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Table of Contents   
CASE REPORT  
Year : 2014  |  Volume : 25  |  Issue : 1  |  Page : 115-118
Benign fibrous histiocytoma of the maxilla: A case report and review of literature


1 Department of Oral and Maxillofacial Surgery, Rural Dental College, Loni, Maharashtra, India
2 Department of Oral Medicine and Radiology, Rural Dental College, Loni, Maharashtra, India
3 Private Practitioner, Pune, Maharashtra, India
4 Department of Oral Pathology and Microbiology, Jamnagar Dental College, Gujarat, India
5 Department of Oral Pathology and Microbiology, Rural Dental College, Loni, Maharashtra, India

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Date of Submission10-Sep-2013
Date of Decision22-Mar-2013
Date of Acceptance17-Jan-2014
Date of Web Publication21-Apr-2014
 

   Abstract 

Benign fibrous histiocytoma (BFH) is a mesenchymal tumor which commonly affects the skin of extremities. Histologically, BFH of soft tissues and bone shows similar features, but BFH of the bone is rare with less than 100 cases reported worldwide and usually it involves tibia, femur, and pelvic bone. As far as the oral cavity is concerned, majority of the BFHs are found in the soft tissues of the buccal mucosa, gingiva, lower and upper lips, soft palate, and floor of the mouth. Occurrence of this tumor in the jaw bones is extremely rare as only one case of the maxilla and six cases of the mandible have been reported so far . Hence, the purpose of this article is to report the second case of BFH of the maxilla in a 23-year-old female patient and to review the literature of this entity affecting the jaws.

Keywords: Benign fibrous histiocytoma, jaw, mandible, maxilla

How to cite this article:
Saluja H, Kasat VO, Rudagi BM, Dehane V, Kalburge JV, Nikam A. Benign fibrous histiocytoma of the maxilla: A case report and review of literature. Indian J Dent Res 2014;25:115-8

How to cite this URL:
Saluja H, Kasat VO, Rudagi BM, Dehane V, Kalburge JV, Nikam A. Benign fibrous histiocytoma of the maxilla: A case report and review of literature. Indian J Dent Res [serial online] 2014 [cited 2021 Oct 28];25:115-8. Available from: https://www.ijdr.in/text.asp?2014/25/1/115/131160
Benign fibrous histiocytoma (BFH) is a well-established soft-tissue neoplasm that was first described by Stout and Lattes in 1967. [1],[2] It is a mesenchymal tumor [3] and most commonly occurs on the skin of the extremities as a solitary, slowly growing nodule in early to mid-adult life. [1],[2] Occasionally, the BFH lesions arise centrally in the bone, with identical histological features as those of soft-tissue BFH. [1],[4] Primary BFH of the bone is rare, and according to the World Health Organization (WHO) classification of tumors, less than 100 cases have been reported so far with frequent involvement of the pelvic bone, femur, and tibia. [5]

BFH has been reported in the oral cavity and majority of the cases were found in the soft tissues of the buccal mucosa, gingiva, lower and upper lips, soft palate, and floor of the mouth. [1],[2] BFH occurring in the jaw bones is extremely rare. The purpose of this article is to describe the clinical, radiographic, microscopic, and immunohistochemical findings of the second reported case of BFH of maxilla in a 23-year-old female and to review the literature of this lesion in the jaws.

A search of PubMed and Google scholar was made with the keywords "benign fibrous histiocytoma of maxilla," "benign fibrous histiocytoma of mandible," and "benign fibrous histiocytoma of jaw." It was supplemented with hand search to identify related published articles in dental journals. For review, articles published in English language with the lesion present in either jaw were selected. From the review, it is clear that occurrence of this tumor in the jaw bones is extremely rare as only one case of the maxilla and six cases of the mandible have been reported till date [Table 1].


   Case Report Top


A 23-year-old female reported to the Department of Oral and Maxillofacial Surgery with the chief complaint of intermittent and dull aching pain since 8 months and gradually increasing swelling since 3 months in the maxillary left posterior region. Other complaints of the patient were frequent nasal obstruction, nasal discharge, and episodes of epistaxis. Her medical and family history was noncontributory and general physical examination revealed no other abnormalities.

Extraoral examination revealed presence of a diffuse swelling on the left side of the face [Figure 1]. Regional lymph nodes were not palpable. On intraoral examination a well-defined, firm, non-tender swelling was present on the left side, extending from the maxillary canine to the first molar and causing expansion of the cortical plates without perforation. Also, loss of buccal vestibular depth was noted [Figure 2]. The overlying mucosa was pink in color and had smooth surface. The involved teeth did not show signs of mobility or tenderness, but second premolar crown was rotated mesially. A provisional diagnosis of a benign lesion was made.
Figure 1: Extraoral photograph showing diffuse swelling over left maxilla

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Figure 2: Preoperative intraoral view

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Radiographic examination included intraoral periapical radiograph (IOPA) of the upper left posterior region, maxillary occlusal view, orthopantomogram (OPG), Waters' view, and computed tomography (CT) scan. It revealed a well-defined, corticated, multilocular radiolucency in relation to 23-27, causing displacement of 25 and 26, expansion of the buccal and palatal cortical plates, and deviation of the nasal septum [Figure 3]a-c]. Also, there was haziness in the left maxillary sinus [Figure 3]d]. The CT scan showed bone destruction and an expansile heterogeneous soft-tissue mass in the left maxilla, which had encroached upon the left nasal cavity and maxillary sinus [Figure 4]a and b]. Routine hematological and biochemical investigations (full blood count, erythrocyte sedimentation rate, renal and liver function tests, etc.) were all within normal limits.
Figure 3: (a) IOPA and (c) OPG revealed a well-defi ned, corticated, multilocular radiolucency in relation to 23-27, causing displacement of 25, 26. (b) Maxillary occlusal view showing expansion of the buccal and palatal cortical plates and deviation of the nasal septum. (d) Waters' view showing haziness in the left maxillary sinus

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Figure 4: (a) Axial computed tomography (CT) scan showing expansile multilocular lesion on the left side of the maxilla, causing destruction of the nasal wall and maxillary sinus. (b) Coronal CT scan showing the lesion encroaching upon the nasal cavity

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Incisional biopsy report was suggestive of BFH. After performing root canal treatment for the involved teeth, surgical removal of the lesion was done under general anesthesia. For that, crevicular incision was made from the left central incisor to the left first molar region and antral lesion was assessed using Caldwell Luc approach. After removing the anterolateral wall of the sinus completely [Figure 5]a, the lesion was curetted out [Figure 5]b. The tissue was sent for the histopathologic and immunohistochemical examination.
Figure 5: (a) Intraoperative view after enucleation of the lesion. (b) Enucleated lesion

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Microscopic examination showed the tumor mass was composed of elongated, spindle-shaped fibroblasts that were arranged in fascicles and whorls. Proliferation of fibrohistiocytic cells with regular vascular pattern and giant cells scattered throughout the lesion were seen [Figure 6]a. Immunohistochemical staining demonstrated positivity for CD68 (BioGenex, Hyderabad India) [Figure 6]b and α-1-antichymotrypsin (BioGenex, Hyderabad India) [Figure 6]c and negativity for smooth muscle actin (SMA; BioGenex, Hyderabad India) [Figure 6]d. Thus, diagnosis of BFH was confirmed. There were no postoperative complications and no recurrence was seen during the 2-year follow-up period.
Figure 6: (a) Photomicrograph of the lesion showing elongated, spindle-shaped fibroblasts arranged in fascicles and whorls (H and E, ×50). (b) Immunohistochemical staining shows strong positivity for CD68 in moderate number of cells (H and E, ×10). (c) Immunohistochemical staining shows moderate to strong positivity for á-1-antichymotrypsin in more than 50% of cells (H and E, ×10). (d) Immunohistochemical staining shows negativity for smooth muscle actin (SMA) (H and E, ×10)

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   Discussion Top


Fibrous histiocytoma represents a benign but diverse group of neoplasms which exhibit both fibroblastic and histiocytic differentiation. [2] The etiology of BFH is not yet clear and there is disagreement amongst pathologists as to whether this tumor represents a true neoplasm, a developmental defect, or a reactive process. [2],[6] The histogenesis of this tumor is controversial. On account of the tissue culture evidence, it was initially proposed that it has a histiocytic origin in which the histiocytes act as facultative fibroblasts. Later on, results of electron microscopic and ultrastructural studies suggested that the histiocytic and fibrocytic cell lines are derived from the small numbers of undifferentiated mesenchymal cells. Apart from this, cell marker studies have supported a fibroblastic origin for these tumors. [7] Histological characteristics of BFH occurring in the bones are similar to those occurring in the soft tissues. [4],[5] There are less than 100 reported cases of BFH of the bone, and though it may occur in virtually any bone, it is usually found in the long bones like femur, tibia, and the pelvic bone. [6] BFH occurring in the jaw bones is extremely rare [2],[3],[5] and there have been only one English report of a maxillary case [4] and six reports of mandibular cases, [1],[2],[3],[5],[6],[8] as shown in [Table 1].
Table 1: Summary of case reports of benign fi brous histiocytoma in either jaw in chronological order

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The age of the patients in the cases reviewed ranged from 13 to 49 years, with the average of 32.85 years. Among the seven case reports summarized in [Table 1], the incidence was almost equal in males (n = 4) and females (n = 3), with a male: female ratio of 1.33:1. In our case, the patient was a 23-year-old female. According to the reports, the most common location for BFH was the mandibular posterior region [1],[2],[3],[5],[6] followed by the maxillary posterior region [4] and the mandibular anterior region. [8] In the present case, it affected the maxillary posterior region. Duration of BFH was reported in only one case. [1] In five cases, it had affected the left side, whereas the right side was affected in two cases. Thus, it appears that BFH has affinity for the left side, which was also evident in the present case. Swelling alone was the most common symptom in the reviewed cases, followed by pain and swelling, while in two cases, the patients were asymptomatic. Radiographic appearance in the cases reviewed varied from well-defined [2],[8] or ill-defined [3],[4] unilocular radiolucency to well-defined multilocular radiolucency [1],[5],[6] with or without reactive hyperostotic border. In the present case, it appeared as a well-defined multilocular radiolucency without hyperostotic border.

Immunohistochemical staining was done in four cases which showed that the tumor cells were positive for vimentin, [1],[3],[5],[6] CD68, [1],[3],[5],[6] α-1-antitrypsin, [5] and α-1-antichymotrypsin [5] and negative for SMA, [1],[3],[5],[6] S-100 protein, [1],[3],[5],[6] epithelial membrane antigen, [5] cytokeratin, [3],[5],[6] desmin, [5],[6] and CD34. [3],[5] The positivity for CD68 and vimentin indicated that the lesion was composed of histiocytic cells and fibroblast-like cells on immunohistochemistry, and the negativity for SMA and S-100 showed that the lesion could be differentiated from leiomyosarcoma and neurogenic tumors. [1] In the present case, immunohistochemical staining demonstrated strong positivity for CD68 in moderate number of cells, moderate to strong positivity for α-1-antichymotrypsin in more than 50% of cells, and negativity for SMA.

According to the WHO histological classification of the tumors, primary BFH of the bone is defined as a benign lesion composed of spindle-shaped fibroblasts arranged in a storiform pattern with a variable admixture of small, multinucleated osteoclast-like giant cells. Foamy cells (xanthoma), chronic inflammatory cells, stromal hemorrhages, and hemosiderin pigment are also commonly present. [6] In our case, it showed spindle-shaped fibroblasts arranged in a storiform pattern, histiocytes, and giant cells.

According to Cale et al., BFH and the metaphyseal fibrous defect (i.e. the fibrous cortical defect and the nonossifying fibroma) together constitute the benign fibrohistiocytic lesions of the bone. [4] Histological findings of BFH are indistinguishable from those of the fibrous cortical defect and nonossifying fibroma. Hence, they should be differentiated on clinical and radiographic presentations. [4],[5],[6],[8] Fibrous cortical defect and nonossifying fibroma are asymptomatic, self-limiting developmental defects confined to the metaphyseal portion of the long bones in children and adolescents. They are usually discovered as incidental radiographic findings. The fibrous cortical defect appears as a small radiolucent lesion isolated to the cortex of the involved bone, while the nonossifying fibroma is slightly larger and involves a portion of or the entire width of the medullary cavity and may cause spontaneous fracture. [4] On the contrary, though BFH lesions may occur at any age, they are more common in adults and are frequently associated with pain even in the absence of fracture. [4],[5] BFH occurs in non-long bones, or even if in a long bone there is lack of metaphyseal involvement. [5] In our case, it was a painful lesion occurring in the adult non-long bone (maxilla) without bone fracture. It is challenging to distinguish BFH from the giant cell tumors, but in the giant cell tumors, massive and diffuse presence of giant cells is seen. [1],[4]

The prognosis for BFH seems to be excellent and the recurrence rate of BFH is low. [1] The follow-up period in the reviewed case ranged from 1 to 3 years and no recurrence had been found. [1],[2],[3],[5] In our case, the patient was followed up for 2 years with no recurrence. Normally, the treatment plan for BFH consists of a wide surgical resection as mentioned in the literature, but considering the age of the patient and location of the lesion, we decided to curette out the lesion instead of resection of the affected area for the best outcomes (our case was a young female patient in whom the maxillary sinus was involved).


   Conclusion Top


A rare case of BFH occurring in the jaw bone (maxilla) of a 23-year-old female is reported. As the lesion has histopathologic similarity with other lesions, clinical and radiographic correlation is important for differential diagnosis. The prognosis for BFH is good and recurrence is seen only if there is incomplete removal of the lesion. The treatment plan will vary from curettage to wide excision, depending on various factors like the area involved and age of the patient.

 
   References Top

1.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. 1
    
2.Gupta P, Godhi SS, Kukreja P, Bhatnagar S, Lall AB, Singh A, et al. Fibrous Histiocytoma of the Mandible-a case report. J Indian Dent Assoc 2011;5:994-5.  Back to cited text no. 2
    
3.Katagiri W, Nakazawa M, Kishino M. Benign fibrous histiocytoma in the condylar process of the mandible: Case report. Br J Oral Maxillofac Surg 2008;46:e1-2.  Back to cited text no. 3
    
4.Cale AE, Freedman PD, Kerpel SM, Lumerman H. Benign fibrous histiocytoma of the maxilla. Oral Surg Oral Med Oral Pathol 1989;68:444-50.  Back to cited text no. 4
    
5.Kishino M, Murakami S, Toyosawa S, Nakatami A, Ogawa Y, Ishida T, et al. Benign fibrous histiocytoma of the mandible. J Oral Pathol Med 2005;34:190-2.  Back to cited text no. 5
    
6.Wagner ME, Rana M, Traenkenschuh W, Kokemueller H, Eckardt AM, Gellrich NC. Piezoelectric-assisted removal of a benign fibrous histiocytoma of the mandible: An innovative technique for prevention of dentoalveolar nerve injury. Head Face Med 2011;7:20.  Back to cited text no. 6
    
7.Shrier DA, Wang AR, Patel U, Monajati A, Chess P, Numaguchi Y. Benign Fibrous Histiocytoma of the Nasal Cavity in a Newborn: MR and CT Findings. AJNR Am J Neuroradiol 1998;19:1166-8.  Back to cited text no. 7
    
8.Ertas U, Buyukkurt MC, Cicek Y. Benign fibrous histiocytoma: Report of case. J Contemp Dent Pract 2003;4:74-9.  Back to cited text no. 8
    

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Correspondence Address:
Vikrant O Kasat
Department of Oral Medicine and Radiology, Rural Dental College, Loni, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-9290.131160

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    Figures

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

  [Table 1]

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