJ, Lechuga VM, Blanco MM (1999) Hepatic inflammatory pseudotumor: apropos a case with a response to steroid treatment. Gastroenterol Hepatol 22:14–17

    Google Scholar 

  16. Coffin CM, Watterson J, Priest JR, Dehner LP (1995) Extrapulmonary inflammatory myofibroblastic tumor (inflammatory pseudotumor). A clinicopathologic and immunohistochemical study of 84 cases. Am J Surg Pathol 19:859–872

    Article  CAS  PubMed  Google Scholar 

  17. Janik JS, Janik JP, Lovell MA, Hendrickson RJ, Bensard DD, Greffe BS (2003) Recurrent inflammatory pseudotumors in children. J Pediatr Surg 38:1491–1495

    Article  PubMed  Google Scholar 

  18. Lazure T, Ferlicot S, Gauthier F, Doz F, Couturier J, Fabre M, Bedossa P (2002) Gastric inflammatory myofibroblastic tumors in children: an unpredictable course. J Pediatr Gastroenterol Nutr 34:319–322

    Article  PubMed  Google Scholar 

Download references

Acknowledgments

The authors are indebted to all participating patients and physicians for their participation in the study. The authors are also indebted to Dr. Lian Chen and Dr. Yangyang Ma who participated in our research and provided lots of suggestions to pathologic analysis.

This study received grants support from Shengkang Hospital Development Center (SHDC12012220 by Xiaolong Zhao, Department of Endocrinology, Huashan Hospital of Fudan University) and Shanghai Human Resources and Social Security (201342, by Kai LI, Department of Pediatric Surgery, Children’s Hospital of Fudan University).

Author information

Authors and Affiliations

Authors

Corresponding author

Correspondence to Kai Li.

Ethics declarations

Conflict of interest

The authors have no conflicts to disclose relative to this article.

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Wang, Z., Zhao, X., Li, K. et al. Analysis of clinical features and outcomes for inflammatory myofibroblastic tumors in China: 11 years of experience at a single center. Pediatr Surg Int 32, 239–243 (2016). https://doi.org/10.1007/s00383-015-3840-7

Download citation

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00383-015-3840-7

Keywords

Navigation