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Table of Contents
CASE REPORT
Year : 2021  |  Volume : 8  |  Issue : 1  |  Page : 27-30

Benign fibrous histocytoma of the maxilla: A rare case report


1 Senior Lecturer, Department of Oral and Maxillofacial, MES Dental College, Perinthalmanna, Kerala, India
2 Junior Resident, Department of Oral and Maxillofacial Surgery, MES Dental College, Perinthalmanna, Kerala, India
3 Professor, Department of Oral and Maxillofacial, MES Dental College, Perinthalmanna, Kerala, India
4 Senior Lecturer, Department of Oral and Maxillofacial, KMCT Dental College, Mukkom, Kerala, India

Date of Submission19-Dec-2020
Date of Acceptance25-Jan-2021
Date of Web Publication27-Mar-2021

Correspondence Address:
Dr. Abhilash Mathews Thomas
Department of Oral and Maxillofacial Surgery, MES Dental College, Perinthalmanna, Kerala
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijpcdr.ijpcdr_51_20

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  Abstract 


Fibrous histiocytoma can present as malignant or benign and may involve either the soft or hard tissues. The treatment of choice to oral BFH is generally en-block resection. The prognosis is good and the oral BFH recurs only if not completely excised. Hereby presenting a case of a 29 year old male with a chief complaint of pain and swelling in the left side of the face since 2 months. Treatment plan of Subtotal maxillectomy was performed under general anesthesia through a Weber Ferguson incision with infraorbital extension after histopathological and radiological confirmation.

Keywords: Benign fibrous histiocytoma, mandible, maxilla


How to cite this article:
Soman S, Das T A, Thomas AM, Aslam S, Thomas T, VIjayakumar D. Benign fibrous histocytoma of the maxilla: A rare case report. Int J Prev Clin Dent Res 2021;8:27-30

How to cite this URL:
Soman S, Das T A, Thomas AM, Aslam S, Thomas T, VIjayakumar D. Benign fibrous histocytoma of the maxilla: A rare case report. Int J Prev Clin Dent Res [serial online] 2021 [cited 2021 Apr 19];8:27-30. Available from: https://www.ijpcdr.org/text.asp?2021/8/1/27/312236




  Introduction Top


The term “fibrous histiocytoma” is an infrequently heard one when discussing about the lesions and pathologies of the oral cavity. Fibrous histiocytoma can present as malignant or benign and may involve either the soft or hard tissues. The term “malignant fibrous histiocytoma” is a soft-tissue sarcoma that arises as a tumor of histiocytes and fibroblasts and has a significant malignant potential. With the advancements in latest immunohistochemical techniques, malignant and benign forms can be differentiated easily. This has resulted in the establishment of benign fibrous histiocytoma (BFH) as a separate clinical entity.[1],[2],[3] BFH is reported to be present at any age with predominance in male adults (2.5:1). The oral BFH clinically presents as a slow-growing painless solitary swelling. The size of the swelling is highly variable, ranging from <2 cm to more than 10 cm.[4],[5] Other clinical features include dysphagia, dyspnea and when the mass is located in the tongue, difficulty in speech may be also present. Computed tomography (CT) scan is beneficial if the involvement of the bone is suspected. The treatment of choice to oral BFH is generally en bloc resection. The prognosis is good and the oral BFH recurs only if not completely excised. Malignant transformation of oral BFH has not been proved. However, a regular clinical follow-up is recommended.[6]


  Case report Top


A 29-year-old male patient reported to the department of oral and maxillofacial surgery with a chief complaint of pain and swelling in the left side of the face for 2 months. The patient gave a history of pain which is of dull aching type, continuous in nature, and gradual in onset. Pain aggravates on mastication and during night time and subsides on taking medication. Swelling was noted by the patient 2 months back which was painful and has grown up to the present size gradually. The patient gave history of pus discharge intraorally from the swelling which subsided 1 week back.

The patient had visited a private dental clinic 25 days back with the same complaint and had undergone extraction of upper left molars because of mobility and was prescribed with antibiotics.

Extraorally, on examination, mild asymmetry was noted on the left mid face region, no palpable lymph nodes were present. On inspection, solitary diffuse swelling was present on the left middle one third of the face, extending from the ala of the nose to 2 cm anterior to the tragus of the ear anteroposteriorly and 1 cm inferior to the infra orbital rim to the corner of the mouth superoinferiorly. On palpation, the swelling was soft in consistency, nonfluctuant, nonpulsatile, and was fixed to the underlying tissue.

Intraorally, vestibular tenderness was noted irt. 25, 26, 27, 28 edentulous region. An erythematous lesion which was tender on palpation was present irt. 26, 27, 28 region, measured approximately 2 cm × 3 cm [Figure 1].
Figure 1: erythematous lesion irt the 26,27,28 region

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It was provisionally diagnosed with a tumor of the maxillary sinus of the left side.

On radiographic examination, a unilocular radiolucency of size 4 cm × 4 cm was noted on the left posterior maxilla. CT scan revealed an osteolytic lesion involving the left maxilla, extending from the maxillary tuberosity till the canine region. The lesion has invaded the entire maxillary sinus and was seen to be encroaching toward the infraorbital region [Figure 2].
Figure 2: CT image showing the invasion of the lesion into the maxillary sinus

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Incisional biopsy was done, and the features were suggestive of fibrous histiocytoma.

Subtotal maxillectomy was performed under general anesthesia through a Weber Ferguson incision with infraorbital extension [Figure 3]. The defect was managed immediately by the placement of an acrylic obturator with bulb.
Figure 3: subtotal maxillectomy done under GA

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Excisional biopsy report showed cellular proliferation of spindle-shaped cells that are arranged in short fascicles and shows some whorling with hyperchromatic and vesiculated nucleus. Cellular pleomorphism and clear cells with basophilic nucleus were seen. Focal areas show necrosis with inflammatory cell neutrophils. These features were suggestive of fibrous histocytoma – cellular variant [Figure 4].
Figure 4: h/p examination showing cellular proliferation of spindle shaped cells

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  Discussion Top


Fibrous histiocytoma represents a benign but diverse group of neoplasms which exhibit both fibroblastic and histiocytic differentiation. The etiology of BFH is not yet fully understood, and there is a difference in opinion among pathologists as to if this tumor is a true neoplasm, a developmental defect, or a reactive process. The histiogenesis of this tumor is controversial. Based on the tissue culture evidence, it was initially proposed to have a histiocytic origin in which the histiocytes act as facultative fibroblasts. Later on, the 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. However, cell marker studies have supported a fibroblastic origin for these tumors. Histological features of BFH in the bones are similar to that of the soft tissues. Less than 100 cases of BFH of the bone have been reported, and though, it may occur in any bone, it is usually associated with long bones such as femur, tibia, and the pelvic bone. BFH of the jaw bones is extremely rare, and there have been only one English case report of a maxillary case and six reports of mandibular cases. According to the reports, the most common location for BFH was the mandibular posterior region followed by the maxillary posterior region and the mandibular anterior region. In the present case, it affected the maxillary posterior region and proceeded to involve the anterior maxilla within 2 months' time – this is what makes this case peculiar. In five cases, it had affected the left side, whereas the right side was affected in two cases.

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 or ill-defined with or without reactive hyperostotic border. In the present case, it appeared as a well-defined multilocular radiolucency without hyperostotic border.

According to Cale et al.,[7] 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. 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. 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, whereas the nonossifying fibroma is slightly larger and involves a portion of or the entire width of the medullary cavity and may cause spontaneous fracture. 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. BFH occurs in nonlong bones, or even if in a long bone there is lack of metaphyseal involvement. In our case, it was a painful lesion occurring in the adult nonlong 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. Normally, the treatment plan for fibrous histiocytoma is wide resection but considering the nature of the lesion in this case we decided to go for hemi maxillectomy. The prognosis for BFH seems to be excellent, and the recurrence rate of BFH is low unlike the malignant fibrous histiocytoma is the most common soft-tissue sarcoma of late adult life. It is now referred to as undifferentiated pleomorphic sarcoma. It has been categorized into five subtypes: (a) Storiform/pleomorphic, (b) myxoid, (c) giant cell, (d) inflammatory, and (e) angiomatoid. One risk factor is radiotherapy; indeed about 20% of all sarcomas are the undifferentiated/unclassified type and about a quarter of those are radiation related.[8]

The follow-up period in the reviewed case ranged from 1 to 3 years, and no recurrence had been found. In our case, the patient was followed up for 2 years with no recurrence.

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

1.
Skoulakis CE, Papadakis CE, Datseris GE, Drivas EI, Kyrmizakis DE, Bizakis JG. Subcutaneous benign fibrous histiocytoma of the cheek. Case report and review of the literature. Acta Otorhinolaryngol Ital 2007;27:90-3.  Back to cited text no. 1
    
2.
Enzinger FM, Weiss SW. Benign fibrohistiocytic tumors. In: Soft Tissue Tumors. St Louis: Mosby; 1995. p. 293-303.  Back to cited text no. 2
    
3.
Kanazawa H, Watanabe T, Kasamatsu A. Primary malignant fibrous histiocytoma of the mandible: Review of literature and report of a case. J Oral Maxillofac Surg 2003;61:1224-7.  Back to cited text no. 3
    
4.
Yamada H, Ishii H, Kondoh T, Seto K. A case of benign fibrous histiocytoma of the upper lip in a 6-month-old infant. J Oral Maxillofac Surg. 2002;60:451-4.  Back to cited text no. 4
    
5.
Whitaker SB, Muller S, Budnick SD, Wunderle RC, Vigneswaran N. Buccal mass with ulceration. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1997;83:527-31.  Back to cited text no. 5
    
6.
Hoffman S, Martinez MG Jr. Fibrous histiocytomas of the oral mucosa. Oral Surg Oral Med Oral Pathol 1981;52:277-83.  Back to cited text no. 6
    
7.
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. 7
    
8.
Seomangal K, Mahmoud N, McGrath JP. Malignant fibrous histiocytoma, now referred to as Undifferentiated Pleomorphic Sarcoma: A Case Report of an unexpected histology of a subcutaneous lesion. Int J Surg Case Rep 2019;60:299-302.  Back to cited text no. 8
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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