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Table of Contents   
CASE REPORT  
Year : 2013  |  Volume : 24  |  Issue : 5  |  Page : 635-638
Benign fibrous histiocytoma of the tongue


Department of Oral Pathology, V S Dental College and Hospital, Bangalore, Karnataka, India

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Date of Submission15-Feb-2013
Date of Decision25-Feb-2013
Date of Acceptance23-Mar-2013
Date of Web Publication21-Dec-2013
 

   Abstract 

Benign fibrous histiocytoma (BFH) is a benign fibrohistiocytic neoplasm. It is documented to occur in all anatomic sites with a strong predilection for sun exposed skin surfaces. Intra oral occurrence of BFH comprises a lesser percentage of cases with tongue being the least reported site. We report a case of BFH of the tongue with an emphasis on its histogenesis and a review of the literature.

Keywords: Fibrous histiocytoma, benign fibrous histiocytoma, tongue

How to cite this article:
Priya N S, Rao K, Umadevi H S, Smitha T. Benign fibrous histiocytoma of the tongue. Indian J Dent Res 2013;24:635-8

How to cite this URL:
Priya N S, Rao K, Umadevi H S, Smitha T. Benign fibrous histiocytoma of the tongue. Indian J Dent Res [serial online] 2013 [cited 2020 Oct 22];24:635-8. Available from: https://www.ijdr.in/text.asp?2013/24/5/635/123419
Tongue being a specialized muscular and hydrostat appendage of the oral cavity, harbors varied pathological entities from hamartomas to neoplasms. One such rare group of neoplasms having a definitive growth potential, but a limited capacity for aggressive behavior occurring in tongue is fibrohistiocytic tumors. This group of neoplasms exhibit benign, intermediate and malignant grades. [1] Until date, very few cases of benign fibrous histiocytoma (BFH) of the tongue are documented in the literature.


   Case Report Top


A 30-year-old female patient presented with a nodular mass of 7 months duration on the anterior dorsum of the tongue, which gradually increased to the present size. Patient complained of difficulty in speech and she could not elicit a definite history of trauma to the region.

On intra-oral examination, a sessile smooth surfaced nodule measuring 3 cm × 3 cm was present on the anterior dorsum of the tongue. It was pink in color with no surface ulceration and was firm in consistency on palpation.

Fibroma was considered for clinical diagnosis and the mass was excised under local anesthesia.

Gross specimen was well-circumscribed and round in shape [Figure 1]. Cut section showed homogeneous white areas.
Figure 1: Gross specimen, well circumscribed nodular mass

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Microscopic examination revealed well encapsulated lesion showing uniform hypercellular areas comprising of spindle cells arranged in short fascicles [Figure 2]. Focal areas exhibited storiform pattern [Figure 3]. Histiocyte like cells with vesiculated nuclei were seen scattered throughout the lesion. Cellular atypia was not evident and mitotic figures were sparse. Lesional cells were seen dissecting the underlying muscle bundles [Figure 4]. Numerous vascular spaces with hemangiopericytoma like areas were evident [Figure 5].
Figure 2: Hypercellular areas with tumor cells arranged in short fascicles. (H and E: ×10)

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Figure 3: Cells arranged in storiform pattern. (H and E, ×20)

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Figure 4: Infiltration of tumor cells dissecting the muscle bundles. (H and E, ×10)

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Figure 5: HPC-Hemangiopericytoma - like areas (H and E, ×10)

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Morphological diagnosis of BFH of the tongue was considered.

Panel of tumor markers comprising CD34, CD68, vimentin, smooth muscle actin (SMA), S-100, and desmin were used to exclude the morphological mimics of BFH.

Immunohistochemistry staining demonstrated a weak and focal reactivity for CD34 [Figure 6] and negativity for CD68, SMA, S-100, and desmin. Stromal cells showed diffuse positivity for vimentin.
Figure 6: Weak and focal reactivity for CD 34 (IHC) Immunohistochemistry

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Morphological and immunohistochemical examination confirmed the diagnosis of BFH.

There was uneventful recovery with no evidence of recurrence with periodic follow-up of 3 years and also patient showed an improvement in tongue function on follow-up after surgery.


   Discussion Top


BFH has a plethora of synonyms like dermal dendrocytoma, dermatofibroma, Fibrous dermatofibroma, sclerosing haemangioma, nodular sub epidermal fibrosis and fibroma simplex when it involves the skin. [1]

Stout and Lattes in 1967 first described BFH as a distinct clinico-pathologic entity. [2]

The word 'fibro-histiocyte' renders the meaning that cell of origin for the tumor is either fibroblast or histiocyte. [3] Ultra structural studies have supported that these tumors arise from a tissue histiocyte that has fibroblastic properties and gives rise to both histiocyte like and fibroblastic cells histologically. [4]

Histogenesis remains elusive and controversial and theories related to histogenesis of tumor have undergone considerable revision [Table 1]. [3]

Now, it is considered that the principal cell is fibroblast and myofibroblast in various morphological manifestations. [3]
Table 1: Histogenesis of BFH


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BFH although not site specific, rarely involves head and neck region. Non-cutaneous sites are buccal mucosa, tongue, gingiva, lower lip, soft palate, alveolar ridge, floor of the mouth, and jaw bones. [5]

In a literature review on the oral and perioral cases of BFH, Gray et al. found that the mean age of patients was 55 years ranging from 12 to 71 years. [6]

Women are more frequently affected than men. Bielamowicz et al. in their study of BFH in the head and neck region found M:F ratio of 2.5:1. [7]

Reactive process, response to viral infection [1] and traumatic injuries are implicated as etiologic agents for non-cutaneous BFH. [5] A case of BFH of the lip is reported secondary to trauma. [8] Cutaneous fibrous histiocytomas can occur as multiple lesions in 1/3 rd of the cases and occur metachronously. Synchronous development can occur in immunosuppressed individuals. [8]

The clinical appearance is related to the location of the tumor. Dysphagia, dyspnea, difficulty in speech and snoring can be the associated symptoms of BFH of tongue. [5] Oral BFH presents as a solitary, painless well-circumscribed nodule. [9]

Microscopically it is composed of biphasic population of fibroblasts and histiocytic cells arranged in storiform pattern and cart wheel patterns with variable proportion of inflammatory cells and foam cells. [1]

Major histopathological variants of BFH are cellular, epithelioid and aneurysmal (angiomatoid). Clear cell change, palisading of nuclei, myxoid change, lipidization (ankle - type), giant cell formation and granular cell change can be attributed to the minor variants. In spite of enhanced cellularity and level of mitotic activity, it is still considered benign and nuclear atypia in the tumor cells may be displayed on a degenerative basis. [1]

Depending upon the cellularity and morphological variations, wide array of histopathological differentials exists for BFH, which includes nodular fasciitis, solitary fibrous tumor (SFT), neurofibroma, [5] leiomyoma and malignant fibrous histiocytoma. [9]

Nodular fasciitis is a poorly circumscribed lesion consisting of short irregular fascicles of fibrous tissue, separated by a myxoid stroma and mitosis is frequent. [4]

SFT is a rare mesenchymal tumor of the oral , which can overlap histologically with BFH by having features such as circumscription, storiform pattern, sclerosis, neural type palisading, and hemangipericytroma like areas. in BFH, uniformly hypercellular areas are seen and the storiform pattern is wide spread with less frequent sclerosis. [10] in SFT, histiocytic differentiation is absent and alternating hypo and hyper cellular areas are seen. [10] immunohistochemically, CD34 shows weak and focal positivity in BFH where as it shows strong and diffuse positivity in SFT. [10]

S-100 protein positivity can be used to differentiate neurofibroma from BFH. [2]

The cellularity in BFH might resemble smooth muscle tumors and it needs to be differentiated. Leiomyoma displays uniform fascicular pattern of plump eosinophilic spindle cells with the cigar shaped nuclei and shows diffuse SMA positivity. [3]

Malignant fibrous histiocytoma (MFH) is a high grade sarcoma and is less frequent in the deep soft-tissues of the head and neck region. [7] Presence of infiltrative pattern, necrosis, cellular atypia, nuclear pleomorphism and abnormal mitosis helps to differentiate BFH from MFH. [9]

The recurrence rate of BFH is low and metastasis is never reported in oral BFH. [2]


   Conclusion Top


The tongue is the rarest intra oral soft-tissue site for BFH and to date, there have been only three documented cases in the literature. [5] BFH is a versatile and distinct soft-tissue neoplasm which is uncommon in the tongue and needs recognition as having an elusive histogenesis and wide spectrum of morphological variants. An emphasis on histogenesis and differentials of BFH is made in the present article along with a case report and review of the literature.

 
   References Top

1.Enzinger FM, Weiss SW. Soft Tissue Tumors. 4 th ed. St Louis: C V Mosby; 1995.  Back to cited text no. 1
    
2.Heo MS, Cho HJ, Kwon KJ, Lee SS, Choi SC. Benign fibrous histiocytoma in the mandible. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2004;97:276-80.  Back to cited text no. 2
[PUBMED]    
3.Montgomery E, Aaron AD. Clinical Pathology of Soft-Tissue Tumors. 1 st ed. Newyork: Marcel Dekker; 2001.  Back to cited text no. 3
    
4.Cawson RA, Binnie WH, Speight PM, Barrett AW, Wright JM. Lucas's Pathology of Tumors of the Oral Tissues. 5 th ed. London: Churchill Livingstone; 1998.  Back to cited text no. 4
    
5.Alves FA, Vargas PA, Coelho Siqueira SA, Coletta RD, de Almeida OP. Benign fibrous histiocytoma of the buccal mucosa: Case report with immunohistochemical features. J Oral Maxillofac Surg 2003;61:269-71.  Back to cited text no. 5
[PUBMED]    
6.Gray PB, Miller AS, Loftus MJ. Benign fibrous histiocytoma of the oral/perioral regions: Report of a case and review of 17 additional cases. J Oral Maxillofac Surg 1992;50:1239-42.  Back to cited text no. 6
[PUBMED]    
7.Bielamowicz S, Dauer MS, Chang B, Zimmerman MC. Noncutaneous benign fibrous histiocytoma of the head and neck. Otolaryngol Head Neck Surg 1995;113:140-6.  Back to cited text no. 7
[PUBMED]    
8.MacLeod SP, Jones JL. Fibrous histiocytoma of the lip secondary to trauma: Report of a case. J Oral Maxillofac Surg 1992;50:1091-3.  Back to cited text no. 8
[PUBMED]    
9.Syed A, Hasan SH, Hosein MM. Oral fibro-histiocytic lesions: Immuno-histological investigations and review of an unusual presentation in the tongue. Pakistan Oral Dent J 2008;28:217-22.  Back to cited text no. 9
    
10.Vargas PA, Alves FA, Lopes MA, Siqueira SA, Menezes LF, Aldred VL, et al. Solitary fibrous tumour of the mouth: Report of two cases involving the tongue and cheek. Oral Dis 2002;8:111-5.  Back to cited text no. 10
[PUBMED]    

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Correspondence Address:
N S Priya
Department of Oral Pathology, V S Dental College and Hospital, Bangalore, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-9290.123419

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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]
 
 
    Tables

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