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ORIGINAL ARTICLE
Year : 2019  |  Volume : 22  |  Issue : 3  |  Page : 127-133

Fibrous histiocytoma of the orofacial region in Nigerians: A retrospective review of 11 cases


1 Department of Dentistry, Faculty of Medical Sciences, University of Jos, Plateau State, Nigeria
2 Department of Oral and Maxillofacial Surgery, Ahmadu Bello University Teaching Hospital, Shika-Zaria, Nigeria
3 Department of Dental and Maxillofacial Surgery, Oral and Maxillofacial Surgery Unit, Aminu Kano Teaching Hospital, Kano State, Nigeria
4 Department of Pathology, Ahmadu Bello University Teaching Hospital, Shika-Zaria, Nigeria

Date of Submission10-Sep-2017
Date of Acceptance24-Apr-2018
Date of Web Publication26-Sep-2019

Correspondence Address:
Dr. Rowland Agbara
Department of Dentistry, Faculty of Medical Sciences, University of Jos
Nigeria
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DOI: 10.4103/smj.smj_65_17

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  Abstract 


Background: Fibrous histiocytoma (FH) is a rare tumor that may exhibit benign, borderline, or malignant features. It has a site predilection for the skin of the extremities. Occurrence in the orofacial area is very rare. Objective: This review highlights the clinic-epidemiologic characteristics of 11 patients with orofacial FH (OFH) diagnosed at a tertiary health facility Materials and Methods: All cases with a histological diagnosis of OFH seen at the oral and maxillofacial surgery clinic of a regional university teaching hospital in Nigeria between May 1995 and June 2016 were retrospectively studied. Data retrieved were analyzed using the Statistical Package for the Social Sciences version 16 (SPSS Inc., Chicago, IL, USA). Findings from descriptive statistics were represented in the form of tables and charts. Results: Eleven patients were seen within the years reviewed, and the most common clinical presentation was jaw swelling. There were gender and site predilections for male individuals and mandible, respectively. Surgery was the main modality of treatment used and this consisted of maxillectomy (n = 1; 9.1%), mandibulectomy (n = 3; 27.3%), soft-tissue excision (n = 2; 18.2%), and a combination of jaw resection with soft-tissue excision (n = 1; 9.1%). Two patients (18.2%) presented with recurrence 1 year and 5 months postsurgery, respectively. Conclusions: OFH is rare. Early diagnosis and adequate margin of excision/resection at the first surgery is important in achieving control, and long-term follow-up remains the rule.

Keywords: Fibrous histiocytoma, males, orofacial, recurrent


How to cite this article:
Agbara R, Fomete B, Obiadazie AC, Omeje KU, Samaila MO, Ajike SO. Fibrous histiocytoma of the orofacial region in Nigerians: A retrospective review of 11 cases. Sahel Med J 2019;22:127-33

How to cite this URL:
Agbara R, Fomete B, Obiadazie AC, Omeje KU, Samaila MO, Ajike SO. Fibrous histiocytoma of the orofacial region in Nigerians: A retrospective review of 11 cases. Sahel Med J [serial online] 2019 [cited 2019 Nov 22];22:127-33. Available from: http://www.smjonline.org/text.asp?2019/22/3/127/267900




  Introduction Top


Fibrous histiocytoma (FH) is the common name for a rare group of tumors with biphasic cell populations, which may exhibit benign, borderline malignant, or malignant features.[1] They may arise from cutaneous or noncutaneous tissues such as subcutaneous tissues, muscles, and bone.[2] FH occurs in various sites such as the retroperitoneum, heart, uterus, lungs, and the head/neck,[3],[4],[5] but the most common site of occurrence is the skin of the extremities. In the head-and-neck region, occurrence in the forehead, cheek, floor of the mouth, neurocranium, and larynx has been reported.[6],[7]

In general, FH has a gender and age-group predilection for male individuals and above the third decade of life, respectively, although it has been reported in all age groups.[8] Etiology is unknown, but some predisposing factors have been implicated in the pathogenesis of FH which include mechanical trauma, chronic burn scar, surgical trauma, chronic infection, and exposure to radiotherapy/chemotherapy.[7],[9],[10]

The clinical presentation of FH is variable and is dependent on the site and nature of the tumor, presence of metastases, and release of inflammatory mediators among other factors. Imaging techniques such as computed tomography (CT) and magnetic resonance imaging (MRI) are useful for preoperative assessment and surgical planning.[11] The definitive diagnosis of FH is histologically aided by other techniques such as immunohistochemistry. Wide surgical excision alone or in combination with chemotherapy and/or radiotherapy is the treatment of choice.[12],[13]

Orofacial FH (OFH) is rare and most of the documented cases are few case reports [Table 1]. From Nigeria, the major publication on OFH (five cases) is from the southwest region. This review highlights the clinicoepidemiologic characteristics of 11 cases of OFH diagnosed in a regional university teaching hospital in Northwest Nigeria.
Table 1: Previous studies on orofacial fibrous histiocytoma

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  Materials and Methods Top


All patients who presented at the oral and maxillofacial surgery clinic of a regional university teaching hospital between May 1995 and June 2016 with orofacial tumors histologically diagnosed as FH were retrospectively studied. Information was sourced from patient case notes and operating theater records. Details sourced included age, sex, site of tumor, duration, signs/symptoms, treatment given, and complications. Data retrieved were analyzed using the Statistical Package for the Social Sciences (SPSS) version 16 (SPSS Inc., Chicago, IL, USA) and Microsoft Office Excel 2007 (Microsoft, Redmond, WA, USA). Findings from descriptive statistics were represented in the form of tables and charts. Ethical approval protocol number ABUTHZ/HREC/G25 was obtained form Ahmadu Bello University Teaching Hospital Health Research Ethics committee.


  Results Top


Age and sex

A total of 11 cases were histologically diagnosed as OFH within the 21-year period reviewed. This consisted of seven males and four females, giving a male-to-female ratio of 1.8:1. Patients' ages ranged from 6 to 52 years with a mean of 32.3 ± 13.8 years. The highest incidence (n = 8; 72.7%) was recorded in patients above the third decade of life [Figure 1].
Figure 1: Age distribution of patients with orofacial fibrous histiocytoma

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Clinical presentation, radiological assessment, and histology

The main presenting complain was jaw swelling (n = 8; 72.7%) [Table 2]. The duration of symptoms ranged from 6 to 72 months with a mean of 36.0 ± 26.7 months. The maximum duration of symptoms for benign FH (BFH) and malignant FH (MFH) in this study was 60 months and 72 months with an average duration of 32.4 months and 40.5 months, respectively. Only one (9.1%) patient had a known predisposing factor, trauma. Of the 11 patients reviewed, the lesions at the initial preoperative consultations were recurrent lesions in 3 (27.3%) patients and primary lesions in 8 (72.3%) patients. The left side of the orofacial region accounted for 5 (45.5%) cases (4 cases of left jaw swellings and 1 case of soft-tissue swellings), while bilateral swellings were recorded in 3 (27.3%) cases (2 cases of bilateral jaw swellings and 1 case of bilateral soft-tissue swellings) [Figure 2]a and [Figure 2]b.
Table 2: Clinical presentation in eleven patients with orofacial fibrous histiocytoma

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Figure 2: (a) Bilateral mandibular benign fibrous histiocytoma in a child: preoperative view. (b) Bilateral mandibular benign fibrous histiocytoma in a child: postoperative view

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Only one (9.1%) patient had a CT scan done [Figure 3], the other patients were reviewed using plain radiographs and one patient had metastases to the lung on chest X-ray at presentation. The histological diagnosis showed that there were slightly more cases of BFH (n = 6; 54.6%) [Figure 4] than MFH [Figure 5]. However, analysis of tumor type relative to sex gave a male-to-female ratio of 1:1 for BFH and 4:1 for MFH, respectively.
Figure 3: Computed tomography scan of a patient with frontonasal benign fibrous histiocytoma. Lesion is isoattenuated to brain tissue

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Figure 4: Section shows whorl of spindle cells having regular nuclei with moderate cytoplasm in benign fibrous histiocytoma (H and E, ×40)

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Figure 5: Section shows sheets of spindle cells having hyperchromatic nuclei and moderate cytoplasm admired with multinucleated giant cells in malignant fibrous histiocytoma (H and E, ×40)

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Treatment and complications

In 7 (63.6%) patients, surgical treatment (maxillectomy [n = 1; 9.1%], mandibulectomy [n = 3; 27.3%], soft-tissue excision [n = 2; 18.2%], and a combination of jaw resection with soft-tissue excision [n = 1; 9.1%]) was used [Figure 6]. Two (18.2%) other patients had oncological (chemotherapy and radiotherapy) treatment. The remaining 2 (18.2%) patients had no treatment documented for reasons such as referral to another center on patient request (following incisional biopsy via Caldwell-Luc antrostomy) and failure to show up for surgery.
Figure 6: Types of treatment used

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Follow-up

The follow-up period ranged from 5 to 27 months with a mean of 10.5 ± 1.1 months and, within this period, of the 9 (81.8%) patients treated, one (11.1%) patient with BFH presented with orbital recurrence 2 years posthemimaxillectomy. A second (11.1%) patient with BFH of the frontonasal region presented 5 months postsurgery with cervical nodal metastases, although the postsurgical histopathological result showed incomplete excision.


  Discussion Top


The orofacial region is involved in many important functions such as speech, mastication, swallowing, vision, and respiration. Moreover, it largely determines the esthetic outlook of an individual. Therefore, tumors in this region, particularly the aggressive and recurrent types, are likely to interfere with these important roles of the orofacial region. Occurrence of FH in the orofacial region is rare,[12] and only few cases have been documented worldwide when compared with cases of FH located in other sites of the body and other tumors of the orofacial region. From the present study, the observation of male gender predilection for OFH is similar to findings from previous studies in the scientific literature.[2],[14]

Occurrence of FH has been reported in all age groups. However, we observed a high incidence of OFH in individuals above the third decade of life, with the highest incidence noted in the 40–49 years' age group (36.4%), followed by the 30–39 years' age group (27.3%). Similar findings of increased preponderance in individuals above the third decade have been reported in previous OFH studies.[6],[15],[16] However, one study [17] showed no increased incidence of OFH in individuals above the third decade of life.

Reported orofacial locations for OFH include the cheek, tongue, floor of the mouth, lip, submandibular and parotid gland regions, forehead, nose, paranasal sinuses, eyelids, zygoma, palate, and jaws.[8],[12],[15],[17] The most common clinical presentation in OFH noted in this study was jaw swelling (buccal, buccolingual, or buccopalatal). This finding is in agreement with earlier studies [16],[17],[18] although the pattern of jaw swelling in relation to buccal and lingual or palatal sites was not well highlighted in these studies. Other presentations reported in the literature include pain, paresthesia, toothache, tooth mobility, epistaxis, and nasal obstruction.[17],[19] Previous studies show a variance in tumor location as some reported a preponderance of soft-tissue OFH,[8],[12] while others report a preponderance of osseous cases.[18] For soft-tissue OFH, the most common affected orofacial location is the buccal mucosa, followed by the tongue,[12],[15] while the most common reported site for the osseous OFH is the mandible.[16] Similarly, we report a mandible osseous-site predilection although other studies report maxillary-site predilection.[7],[20]

FH may also be associated with systemic manifestations and this is observed in the inflammatory subtype of MFH. Clinical features include fever, weight loss, malaise, and acute-phase response. These systemic manifestations are attributed to the effect of cytokines such as tumor necrosis factor, interleukin (IL)-2, and IL-6.[21] In some other cases, patients may present with paraneoplastic syndromes or local/distant metastases.[22] None of the patients in this study presented with systemic inflammatory manifestations or paraneoplastic syndromes. However, one patient had distant metastases to the lungs at the first presentation. Histological vascular invasion and the presence of leukemia/lymphoma have been identified as significant risk factors for distant metastases.[23],[24]

From this study, the peak durations of symptoms prior to presentations for both BFH and MFH were higher than peak duration reports from previous studies.[7],[8],[17] Delayed presentation remains a challenge in our environment, and some of the factors responsible in this part of the country include poverty, inadequate number of maxillofacial surgeons/oral pathologists, and cultural and religious beliefs. In some cases, patients presented early to hospital within their locality but symptoms were treated as “simple toothache” without proper examination and then later referred following disease progression. Therefore, nondental causes of toothache should always be bore in mind when evaluating patients with odontalgia. However, it is unclear if these patients were examined by qualified dental surgeons since patients in this region commonly refer to most health workers as “likita” (doctor).

Various risk factors have been implicated in the development of FH and these include trauma, postradiation exposure, chronic burn scar, surgical incisions, and some inherited conditions such as hereditary bone dysplasia with MFH.[9],[10] In this study, only one patient had a known risk factor, trauma.

Radiological features of FH depend on the imaging technique used. Plain radiography, CT, and MRI have been used in patient assessment. The features on CT and MRI are usually nonspecific.[11] MFH appears isoattenuated to muscle on CT scan, while with MRI, it is isointense to muscle on T1-weighted images and heterogeneously hyperintense on T2-weighted images. However, some lesions may appear hypoattenuated or a mixture of isoattenuated and hypoattenuated on CT.[8],[11] BFH has no specific features and cannot be safely differentiated from MFH.[25] The radiographic features of FH on plain radiograph include cortical thinning and expansion, unilocular/multilocular radiolucency, and rarely periosteal reaction.[17],[26] In this study, only one patient had CT scan done, the others were reviewed with plain radiographs. The absence of CT machine in our center prior to the year 2006 and patient's inability to afford the cost of CT scanning accounted for the high use of plain radiographs in preoperative assessment. CT scanning in comparison to plain radiography and MRI has the added advantage of enabling the assessment of mineralization pattern, lesion density, and bone and vascular involvement in soft-tissue cases.[27] However, plain radiographs when combined with good clinical assessment by experienced clinicians is usually adequate for preoperative evaluation in resource-limited settings.

Histologically, there are differences between BFH and MFH. Under hematoxylin and eosin stain, the latter has pleomorphic appearance with numerous typical and atypical mitotic figures and prominent areas of hemorrhage and necrosis.[7] Controversy among pathologists over the years has been on whether FH is a morphologic pattern or a true pathologic entity. The earlier notion that it is a biphasic tumor with histiocytic and fibroblastic origin has been disputed and traditionally known histiocytic markers (CD68, 1-antitrypsin, 1-antichymotrypsin, and factor XIII) have been found to be nonspecific and therefore no longer play a useful role in the diagnosis of FH.[27] The availability of advanced histopathological techniques (such as immunohistochemical and electron microscopic techniques) in recent years has led to a better understanding of the true nature of this tumor. As a result, the hypothesis that histiocytes are the progenitor cells is presently no longer acceptable and FH is viewed rather as a morphologic pattern and most cases have been found to be well-differentiated or dedifferentiated sarcomas.[27] It has been suggested that the term MFH should be reserved for undifferentiated tumors and the World Health Organization classification (2013) grouped MFH under undifferentiated sarcomas.[28] However, the use of the term MFH has persisted among pathologists and clinicians.[29] MFH exhibits a wide range of histologic appearance and this has been classified into five subtypes: myxoid (myxofibrosarcoma), storiform-pleomorphic, inflammatory (xanthosarcoma and malignant xanthogranuloma), giant cell (malignant giant cell tumor of soft parts), and angiomatoid variants.[29] In this retrospective study, the histological subtypes were not documented in all the 11 cases.

Wide surgical excision/bone resection alone or in combination with chemotherapy and radiotherapy is the main modality of treatment. Adequate treatment for BFH is achieved by wide local excision/resection into apparently normal tissues.[12],[15],[16] Local recurrence following adequate margin of excision/resection is low and generally, BFH has low metastatic potential. However, where frozen section is not available to determine adequacy of the margin of resection, we recommend a delayed reconstruction. Radiotherapy and chemotherapy have not been found to confer significant benefit in the treatment of BFH and therefore have no role in its management.[30] In this study, two (18.2%) patients presented with recurrence 2 years and 5 months postsurgery, respectively. Although a benign lesion, cases of metastases have been recorded in the literature and the risks of recurrence/metastasis appear to be related to the variant of the tumor, incomplete excision, and multiple surgeries.[1] One of the patients with BFH in this study who had recurrence and metastases have been operated upon twice previously and his histological report showed incomplete excision.

The use of adjuvant radiotherapy in MFH has been associated with increased local tumor control but appears to have no benefit on survival rate and in preventing distant metastases. Similarly, adjuvant chemotherapy using intra-arterial cisplatinum and intravenous doxorubicin has been shown to improve survival.[13] Prognostic factors in MFH include clinical stage, histological grade of malignancy, histological subtype, and local recurrences. In this study, two patients with MFH were mainly managed using radiotherapy due to the inoperable nature of their disease. Poor outcome has been associated with chemotherapy alone in our center. This study being retrospective is constrained by missing data.


  Conclusions Top


FH of the orofacial is rare, common above the third decade of life, and appears to be predominantly osseous in occurrence in this environment. Early diagnosis and adequate margin of excision/resection at the first surgery is important in achieving control. In view of the possibility of recurrence, long-term follow-up remains the rule.

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.



 
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Hugate RR, Wilkins RM, Kelly CM, Madsen W, Hinshaw I, Camozzi AB, et al. Intraarterial chemotherapy for extremity osteosarcoma and MFH in adults. Clin Orthop Relat Res 2008;466:1292-301.  Back to cited text no. 13
    
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