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CORRESPONDENCE
Year : 2019  |  Volume : 15  |  Issue : 3  |  Page : 725-728

Inflammatory myofibroblastic tumor of the oral cavity: A case report and literature review


1 Department of Oral Pathology and Microbiology, School of Dentistry, D. Y. Patil University, Mumbai, Maharashtra, India
2 Department of Oral Pathology and Microbiology, Nair Hospital Dental College, Mumbai, Maharashtra, India

Date of Web Publication29-May-2019

Correspondence Address:
Dr. Subraj J Shetty
Department of Oral Pathology and Microbiology, School of Dentistry, D. Y. Patil University, Sector 7, Nerul, Mumbai - 400 706, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jcrt.JCRT_1044_16

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 > Abstract 


Inflammatory myofibroblastic tumor (IMT) is a rare tumor of unknown etiology and pathogenesis. The lesion has been recognized to occur at various sites but rarely affects the head and neck region. A 29-year-old male presented with a 13 months' history of a slow growing, painless growth in maxillary left posterior gingiva. An excisional biopsy was performed under local anesthesia. Microscopic examination revealed a compact cellular spindle cell proliferation with collagenous stroma having storiform architecture. Immunohistochemistry revealed that the tumor cells were positive for smooth muscle actin, CD-68 and negative for anaplastic lymphoma kinase. Oral IMT should be included in the differential diagnosis of localized gingival enlargement mimicking oral hyperplastic/reactive lesions.

Keywords: Inflammatory myofibroblastic tumor, myofibroblasts, oral cavity


How to cite this article:
Shetty SJ, Pereira T, Desai RS. Inflammatory myofibroblastic tumor of the oral cavity: A case report and literature review. J Can Res Ther 2019;15:725-8

How to cite this URL:
Shetty SJ, Pereira T, Desai RS. Inflammatory myofibroblastic tumor of the oral cavity: A case report and literature review. J Can Res Ther [serial online] 2019 [cited 2019 Jun 17];15:725-8. Available from: http://www.cancerjournal.net/text.asp?2019/15/3/725/244198




 > Introduction Top


Inflammatory myofibroblastic tumor (IMT) is a rare mesenchymal tumor, primarily occurring in children and young adults.[1] The first case was first observed in the lung and described by Brunn in 1939 and was so named by Umiker et al. in 1954 because its clinical and radiological behavior mimics a malignant process.[2] The most common sites of involvement include the lung, liver, and orbit, but it has been reported to occur in nearly every site of the body, including the major salivary glands and the oral cavity.[3]

Until 1998, it was called inflammatory pseudotumor (IPT), when the term IMT was proposed as being a more descriptive name. The etiology and pathogenesis of IMT remains elusive.[3] Trauma and viral infections such as herpes simplex virus type 8 and Epstein–Barr virus, as well as autoimmune reactions, have been suggested as etiological factors.[4]

Here, we report and describe the clinicopathologic features of a case of IMT arising from the gingiva.


 > Case Report Top


A 29-year-old male presented with a 13 months' history of a slow growing, painless growth in maxillary left posterior gingiva. The lesion was a well-circumscribed, pedunculated nodule, the same color as the surrounding mucosa, in the area between teeth numbers 26 and 27 [Figure 1]. There was no bleeding or exudates in the area. The patients' medical and social history was noncontributory. Orthopantomogram showed the presence of slight bony erosion in between 26 and 27 [Figure 2]. The initial clinical differential diagnosis was fibroma, peripheral giant cell lesion, pyogenic granuloma, and peripheral ossifying fibroma.
Figure 1: Intraoral photograph showing a gingival growth in the maxillary left posterior region

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Figure 2: Orthopantomogram showing slight bony erosion between maxillary left first and second molar

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An excisional biopsy was performed under local anesthesia. Gross examination of the specimen revealed a brownish white colored mass, measuring approximately 3 cm × 2.5 cm in size. The cut surface of the lesion was yellowish-white in color and firm in consistency [Figure 3]. Microscopic examination of the soft-tissue specimen, using hematoxylin and eosin stain, revealed a compact cellular spindle cell proliferation with collagenous stroma having storiform architecture. The inflammatory component was minimal with single or small groups of lymphocytes scattered around the spindle cell proliferation [Figure 4]. Immunohistochemistry revealed that the tumor cells were positive for smooth muscle actin [Figure 5], CD-68 [Figure 6], and negative for anaplastic lymphoma kinase (ALK). The final diagnosis was IMT.
Figure 3: Cut surface of gross specimen

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Figure 4: Photomicrograph showing a proliferation of spindle cells admixed with numerous chronic inflammatory cells (H and E, ×40)

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Figure 5: Photomicrograph showing immunopositivity for smooth muscle actin (×10)

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Figure 6: Photomicrograph showing immunohistochemical expression of CD-68 showing positivity of histiocytes (×10)

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 > Discussion Top


Myofibroblasts are ubiquitous cells showing ultrastructural features of both muscle cells and fibroblasts.[5] IMT is a benign neoplasm consisting of variable numbers of inflammatory cells and myofibroblastic spindle cells.[3] Liston et al. in 1981 were the first to report IMTs of oral cavity in three children.[6] The numerous terms used to describe this lesion like pseudosarcomatous myofibroblastic proliferation, inflammatory sarcoma, plasma-cell granuloma, IPT, and inflammatory histiocytic proliferation reflect the initial controversies regarding its etiopathogenesis, histopathology, and nomenclature. Eventually, electron microscopic and immunohistochemical examinations clarified that the proliferating spindle cells in these tumors were myofibroblasts; and therefore, these distinctive tumors are today preferentially called IMTs.[5]

The histologic differential diagnosis of IMT is extensive and includes benign and malignant spindle cell tumors such as nodular fasciitis, solitary fibrous tumor, benign fibrous histiocytoma, calcifying fibrous tumor, myofibroma, fibrosarcoma, follicular dendritic cell tumor, and leiomyosarcoma.[3]

Histologically, IMTs contain a much more prominent inflammatory infiltrate than nodular fasciitis. In addition, they lack the “C-” shaped fascicles and mucin-rich stroma which is responsible for the characteristic “tissue culture-like or feathery” appearance in nodular fasciitis. Solitary fibrous tumor was excluded due to the lack of hemangiopericytoma-like areas and strong CD-34 immunoreactivity.[3]

The diagnosis of benign fibrous histiocytoma was not favored because of the lack of characteristic storiform pattern. Calcifying fibrous tumor, a rare benign neoplasm, is uniformly hypocellular and contains scattered dystrophic calcifications.[3]

The diagnosis of myofibroma was excluded due to the lack of biphasic growth pattern with hemangiopericytoma-like blood vessels. Fibrosarcoma was excluded due to the lack of malignant features, collagenous areas and herringbone pattern that characterize it. In addition, it typically lacks a significant inflammatory infiltrate.[3]

There is no evidence of recurrence, malignant transformation, metastasis, or death reported with oral IMTs.[3]

According to the World Health Organization, IMTs are classified as tumors of intermediate biological potential due to a tendency of local recurrence and small risk of distance metastasis.[3]

The ALK (chromosome band 2p23) gene has been implicated in the pathogenesis of IMT supporting neoplastic origin of tumor.[4] Approximately 50% of all IMTs have been associated with ALK positivity. Most of the previous oral IMT cases have been reported to be ALK negative.[4] Indeed, a question has been raised whether ALK-positive IMTs are a distant clinicopathologic entity with neoplastic origin with more aggressive behavior and even potential for malignancy.[4] The present case was ALK negative.

Including the present case, 28 cases of oral IMT have been reported [Table 1], with lesions occurring over a wide age range of 2–82 years and showing a 5:4 female predilection.[1],[2],[3],[4],[5],[6],[7],[8],[9],[10],[11],[12],[13],[14],[15],[16],[17],[18],[19],[20],[21],[22],[23],[24],[25] The lesion typically presents as a firm, indurated swelling and does not produce significant systemic symptoms, unlike its counterpart in visceral organs. Intrabony presentation of IMT is rare. Only three cases have been reported. Brooks et al. described a resorptive effect of soft tissue on the underlying alveolar bone of the edentulous mandible.[17] A computed tomography scan or magnetic resonance imaging was done in previous reports which characterized lesions with ill-defined infiltrative or erosive bony margins. There was no evidence of recurrence in any previous reports.
Table 1: Clinical features of inflammatory myofibroblastic tumor cases in the oral cavity

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In conclusion, IMT is a rare tumor of the gingiva, and diagnosis of the tumor or the differential diagnosis from other lesions of the gingival may be difficult. Even though being an extremely rare neoplasm, oral IMT should be included in the differential diagnosis of localized gingival enlargement mimicking oral hyperplastic/reactive lesions.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
 > References Top

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Yucel Ekici N, Bayindir T, Kizilay A, Aydin NE. Inflammatory myofibroblastic tumor: A rare tumor in the tongue. Case Rep Otolaryngol 2013;2013:787824.  Back to cited text no. 1
    
2.
Umiker WO, Iverson L. Postinflammatory tumors of the lung; report of four cases simulating xanthoma, fibroma or plasma cell tumor. J Thorac surg 1954;28:55-63.  Back to cited text no. 2
    
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Rautava J, Soukka T, Peltonen E, Nurmenniemi P, Kallajoki M, Syrjänen S, et al. Unusual case of inflammatory myofibroblastic tumor in maxilla. Case Rep Dent 2013;2013:876503.  Back to cited text no. 4
    
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Deshingkar SA, Tupkari JV, Barpande SR. Inflammatory myofibroblastic tumour of maxilla. J Oral Maxillofac Pathol 2007;11:76-9.  Back to cited text no. 5
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6.
Liston SL, Dehner LP, Jarvis CW, Pitzele C, Huseby TL. Inflammatory pseudotumors in the buccal tissues of children. Oral Surg Oral Med Oral Pathol 1981;51:287-91.  Back to cited text no. 6
    
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Earl PD, Lowry JC, Sloan P. Intraoral inflammatory pseudotumor. Oral Surg Oral Med Oral Pathol 1993;76:279-83.  Back to cited text no. 7
    
8.
Ramachandra S, Hollowood K, Bisceglia M, Fletcher CD. Inflammatory pseudotumour of soft tissues: A clinicopathological and immunohistochemical analysis of 18 cases. Histopathology 1995;27:313-23.  Back to cited text no. 8
    
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Shek AW, Wu PC, Samman N. Inflammatory pseudotumour of the mouth and maxilla. J Clin Pathol 1996;49:164-7.  Back to cited text no. 9
    
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Ide F, Shimoyama T, Horie N. Intravenous myofibroblastic pseudotumour of the buccal mucosa. Oral Oncol 1998;34:232-5.  Back to cited text no. 10
    
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Ide F, Shimoyama T, Horie N. Inflammatory pseudotumor in the mandibular retromolar region. J Oral Pathol Med 1998;27:508-10.  Back to cited text no. 11
    
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Cable BB, Leonard D, Fielding CG, Hommer DH. Pathology forum: Quiz case 1. Diagnosis: Inflammatory myofibroblastic tumor (IMT). Arch Otolaryngol Head Neck Surg 2000;126:900, 904-5.  Back to cited text no. 12
    
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Ide F, Shimoyama T, Horie N. Sclerosing inflammatory myofibroblastic tumour of the tongue: An immunohistochemical and ultrastructural study. Oral Oncol 2000;36:300-4.  Back to cited text no. 13
    
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Pankaj C, Uma C. How to manage oral inflammatory myofibroblastic tumor (inflammatory pseudotumor)? Oral Dis 2001;7:315-6.  Back to cited text no. 14
    
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Jordan RC, Regezi JA. Oral spindle cell neoplasms: A review of 307 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2003;95:717-24.  Back to cited text no. 15
    
16.
Fang JC, Dym H. Myofibroblastic tumor of the oral cavity. A rare clinical entity. N Y State Dent J 2004;70:28-30.  Back to cited text no. 16
    
17.
Brooks JK, Nikitakis NG, Frankel BF, Papadimitriou JC, Sauk JJ. Oral inflammatory myofibroblastic tumor demonstrating ALK, p53, MDM2, CDK4, pRb, and ki-67 immunoreactivity in an elderly patient. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2005;99:716-26.  Back to cited text no. 17
    
18.
Poh CF, Priddy RW, Dahlman DM. Intramandibular inflammatory myofibroblastic tumor – A true neoplasm or reactive lesion? Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2005;100:460-6.  Back to cited text no. 18
    
19.
Johann AC, Caldeira PC, Abdo EN, Sousa SO, Aguiar MC, Mesquita RA, et al. Inflammatory myofibroblastic tumor of the alveolar mucosa of the mandible. Minerva Stomatol 2008;57:59-63.  Back to cited text no. 19
    
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Oh JH, Yim JH, Yoon BW, Choi BJ, Lee DW, Kwon YD, et al. Inflammatory pseudotumor in the mandible. J Craniofac Surg 2008;19:1552-3.  Back to cited text no. 20
    
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Eley KA, Watt-Smith SR. Intraoral presentation of inflammatory myofibroblastic tumor (pseudotumor) at the site of dental extraction: Report of a case and review of the literature. J Oral Maxillofac Surg 2010;68:2016-20.  Back to cited text no. 22
    
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Satomi T, Watanabe M, Matsubayashi J, Nagao T, Chiba H. A successfully treated inflammatory myofibroblastic tumor of the mandible with long-term follow-up and review of the literature. Med Mol Morphol 2010;43:185-91.  Back to cited text no. 23
    
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Palaskar S, Koshti S, Maralingannavar M, Bartake A. Inflammatory myofibroblastic tumor. Contemp Clin Dent 2011;2:274-7.  Back to cited text no. 24
[PUBMED]  [Full text]  
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Stringer DE, Allen CN, Nguyen K, Tandon R. Intraosseous inflammatory myofibroblastic tumor in the mandible: A rare pathologic case report. Case Rep Surg 2014;2014:565478.  Back to cited text no. 25
    


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