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CORRESPONDENCE
Year : 2014  |  Volume : 10  |  Issue : 2  |  Page : 422-424

Malignant fibrous histiocytoma of maxillary alveolar ridge extending to the hard palate


1 Department of Oral Medicine and Radiology, Rural Dental College, Loni, India
2 Department of Oral and Maxillofacial Surgery, Rural Dental College, Loni, India
3 Department of Oral Pathology and Microbiology, Jamnagar Dental College, Gujarat, India
4 Department of Periodontics, Government Dental College, Aurangabad, Maharashtra, India

Date of Web Publication14-Jul-2014

Correspondence Address:
Vikrant O Kasat
Department of Oral Medicine and Radiology, Rural Dental College, Loni - 413 736, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0973-1482.136678

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 > Abstract 

Malignant fibrous histiocytoma (MFH) is the most common soft-tissue sarcoma occurring in adults. This entity was first described by O'Brian and Stout in 1964. Apart from the soft-tissues, this tumor has been reported to occur in all other parts of the body including bone. It has male predilection and is usually noticed in the fifth to seventh decades of life. It commonly affects extremities and retroperitoneum, whereas involvement of the head and neck region is comparatively rare. However, when the head and neck region is affected, the most common sites are the maxillary sinus and the nasal cavity, in which case, later involvement of the maxillary alveolar bone can happen. The oral cavity is a relatively less common site for MFH. This article describes clinical, computed tomographic and microscopic features of a MFH of the maxillary alveolar ridge extending to the hard palate in a 46-year-old female patient. Histogenesis, advanced imaging features, differential diagnosis, and management of MFH are also discussed.

 > Abstract in Chinese 

上颌牙槽嵴延伸至硬腭的恶性纤维组织细胞瘤
摘要
恶性纤维组织细胞瘤(MFH)是发生在成人的最常见的软组织肉瘤。这种病例实体在1964就由O'Brian和Stout第一次描述了。除了发生在软组织,该肿瘤已经有报道说在身体的所有其他部分发生包括骨都能发生。它偏向于发生于男性,通常是在一生的第五到第七个十年。它通常影响四肢及后腹腔,而头部和颈部区域是比较罕见的。然而,在头部和颈部区域的影响,最常见的部位为上颌窦和鼻腔,在这种情况下,较晚可发生上颌牙槽骨受累。口腔是一个发生恶性纤维组织细胞瘤相对少见的部位。本文介绍了一个46岁的女性患者的上颌牙槽嵴延伸至硬腭的恶性纤维组织细胞瘤的临床,CT和微观特征。组织发生,晚期的显像特点,鉴别诊断以及恶性纤维组织细胞瘤的处理。
关键词:组织细胞瘤,恶性纤维瘤,上颌骨,口腔,肉瘤


Keywords: Histiocytoma, malignant fibrous, maxilla, mouth, sarcoma


How to cite this article:
Kasat VO, Saluja H, Rudagi BM, Kalburge JV, Sachdeva S. Malignant fibrous histiocytoma of maxillary alveolar ridge extending to the hard palate. J Can Res Ther 2014;10:422-4

How to cite this URL:
Kasat VO, Saluja H, Rudagi BM, Kalburge JV, Sachdeva S. Malignant fibrous histiocytoma of maxillary alveolar ridge extending to the hard palate. J Can Res Ther [serial online] 2014 [cited 2019 Sep 17];10:422-4. Available from: http://www.cancerjournal.net/text.asp?2014/10/2/422/136678


 > Introduction Top


Malignant fibrous histiocytoma (MFH) accounts for 20-30% of all soft-tissue sarcomas [1] and is considered as the most common soft-tissue sarcoma in adults. [2],[3],[4],[5],[6] O'Brian and Stout were the first to delineate MFH in 1964. [2],[3],[4],[5],[6] Later on, Kempson and Kyriakos described the histopathological features of MFH. [7] Apart from the soft-tissues, this tumor can occur in all other parts of the body, including bone. [1],[8] MFH of bone was first described by Feldman and Norman in 1972. [9]

The extremities and the retroperitoneum are the most common locations for MFH. [2],[3],[5],[6] Head and neck remains an uncommon location for MFH, as it accounts for only 3-10% of all cases. [1],[6],[8],[10] In the head and neck region, the nasal cavity and paranasal sinuses are the most common sites from which subsequent involvement of maxillary alveolar bone can occur. [3],[4],[6] The oral cavity as such is a less common site for primary MFH and when involved, it mainly affects the jawbones. However, it can also occur in the buccal mucosa, lip, gingiva, tongue, alveolar ridge, and hard palate. [2]

The purpose of this article is to describe a case of MFH in a 46-year-old female affecting the right side of the maxillary alveolar ridge.


 > Case Report Top


A 46-year-old female complained of a painful, rapidly increasing soft-tissue mass in the maxillary right posterior region, beginning 1 month ago. Past dental history revealed that the upper right first molar was extracted 1 year back. Her medical and family history was non-contributory.

Intraoral examination revealed a firm and tender growth on the right side of the hard palate measuring approximately 4 × 3 cm. It extended from the mesial of the canine to the distal of the third molar and was occupying the space of the extracted first molar [Figure 1]. Regional lymph nodes were not palpable. Due to the rapid growth of the lesion, a provisional diagnosis of malignancy was made.
Figure 1: Intra-oral photograph showing growth on the maxillary alveolar ridge and hard palate on right side

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Conventional radiographs revealed an ill-defined radiolucency in the involved region. Computed tomography (CT) scan showed a soft-tissue mass on right side of the hard palate having density similar to that of muscles [Figure 2]a. The lesion involved the alveolar process of the posterior maxilla [Figure 2]b. Routine hematological investigations were within the normal limits.
Figure 2: (a) Axial computed tomography (CT) scan using soft-tissue algorithm showing a soft-tissue mass with uniform density similar to that of muscles on the right side of maxilla. (b) coronal CT scan using bone algorithm showing the lesion on the right side of maxilla causing destruction of alveolar bone without invading the floor of the maxillary sinus

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Incisional biopsy was suggestive of MFH. The tumor was resected along with the alveolar part of the maxilla, extending from first premolar to the maxillary tuberosity antero-posteriorly; medially, the extent of the resection was approximately 1.5 cm towards the midline considering 1 cm of safe margin. The approximate size of the resected specimen was 4 × 3 × 2 cm [Figure 3]a and b. Tissue was sent for histopathological examination, which revealed proliferating pleomorphic fibroblasts and histiocytes with abundance of blood vessels. The fibro-histiocytic cells were arranged in the form of interlacing strands forming a typical storiform pattern. Deeper areas showed metaplastic osteoid and chondroid deposition. Interstitial and perivascular hyalinization was also seen [Figure 3]c and d. Thus, the findings of the incisional biopsy were confirmed. Healing was uneventful [Figure 4] and the patient has been regularly followed-up for the last 2 years without any recurrence.
Figure 3: (a) Intra-operative view after resection of the lesion. (b) Resected specimen. (c and d) Photomicrograph of the lesion (magnification ×20 and ×50 respectively) showing proliferating pleomorphic fibroblasts and histiocytes arranged in the form of interlacing strands forming typical storiform pattern

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Figure 4: Post-operative photograph

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 > Discussion Top


MFH tumors are either primary or secondary. Primary tumors are less aggressive and more common than secondary tumors. [1] Secondary tumors are associated with an underlying condition such as prior radiotherapy, [5],[8] prior trauma, [1] fibrous dysplasia, [6] or benign bone tumors like enchondroma. [1] MFH may occur at any age, with a peak incidence in the fifth to seventh decades. [1],[3],[4],[8],[10] The majority of investigators have reported male predominance with a male: female ratio of 2:1. [1],[3],[4],[8],[10] However, contrary to this, Chen et al. found female predominance with male: female ratio of 1:2. [2] In our case, it was a primary tumor reported in a female who was in her fifth decade.

The head and neck is an uncommon site for MFH. In this region, prevalence of the tumor in the nasal cavity and paranasal sinuses is 30%, craniofacial bones is 15-25%, larynx and soft-tissue of neck is 10-15% each, and oral cavity 5-15%. [1] In the present case, it has affected the maxillary alveolar ridge and the hard palate.

Clinically, MFH in the oral cavity usually presents as an enlarging, painless soft-tissue mass. [1],[2],[6] However, cases have been reported, including the present one, wherein initial presentation was a rapidly, enlarging painful mass. [2] The differential diagnosis of MFH includes squamous cell carcinoma, malignant lymphoma, malignant giant cell tumor, and osteolytic osteosarcoma. [3]

On conventional radiographs, MFH appears as an ill-defined, osteolytic lesion. [6] Park et al. described advanced imaging findings of MFH. On CT scans, MFH appears as a lobulated soft-tissue mass iso-attenuated to muscle. The center of the lesion may show less attenuation due to necrosis, hemorrhage or myxoid material. In 5-20% of the cases calcification can be seen. On MRI, MFH appears as a mass that is isointense to muscle on T1-weighted images and heterogeneously hyperintense on T2-weighted images. [1] In our case, similar findings were observed on conventional radiographs and CT scan, however, an area of necrosis or calcification was not present.

The exact histogenesis of MFH remains controversial. [1],[2],[4],[10] Initially, it was proposed that histiocytes, which characterize the disease were acting as facultative fibroblasts. [4],[8] However, the majority of investigators have suggested that histiocytic and fibrocytic cell lines are derived from the small numbers of undifferentiated mesenchymal cells. [2],[4] Histologically, MFH has been divided into the following subclasses (1) storiform-pleomorphic (2) myxoid (3) giant cell (4) inflammatory (5) angiomatoid. [6],[8] In our patient, the most common type, i.e. storiform-pleomorphic, was present.

The primary treatment for MFH is surgical excision with or without radical neck dissection. [1],[2] As the incidence of regional lymph node metastasis is relatively low (4-17%), [1],[10] elective neck dissection is performed only when there is an evidence of metastasis. In our patient, type I maxillectomy was carried out without radical neck dissection as there was no evidence of regional lymphadenopathy.

The prognosis of MFH depends on various factors such as the tumor grade, size, depth, anatomic location, histologic subtype, and distant metastases. [1],[6] It has been suggested that 25-35% of patients with MFH of head and neck region will develop metastasis, most often to the lung (90%) followed by bone (8%), and liver (1%). [1],[2] The giant cell type of MFH has been thought to carry the worst prognosis. [2] Considering all the above factors, this patient seems to have a good prognosis.

Recurrence rate of MFH varies from 16% to 52%. Hence, close follow-up after treatment is important. The single most important factor responsible for local recurrence is the presence of a positive surgical margin after surgical resection. [8] Our patient has been regularly followed-up for 2 years and no recurrence has been noted so far.

 
 > References Top

1.Park SW, Kim HJ, Lee JH, Ko YH. Malignant fibrous histiocytoma of the head and neck: CT and MR imaging findings. AJNR Am J Neuroradiol 2009;30:71-6.  Back to cited text no. 1
    
2.Chen YK, Lin LM, Lin CC. Malignant fibrous histiocytoma of the tongue. J Laryngol Otol 2001;115:763-5.  Back to cited text no. 2
    
3.Sato T, Kawabata Y, Morita Y, Noikura T, Mukai H, Kawashima K, et al. Radiographic evaluation of malignant fibrous histiocytoma affecting maxillary alveolar bone: A report of 2 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2001;92:116-23.  Back to cited text no. 3
    
4.Senel FC, Bektas D, Caylan R, Onder E, Gunhan O. Malignant fibrous histiocytoma of the mandible. Dentomaxillofac Radiol 2006;35:125-8.  Back to cited text no. 4
    
5.Balaji SM. Malignant fibrous histiocytoma-case report. J Maxillofac Oral Surg 2010;9:292-6.  Back to cited text no. 5
[PUBMED]    
6.Dalirsani Z, Mohtasham N, Falaki F, Bidram F, Nosratzehi T. Malignant fibrous histiocytoma of mandible: A review of literatures and report a case. Aust J Basic Appl Sci 2011;5:936-42.  Back to cited text no. 6
    
7.Kempson RL, Kyriakos M. Fibroxanthosarcoma of the soft tissues. A type of malignant fibrous histiocytoma. Cancer 1972;29:961-76.  Back to cited text no. 7
[PUBMED]    
8.Al-Salihi KA, Al-Jashamy KA, Ab Rahman S, Samsudin AR. Maxilla tuberosity malignant fibrous histiocytoma with giant fibroblastic cells: Case report and review of literature. Oral Oncology Extra 2006; 42:115-22.  Back to cited text no. 8
    
9.Feldman F, Norman D. Intra- and extraosseous malignant histiocytoma (malignant fibrous xanthoma). Radiology 1972;104:497-508.  Back to cited text no. 9
[PUBMED]    
10.Alba García JR, Armengot Carceller M, Zapater Latorre E, Pérez Valles A, Basterra Alegría A. Malignant fibrohistiocytoma of the parotid region. Report of a case. Med Oral Patol Oral Cir Bucal 2008;13:E148-50.  Back to cited text no. 10
    


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