Tuberc Respir Dis.  2013 Oct;75(4):165-169.

A Case of Recurrent Pulmonary Inflammatory Myofibroblastic Tumor with Aggressive Metastasis after Complete Resection

Affiliations
  • 1Department of Internal Medicine, Korea Cancer Center Hospital, Korea Institute of Radiological and Medical Sciences, Seoul, Korea. cheol@kcch.re.kr

Abstract

An inflammatory myofibroblastic tumor (IMT) is a rare disease entity reported to arise in various organs. It is thought to be a neoplastic or reactive inflammatory condition, controversially. The treatment of choice for myofibroblastic tumor is surgery, and recurrence is known to be rare. The optimal treatment method is not well-known for patients ineligible for surgery. We report a 47-year-old patient with aggressive recurrent IMT of the lungs. The patient had been admitted for an evaluation of back-pain two years after a complete resection of pulmonary IMT. Radiation therapy was performed for multiple bone recurrences, and the symptoms were improved. However the patient presented again with aggravated back-pain six months later. High-dose steroid and non-steroidal anti-inflammatory drugs were administered, but the disease progressed aggressively, resulting in spinal cord compression and metastasis to intra-abdominal organs. This is a very rare case of aggressively recurrent pulmonary IMT with multi-organ metastasis.

Keyword

Lung Neoplasms; Myofibroblasts; Lung; Recurrence; Neoplasm Metastasis

MeSH Terms

Humans
Lung
Lung Neoplasms
Middle Aged
Myofibroblasts*
Neoplasm Metastasis*
Rare Diseases
Recurrence
Spinal Cord Compression

Figure

  • Figure. 1 Simple chest radiography (A) and contrast-enhanced computed tomography (CT) (B) taken before the operation. Chest CT shows a 3-cm sized, well-marginated oval mass in the left upper lobe of lung.

  • Figure. 2 Pathologic findings of resected specimens from the patient. (A) Grossly, there is a solitary mass with a yellowish-gray color and rubbery consistency. (B) Tumor contains a mixture of spindle cells and infiltrating inflammatory cells containing lymphocytes and plasma cells (H&E stain, ×500). (C) Immunohistochemical staining result for anaplastic lymphoma kinase-1 shows focal positivity in cytoplasm of tumor cells (×1,000).

  • Figure. 3 (A) Fluorodeoxyglucose positron emission tomography/computed tomography (PET/CT) images obtained before the first radiation therapy. PET/CT shows abnormal metabolic activity with a high standardized uptake value in the right 3rd rib, the thoracic 12th vertebra and the left sacroiliac bone. (B) PET/CT performed before the second radiation therapy. PET/CT shows remarkably increased number of metastatic lesions with increased fluorodeoxyglucose uptake at multiple bones during the time interval.


Reference

1. Cerfolio RJ, Allen MS, Nascimento AG, Deschamps C, Trastek VF, Miller DL, et al. Inflammatory pseudotumors of the lung. Ann Thorac Surg. 1999; 67:933–936.
2. Hagenstad CT, Kilpatrick SE, Pettenati MJ, Savage PD. Inflammatory myofibroblastic tumor with bone marrow involvement: a case report and review of the literature. Arch Pathol Lab Med. 2003; 127:865–867.
3. Kim DY, Park HS, Kim SM, Park JH, Hong YS, Lee JL, et al. Inflammatory myofibroblastic tumor showing durable remission after anthracycline-containing cytotoxic chemotherapy: report of a case. Korean J Med. 2012; 82:749–753.
4. Sasagawa Y, Akai T, Itou S, Iizuka H. Multiple intraosseous inflammatory myofibroblastic tumors presenting with an aggressive clinical course: case report. Neurosurgery. 2011; 69:E1010–E1015.
5. Brunn H. Two interesting benign lung tumors of contradictory histopathology. J Thorac Surg. 1939; 9:119–131.
6. Coffin CM, Fletcher JA. Fletcher CD, Unni KK, Mertens F, editors. World Health Organization classification of tumours: pathology and genetics of tumours of soft tissue and bone. Lyon: IARC Press;2002. p. 91–93.
7. Coffin CM, Patel A, Perkins S, Elenitoba-Johnson KS, Perlman E, Griffin CA. ALK1 and p80 expression and chromosomal rearrangements involving 2p23 in inflammatory myofibroblastic tumor. Mod Pathol. 2001; 14:569–576.
8. Suetsugu S, Yamamoto H, Izumi M, Takayama K, Inoue H, Nakanishi Y. A case of rapidly growing inflammatory myofibroblastic tumor in the lung. Nihon Kokyuki Gakkai Zasshi. 2009; 47:1156–1160.
9. Kubo N, Harada T, Anai S, Otsubo K, Yoneshima Y, Ijichi K, et al. Carboplatin plus paclitaxel in the successful treatment of advanced inflammatory myofibroblastic tumor. Intern Med. 2012; 51:2399–2401.
10. Applebaum H, Kieran MW, Cripe TP, Coffin CM, Collins MH, Kaipainen A, et al. The rationale for nonsteroidal antiinflammatory drug therapy for inflammatory myofibroblastic tumors: a Children's Oncology Group study. J Pediatr Surg. 2005; 40:999–1003.
Full Text Links
  • TRD
Actions
Cited
CITED
export Copy
Close
Share
  • Twitter
  • Facebook
Similar articles
Copyright © 2024 by Korean Association of Medical Journal Editors. All rights reserved.     E-mail: koreamed@kamje.or.kr