Skip to main content

Advertisement

SpringerLink
Log in

A successfully treated inflammatory myofibroblastic tumor of the mandible with long-term follow-up and review of the literature

  • Original Paper
  • Published:
Medical Molecular Morphology Aims and scope Submit manuscript

Abstract

Inflammatory myofibroblastic tumor (IMT) of the oral cavity is an extremely rare clinical and pathological disease entity. It was originally described in the lung but has recently been reported in various anatomic sites. We report such a case of inflammatory myofibroblastic tumor of the mandible in a 14-year-old girl. The patient presented with an aggressive ulcerative soft tissue mass of 3 months duration in the mandibular molar gingiva. Histologically, the lesion was composed of fibroblastic or myofibroblastic spindle cell proliferations with infiltrative margins in an inflammatory background. Immunohistochemically, the fibroblastic or myofibroblastic spindle cells were positive for vimentin, α-smooth muscle actin, and Ki-67 (MIB-1) but negative for desmin, pan-cytokeratin, S-100 protein, CD34, CD68, CD99, bcl-2, β-catenin, estrogen receptor, progesterone receptor, ALK-1, and p53. These spindle cells were focally and weakly Ki-67- (MIB-1-) positive. The MIB-1 labeling index was 5%. The results of in situ hybridization for Epstein-Barr virus-encoded-RNA were negative. The ratio of IgG4+/IgG+ plasma cells was about 10%. A pathological diagnosis of inflammatory myofibroblastic tumor was made. The postoperative course was uneventful, and the patient has had no recurrence in the 10-year follow-up period. Although no evidence of oral inflammatory myofibroblastic tumor recurrence or malignant transformation has been reported, it has been observed that in inflammatory myofibroblastic tumors of other regions, a prolonged follow-up is necessary after surgical resection. No other case of an IMT patient under 20 years of age has appeared in either the English or the Japanese literature.

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

Access this article

Log in via an institution

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

Instant access to the full article PDF.

Institutional subscriptions

Similar content being viewed by others

References

  1. Coffin CM, Fletcher JA (2002) Inflammatory myofibroblastic tumour. In: Fletcher CDM, Unni KK, Mertens F (eds) World Health Organization classification of tumours. Pathology and genetics of tumours of soft tissue and bone. IARC Press, Lyon, pp 91–93

    Google Scholar 

  2. Coindre JM (1994) Histologic classification of soft tissue tumors (WHO, 1994). Ann Pathol 14:426–427

    CAS  PubMed  Google Scholar 

  3. Coffin CM, Humphrey PA, Dehner LP (1998) Extrapulmonary inflammatory myofibroblastic tumor: a clinical and pathological survey. Semin Diagn Pathol 15:85–101

    CAS  PubMed  Google Scholar 

  4. 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 

  5. Nikitakis NG, Drachenberg CB, Papadimitriou JC (2002) MDM2 and CDK4 expression in carcinosarcoma of the esophagus: comparison with squamous cell carcinoma and review of the literature. Exp Mol Pathol 73:198–208

    Article  CAS  PubMed  Google Scholar 

  6. Bahadori M, Liebow AA (1973) Plasma cell granulomas of the lung. Cancer (Phila) 31:191–208

    Article  CAS  Google Scholar 

  7. Miettinen M (2003) Fibroblastic proliferations in children. In: Miettinen M (ed): Diagnostic soft tissue pathology. Churchill Livingstone, Philadelphia, pp 181–184

    Google Scholar 

  8. Brooks JK, Nikitakis NG, Frankel BF, Papadimitriou JC, Sauk JJ (2005) Oral inflammatory myofibroblastic tumor demonstrating ALK, pRb, and Ki-67 immunoreactivity in an elderly patient. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 99:716–726

    Article  PubMed  Google Scholar 

  9. Kuroda N, Tada H, Takahashi J, Ohara M, Hirouchi T, Mizuno K, Miyazaki E, Enzan H (2006) Myofibroblasts in the stroma of metastatic pulmonary calcification in a patient with chronic renal failure. Med Mol Morphol 39:161–163

    Article  PubMed  Google Scholar 

  10. Kuroda N, Tamura M, Ohara M, Hirouchi T, Mizuno K, Miyazaki E, Hayashi Y, Lee GH (2008) Possible identification of third stromal component in extraadrenal paraganglioma: myofibroblast in fibrous band and capsule. Med Mol Morphol 41:59–61

    Article  PubMed  Google Scholar 

  11. Karakök M, Ozer E, Sari I, Mumbuç S, Aydin A, Kanlikama M, Kervancioglu R (2002) Inflammatory myofibroblastic tumor (inflammatory pseudotumor) of the maxillary sinus mimicking malignancy: a case report of an unusual location (is that a true neoplasm?). Auris Nasus Larynx 29:383

    Article  PubMed  Google Scholar 

  12. Montgomery E, Goldblum JR, Fisher CF (2001) Myofibrosarcoma. A clinicopathological study. Am J Surg Pathol 25:219

    Article  CAS  PubMed  Google Scholar 

  13. Watanabe K, Tajino T, Sekiguchi M, Suzuki T (2000) Inflammatory myofibroblastic tumor (inflammatory fibrosarcoma) of the bone. Arch Pathol Lab Med 124:1514

    CAS  PubMed  Google Scholar 

  14. Chen JK, Cheuk W, Shimizu M (2001) Anaplastic lymphoma kinase expression in inflammatory pseudotumors. Am J Surg Pathol 25:761–768

    Article  Google Scholar 

  15. 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 

  16. Griffin CA, Hawkins AL, Dvorak C, Henkle C, Ellingham T, Perlman EJ (1999) Recurrent involvement of 2p23 in inflammatory myofibroblastic tumors. Cancer Res 59:2776–2780

    CAS  PubMed  Google Scholar 

  17. Lawrence B, Perez-Atayde A, Hibbard MK, Rubin BP, Dal Cin P, Pinkus JL, Pinkus GS, Xiao S, Yi ES, Fletcher CDM, Fletcher JA (2000) TPM3-ALK and TPM4-ALK oncogenes in inflammatory myofibroblastic tumors. Am J Pathol 157:377–384

    CAS  PubMed  Google Scholar 

  18. Coffin CM, Patel A, Perkins S, Elenitoba-Johnson KSJ, Perlman E, Griffin CA (2001) ALK1 and p80 expression and chromosomal rearrangements involving 2p23 in inflammatory myofibroblastic tumor. Mod Pathol 14:569–576

    Article  CAS  PubMed  Google Scholar 

  19. Kovarik P, Pyle J, Chou PM (1998) Ploidy, proliferative activity, and p53 as biologic markers in inflammatory myofibroblastic tumors [abstract]. Lab Invest 78:11A

    Google Scholar 

  20. Meis-Kindblom JM, Kjellström C, Kindblom LG (1998) Inflammatory fibrosarcoma: update, reappraisal, and perspective on its place in the spectrum of inflammatory myofibroblastic tumors. Semin Diagn Pathol 15:133–143

    CAS  PubMed  Google Scholar 

  21. Hussong JW, Brown M, Perkins SL, Dehner LP, Coffin CM (1999) Comparison of DNA ploidy, histologic, and immunohistochemical findings with clinical outcome in inflammatory myofibroblastic tumors. Mod Pathol 12:279–286

    CAS  PubMed  Google Scholar 

  22. Rassidakis GZ, Sarris AH, Herling M, Ford RJ, Cabanillas F, McDonnell TJ, Medeiros LJ (2001) Differential expression of BCL-2 family proteins in ALK-positive and ALK-negative anaplastic large cell lymphoma of T/null-cell lineage. Am J Pathol 159:527–535

    CAS  PubMed  Google Scholar 

  23. Villalva C, Bougrine F, Delsol G, Brousset P (2001) Bcl-2 expression in anaplastic large cell lymphoma [letter]. Am J Pathol 158:1889–1890

    CAS  PubMed  Google Scholar 

  24. Arber DA, Kamel OW, van de Rijn M, Davis RE, Medeiros LJ, Jaffe ES, Weiss LM (1995) Frequent presence of the Epstein-Barr virus in inflammatory pseudotumor. Hum Pathol 26:1093–1098

    Article  CAS  PubMed  Google Scholar 

  25. Arber DA, Weiss LM, Chang KL (1998) Detection of Epstein-Barr virus in an inflammatory pseudotumor. Semin Diagn Pathol 15:155–160

    CAS  PubMed  Google Scholar 

  26. Lewis JT, Gaffney RL, Casey MB, Farrell MA, Morice WG, Macon WR (2003) Inflammatory pseudotumor of the spleen associated with a clonal Epstein-Barr virus genome. Am J Clin Pathol 120:56–61

    Article  PubMed  Google Scholar 

  27. Cheuk W, Woo PC, Yuen KY, Yu PH, Chan JK (2000) Intestinal inflammatory pseudotumour with regional lymph node involvement: identification of a new bacterium as the aetiological agent. J Pathol 192:289–292

    Article  CAS  PubMed  Google Scholar 

  28. Yamamoto H, Yamaguchi H, Aishima S, Oda Y, Kohashi K, Oshiro Y, Tsuneyoshi M (2009) Inflammatory myofibroblastic tumor versus IgG4-related sclerosing disease and inflammatory pseudotumor. Am J Surg Pathol 33:1330–1340

    Article  PubMed  Google Scholar 

  29. Gale N, Zidar N, Podboj J, Volavsek M, Luzar B (2003) Inflammatory myofibroblastic tumor of paranasal sinuses with fatal outcome: reactive lesion or tumor? J Clin Pathol 56:715–717

    Article  CAS  PubMed  Google Scholar 

  30. Kempson RL, Fletcher CD, Evans HL, Hendrickson MR, Sibley RK (2001) Tumors of the soft tissues. Atlas of tumor pathology, third series. Armed Forces Institute of Pathology, Washington, DC, pp 83–86

    Google Scholar 

  31. 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 

  32. Rudy HN, Conn H, Scheingold SS (1964) Solitary xanthogranuloma of the mandible. Oral Surg Oral Med Oral Pathol 18:262–271

    Article  CAS  PubMed  Google Scholar 

  33. Zegarelli DJ, Rankov RM, Zegarelli EV (1974) A large dental granuloma (? inflammatory pseudotumor) with unusual features: report of a case. J Am Dent Assoc 89:891–894

    CAS  PubMed  Google Scholar 

  34. Earl PD, Lowry JC, Sloan P (1993) Intraoral inflammatory pseudotumor. Oral Surg Oral Med Oral Pathol 76:279–283

    Article  CAS  PubMed  Google Scholar 

  35. Inui M, Tagawa T, Mori A, Yoneda J, Nomura J, Fukumori T (1993) Inflammatory pseudotumor in the submandibular region. Clinicopathologic study and review of literature. Oral Surg Oral Med Oral Pathol 76:333–337

    Article  CAS  PubMed  Google Scholar 

  36. Ide F, Shimoyama T, Horie N (1998) Inflammatory pseudotumor in the mandibular retromolar region. J Oral Pathol Med 27:508–510

    Article  CAS  PubMed  Google Scholar 

  37. Araki M, Hashimoto K, Shinoda K, Komiyama K (2002) Plasma cell granuloma with severe cortical bone destruction of the mandible appearing as a malignant lesion: a case report. Oral Radiol 18:45–51

    Article  Google Scholar 

  38. Fang JC, Dym H (2004) Myofibroblastic tumor of the oral cavity. A rare clinical entity. NY State Dent J 70:28–30

    Google Scholar 

  39. Poh CF, Priddy RW, Dahlman DM (2005) Intramandibular inflammatory myofibroblastic tumor: a true neoplasm or reactive lesion? Oral Surg Oral Med Oral Pathol Oral Radiol Endod 100:460–466

    Article  PubMed  Google Scholar 

  40. Aota K, Yui S, Adachi M, Shimizu H, Kinoshita F, Yura Y (2005) A case of inflammatory myofibroblastic tumor of the mandible. Jpn J Oral Maxillofac Surg 51:89–92

    Google Scholar 

  41. Oh JH, Yim JH, Yoon BW, Choi BJ, Lee DW, Kwon YD (2008) Inflammatory pseudotumor in the mandible. J Craniofac Surg 19:1552–1553

    Article  PubMed  Google Scholar 

  42. Johann AC, Caldeira PC, Abdo EN, Sousa SO, Aguiar MC, Mesquita RA (2008) Inflammatory myofibroblastic tumor of the alveolar mucosa of the mandible. Minerva Stomatol 57:59–63

    CAS  PubMed  Google Scholar 

Download references

Author information

Authors and Affiliations

  1. Department of Oral and Maxillofacial Surgery, Tokyo Medical University Hospital, 6-7-1 Nishi-Shinjuku, Shinjuku-ku, Tokyo, 160-0023, Japan

    Takafumi Satomi, Masato Watanabe & Hiroshige Chiba

  2. Department of Diagnostic Pathology, Tokyo Medical University, Tokyo, Japan

    Jun Matsubayashi & Toshitaka Nagao

Authors
  1. Takafumi Satomi
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Masato Watanabe
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Jun Matsubayashi
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Toshitaka Nagao
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. Hiroshige Chiba
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Takafumi Satomi.

Rights and permissions

Reprints and permissions

About this article

Cite this article

Satomi, T., Watanabe, M., Matsubayashi, J. et al. A successfully treated inflammatory myofibroblastic tumor of the mandible with long-term follow-up and review of the literature. Med Mol Morphol 43, 185–191 (2010). https://doi.org/10.1007/s00795-009-0483-6

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00795-009-0483-6

Key words

Search

Navigation