Skip to main content
SpringerLink
Log in

Sinonasal Inflammatory Myofibroblastic Tumor with Anaplastic Lymphoma Kinase 1 Rearrangement: Case Study and Literature Review

  • Original Paper
  • Published:
Head and Neck Pathology Aims and scope Submit manuscript

Abstract

Inflammatory myofibroblastic tumors (IMTs) are rare mesenchymal tumors initially described in the lung. About half of them exhibit expression of the ALK1 protein, generally resulting from a gene rearrangement. Paranasal sinus IMTs are extremely uncommon, and gene rearrangement of ALK1 is very rare in this localization. A 47-year-old woman presented with rapidly progressive vision loss in her left eye. Clinical and imaging work-up revealed a tumor invading the left ethmoidal and sphenoidal sinuses and extending into the nasal cavity, the orbit and the skull base. Complete tumor resection was performed using an endonasal approach. Pathological examination revealed a paranasal localization of IMT, positive for ALK1 immunostaining. FISH analysis showed an ALK1 gene rearrangement. This case illustrates the local aggressive potential for IMTs. Treatment is primarily surgical, but targeted therapies (crizotinib) might be a solution for ALK1 rearranged cases with a poor prognosis.

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

Fig. 1
Fig. 2
Fig. 3
Fig. 4
Fig. 5
Fig. 6

Similar content being viewed by others

References

  1. 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(6):569–76.

    Article  CAS  PubMed  Google Scholar 

  2. Castelnuovo P, Nicolai P, Turri-Zanoni M, Battaglia P, BolzoniVillaret A, Gallo S, Bignami M, Dallan I. Endoscopic endonasal nasopharyngectomy in selected cancers. Otolaryngol Head Neck Surg. 2013;149(3):424–30.

    Article  PubMed  Google Scholar 

  3. Pettinato G, Manivel J, De Rosa N, Dehner L. Inflammatory myofibroblastic tumor (plasma cell granuloma). Clinicopathologic study of 20 cases with immunohistochemical and ultrastructural observations. Am J Clin Pathol. 1990;95(5):538–46.

    Article  Google Scholar 

  4. Coffin C, Fletcher J. Inflammatory myofibroblastic tumor. In: Fletcher CDM, Unni KK, Mertens F, editors. Pathology and genetics of tumours of soft tissue and bone. Lyon, France: IARC Press; 2002. pp. 91–93.

    Google Scholar 

  5. Coffin C, Watterson J, Priest J, Dehner L. Extrapulmonary inflammatory myofibroblastic tumor (inflammatory pseudotumor). A clinico-pathologic and immunohistochemical study of 84 cases. Am J Surg Pathol. 1995;19(8):859–72.

    Article  CAS  PubMed  Google Scholar 

  6. He C, Dong G, Yang D, Liu H. Inflammatory myofibroblastic tumors of the nasal cavity and paranasal sinus: a clinicopathologic study of 25 cases and review of literature. Eur Arch Otorhinolaryngol. 2015;272(4):789–97.

    Article  PubMed  Google Scholar 

  7. Miller M, Fales W, McCracken W, O’Bryan M, Jarnagin J, Payeur J. Inflammatory pseudotumor in a cat with cutaneous mycobacteriosis. Vet Pathol. 1999;36(2):161–3.

    Article  CAS  PubMed  Google Scholar 

  8. Priebe-Richter C, Ivanyi P, Buer J, Langer F, Lotz J, Hertenstein B, Ganser A, Franzke A. Inflammatory pseudotumor of the lung following invasive aspergillosis in a patient with chronic graft-vs. -host disease. Eur J Haematol. 2005;75(1):68–72.

    Article  PubMed  Google Scholar 

  9. Constantino G, Sasaki F, Tavares R, Voegels R, Butugan O. Inflammatory pseudotumor of the paranasal sinuses. Rev Bras Otorhinolaryngol. 2008;74(2):297–302.

    Google Scholar 

  10. Gadde J, Franck B, Liu X, Teixido M, Rizk H. Inflammatory pseudotumor of the nasopharynx with spread along the trigeminal nerve. Am J Otolaryngol. 2013;34(3):252–4.

    Article  PubMed  Google Scholar 

  11. Huang W-H, Dai Y-C. Inflammatory pseudotumor of the nasal cavity. Am J Otolaryngol. 2006;27(4):275–7.

    Article  CAS  PubMed  Google Scholar 

  12. Lee H-M, Choi G, Choi CS, Kim CH, Lee SH. Inflammatory pseudotumor of the maxillary sinus. Otolaryngol–Head Neck Surg. 2001;125(5):565–6.

    CAS  PubMed  Google Scholar 

  13. Maire J, Eimer S, Galli FS, Franco-vidal V, Galland-girodet S, Huchet A, Darrouzet V. Case report inflammatory myofibroblastic tumour of the skull base. Case Rep Otolaryngol. 2013;2013:1–6.

  14. Fang S, Dong D, Jin M. Inflammatory myofibroblastic tumour of the maxillary sinus: CT appearance, clinical and pathological findings. Eur J Radiol Extra. 2006;60(1):5–9.

    Article  Google Scholar 

  15. Soysal V, Yigitbasi O, Kontas O, Kahya H, Guney E. Inflammatory myofibroblastic tumor of the nasal cavity: a case report and review of the literature. Int J Pediatr Otorhinolaryngol. 2001;61(2):161–5.

    Article  CAS  PubMed  Google Scholar 

  16. Dimitrakopoulos I, Psomaderis K, Iordanidis F, Karakasis D. Inflammatory myofibroblastic tumor of the maxillary sinus: a case report. J Oral Maxillofac Surg. 2007;65(2):323–6.

    Article  PubMed  Google Scholar 

  17. Karakok M, Enver O, Sari I, Mumbuc S, Aydin A, Kanlikama M, Kervancioglu R. 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. 2002;29(4):383–6.

    Article  PubMed  Google Scholar 

  18. Lawson SLA, Azoumah DK, Lawson-Evi K, N’Timon B, Savi de Tove HM, Yehouessi-Vignikin B, Kpemissi E. Tumeur myofibroblastique inflammatoire chez une fillette de 7 ans. Arch Pédiatr. 2010;17(1):34–7.

    Article  CAS  PubMed  Google Scholar 

  19. Lee DK, Cho Y-S, Hong SH, Chung WH, Ahn YC. Inflammatory pseudotumor involving the skull base: response to steroid and radiation therapy. Otolaryngol Head Neck Surg. 2006;135(1):144–8.

    Article  PubMed  Google Scholar 

  20. Sasagawa Y, Akai T, Itou S, Iizuka H. Multiple intraosseous inflammatory myofibroblastic tumors presenting with an aggressive clinical course: case report. Neurosurgery. 2011;69(4):1010–5.

    Article  Google Scholar 

  21. Saïji E, Guillou L. Tumeurs fibroblastiques et myofibroblastiques de la tête et du cou. Ann Pathol. 2009;29(4):335–46.

    Article  PubMed  Google Scholar 

  22. Morris SW, Kirstein MN, Valentine MB, Dittmer KG, Shapiro DN, Saltman DL, Look AT. Fusion of a Kinase Gene, ALK, to a nucleolar protein gene, NPM, in non-Hodgkin’s Lymphoma”. Science. 1994;263(5151):1281–4.

    Article  CAS  PubMed  Google Scholar 

  23. Griffin CA, Hawkins AL, Dvorak C, Henkle C, Ellingham T, Perlman EJ. Advances in brief recurrent involvement of 2p23 in inflammatory myofibroblastic tumors. Cancer Res. 1999;59(12):2776–80.

    CAS  PubMed  Google Scholar 

  24. Coffin CM, Hornick JL, Fletcher CD. Inflammatory myofibroblastic tumor: comparison of clinicopathologic, histologic, and immunohistochemical features including ALK expression in atypical and aggressive cases. Am J Surg Pathol. 2007;31(4):509–20.

    Article  PubMed  Google Scholar 

  25. Chan J, Cheuk W, Shimizu M. Anaplastic Lymphoma Kinase expression in inflammatory pseudotumors. Am J Surg Pathol. 2011;25(6):761–8.

    Article  Google Scholar 

  26. Jacob S, Reith J, Kojima A, Williams DW, Liu C, Vila Duckworth L. An unusual case of systemic inflammatory myofibroblastic tumor with successful treatment with ALK-Inhibitor. Case Rep Pathol. 2014;2014:1–5.

  27. Chiarle R, Voena C, Ambrogio C, Piva R, Inghirami G. The anaplastic lymphoma kinase in the pathogenesis of cancer. Nat Rev Cancer. 2008;8(1):11–23.

    Article  CAS  PubMed  Google Scholar 

  28. Butrynski JE, D’Adamo DR, Hornick JL, Dal Cin P, Antonescu C, Jhanwar S, Ladanyi M, Capelletti M, Rodig S, Ramaiya N, Kwak E, Clark J, Wilner KD, Christensen JG, Jänne P, Maki RG, Demetri GD, Shapiro GI. Crizotinib in ALK-rearranged inflammatory myofibroblastic tumor. New Engl J Med. 2010;363(18):1727–33.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

Download references

Author information

Authors and Affiliations

  1. ENT Department, Lariboisiere Hospital, 2 Rue Ambroise Paré, 75010, Paris, France

    Ghizlene Lahlou, Nicolas Le Clerc, Philippe Herman & Benjamin Verillaud

  2. Pathology Department, Lariboisiere Hospital, APHP, Paris 7 University, Paris, France

    Marion Classe & Michel Wassef

  3. Pathology Department, Cochin Hospital, APHP, Paris 5 University, Paris, France

    Pierre-Alexandre Just

  4. EA 7334 REMES, Paris 7 University, Paris, France

    Philippe Herman

Authors
  1. Ghizlene Lahlou
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Marion Classe
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Michel Wassef
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Pierre-Alexandre Just
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. Nicolas Le Clerc
    View author publications

    You can also search for this author in PubMed Google Scholar

  6. Philippe Herman
    View author publications

    You can also search for this author in PubMed Google Scholar

  7. Benjamin Verillaud
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Ghizlene Lahlou.

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Lahlou, G., Classe, M., Wassef, M. et al. Sinonasal Inflammatory Myofibroblastic Tumor with Anaplastic Lymphoma Kinase 1 Rearrangement: Case Study and Literature Review. Head and Neck Pathol 11, 131–138 (2017). https://doi.org/10.1007/s12105-016-0744-3

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s12105-016-0744-3

Keywords

Search

Navigation