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E-JCRT CORRESPONDENCE
Year : 2015  |  Volume : 11  |  Issue : 4  |  Page : 1023

Liposarcoma of the maxillary antrum: A case report


1 Department of Oral Pathology, Yenepoya Dental College, Yenepoya University, Mangalore, Karnataka, India
2 Department of Orthodontics and Dentofacial Orthopaedics, Yenepoya Dental College, Yenepoya University, Mangalore, Karnataka, India

Date of Web Publication15-Feb-2016

Correspondence Address:
G Priyal
PG Student, Department of Oral Pathology and Microbiology, Yenepoya Dental College, Yenepoya University, Nithyanand Nagar, Mangalore - 575 018, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0973-1482.147739

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 > Abstract 


Atypical Lipomatous Tumor/Well-Differentiated Liposarcoma. (ALT/WDL) is a soft tissue sarcoma of intermediate malignant behavior, which most frequently affects the retroperitoneum and lower extremities. Liposarcomas of head and neck are rare, representing only 2-8% of all sarcomas in this region. The majority of liposarcomas occur in middle-aged adults; however, very uncommonly cases have been reported in infancy and early childhood. We report a case of a 14-year-old girl diagnosed as ALT/WDL of the maxillary antrum.

Keywords: Atypical lipomatous tumor, lipoma, liposarcoma


How to cite this article:
Priyal G, Shakil M, Jose M, Hussain A. Liposarcoma of the maxillary antrum: A case report. J Can Res Ther 2015;11:1023

How to cite this URL:
Priyal G, Shakil M, Jose M, Hussain A. Liposarcoma of the maxillary antrum: A case report. J Can Res Ther [serial online] 2015 [cited 2019 Sep 21];11:1023. Available from: http://www.cancerjournal.net/text.asp?2015/11/4/1023/147739




 > Introduction Top


Liposarcoma, an entity described by Virchow, is regarded as the most common soft tissue sarcoma, accounting for 20% of all soft tissue malignancies in adults.[1] It usually involves the deep soft tissues of the trunk and lower extremities, in particular the retro peritoneum, thighs, and buttocks usually arising in adults, with no gender predilection. The occurrence of liposarcoma in the head and neck region is rare, comprising 2-8% of all cases. Most common sites of occurrence in the head and neck region include the larynx, hypo pharynx, soft tissues of the neck, orbit, scalp and oral cavity. Liposarcoma of the oral cavity shows predilection for the cheek,[2] with other sites including the palate, floor of the mouth, gingiva, mandible, and tongue. The World Health Organization (WHO) identifies five variants; WDL/ALT, myxoid, pleomorphic, de-differentiated and mixed. The first two variants are of low grade and are the commonest. WDL/ALT constitutes more than half of the low grade cases. These tumors are often incited in deep soft tissues and affirmed itself as a slowly growing painless mass. Factors considered to be significant in the etiology of liposarcomas include genetics, trauma and irradiation. Usually, the tumor appears firm, relatively fixed to adjacent tissues, encapsulated, and to be growing steadily. Radiological investigations are essential in most cases to determine precise size, identify its localization, limits, extensions of the tumor and its relations with neurovascular structures. It is also necessary for detecting distant metastases.{Table 4}


 > Case Report Top


A 14-year-old girl presented to the department of Oral and Maxillofacial Pathology, Yenepoya Dental College, Mangalore, with the swelling on the right maxillary antrum. The patient had noticed a small swelling 2 years back. There was no history of pain. The size of the lesion increased between discovery and presentation of the lesion at Yenepoya dental college over a time period of one month. Her past medical history and family history was noncontributory. Initial examination revealed a ovoid mass which on palpation was elastic, soft and movable. There was asymmetry in the patient's facial appearance. [Figure 1] and [Figure 2].
Figure 1: Extraoral clinical photograph showing swelling over the right cheek region and asymmetry of face

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Figure 2: Intraoral clinical photograph showing excruciating mass of tissue from the maxillary antrum

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CT scan

Multidirectional CT of paranasal sinus revealed the presence of large soft tissue mass involving the right maxillary antra measuring 36 × 40 mm in size. There was erosion of the lateral walls, floor and anterior maxilla. Exposure of alveolar process of maxillary and apical aspect of the tooth was also observed. Superiorly there was compression of the orbital wall. The lesion showed extrusion onto the soft tissues of cheek and involving the inferior turbinate medially [Figure 3].
Figure 3: Computed tomography image showing tumor mass extruding onto the soft tissues of cheek

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Histopathology

Macroscopically, the pathologic specimen measured 36 × 40 mm in size, soft in consistency and some part of the specimen found to be floating in the bottle filled with formalin. Microscopic examination of formalin-fixed, paraffin embedded tissue sections stained with Hematoxylin and eosin (H and E) showed sheets of cells having clear cytoplasm with eccentrically placed nucleus suggestive of mature fat cells [Figure 4]. In few areas these cells were exhibiting large atypical nuclei with marked pleomorphism. Periphery of the section shows fibrous tissue with spindle shaped cells which are also found to be extending between the tumor cells with presence of lipoblast like cells. Immunohistochemistry was done with S100 and Cytokeratin markers. Immunohistochemistry revealed that tumor cells were positive for S-100 protein [Figure 5] but negative for Cytokeratin [Figure 6], suggesting the presence of fat cells. Based on the microscopic examination and supported by immunohistochemical results, a final diagnosis of Atypical Lipomatous Tumour was established.
Figure 4: H and E showing sheets of cells having clear cytoplasm with eccentrically placed nucleus suggestive of mature fat cells. (×10 magnification)

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Figure 5: Tumor cells showing positivity to S-100. (×10 magnification)

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Figure 6: Tumor cells showing negativity to Cytokeratin. (×10 magnification)

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 > Discussion Top


Liposarcoma of maxillary sinus is extremely rare and only one case has been reported till date. In a study of 256 non-epithelial neoplasms involving the nasal cavity, paranasal sinuses and nasopharynx, only two lesions were classified as adipose tissue tumors, one lipoma and one liposarcoma. Even though adipose tissue tumors are among the most common soft tissue neoplasms, these lesions rarely occur in the nasal cavity, paranasal sinuses and nasopharynx.[3] Liposarcoma usually present as slow growing painless mass in individuals with a peak incidence between 40-60 years of age.[4] Enzinger and Winslow proposed the first classification of liposarcoma, currently used by the WHO, which is grouped histologically into four types- myxoid, well differentiated, round cell, and pleomorphic.[5] The well-differentiated liposarcoma can be further sub-divided into several types, including lipoma like, inflammatory, sclerosing and de-differentiated type. Majority of the head and neck liposarcoma are myxoid type.[6] Liposarcoma clinically can be easily misdiagnosed. Many authors report difficulty in diagnosing and hence histopathology is required for an appropriate diagnosis. Lesions which are considered in the differential diagnosis of ALT/WDL include lipoma, hibernoma with predominant mature fat, myxoid liposarcoma, pleomorphic liposarcoma, fat necrosis, angiomyolipoma and malignant fibrous histiocytoma. Liposarcoma has been described as more firm, less easily compressed and more fixed to adjacent tissue than lipoma.[7] Hibernoma and myxoid liposarcoma lacks atypical nuclei when compared to ALT/WDL. Pleomorphic lipoma lacks mature fat and is highly cellular, fat necrosis is composed of small central nuclei and angiomyolipoma is composed of large epitheloid cells which are absent in ALT/WDL. The treatment of this tumor involves surgical resection that includes a safety margin. Although recurrence of ALT/WDL is thought to be unlikely after complete excision, long term follow up is necessary when considering the pathologic conditions of this tumor at other sites.

In summary, liposarcomas rarely arises in the maxillary sinus. Although, at this time, we have only limited follow-up data for our case, these tumors should be regarded as having the potential for local recurrence if not excised with uninvolved margins. Careful examination of any ALT is advisable to exclude the presence of a small dedifferentiated element, with inherent metastatic risk.

 
 > References Top

1.
Christopher DM. Fletcher, Krishnan Unni K, Fredrik Mertens, Adipocytic tumors. In: Diagnostic Histopathology of Tumors. 2nd ed. London: Churchill Livingstone; 2000;2:1474-85.  Back to cited text no. 1
    
2.
Nikitakis NG, Lopez MA, Pazoki AE, Ord RA, Sauk JJ. MDM2+/CDK4+/p53+oral liposarcoma: A case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2001;92:194-201.  Back to cited text no. 2
    
3.
Fu YS, Perzin KH. Non-epithelial tumors of the nasal cavity, paranasal sinuses and nasopharynx: A clinicopathologic study. VIII. Adipose tissue tumors (lipoma and liposarcoma). Cancer 1977;40:1314-7.  Back to cited text no. 3
    
4.
Enterline HT, Culberson JD, Rochlin DB, Brady LW. Liposarcoma. A clinical and pathological study of 53 cases. Cancer 1960;13:932-50.  Back to cited text no. 4
    
5.
Enzinger FM, Winslow DJ. Liposarcoma. A study of 103 cases. Virchows Arch Pathol Anat Physiol Klin Med 1962;355:367-88.  Back to cited text no. 5
    
6.
Enzinger FM, Weiss SW. Liposarcoma. In: Enzinger FM, Weiss SW, editors. Soft Tissue Tumors. 2nd ed. St Louis: C.V. Mosby; 1988. p. 346-82.  Back to cited text no. 6
    
7.
Wescott WB, Correll RW. Multiple recurrences of a lesion at the base of the tongue. J Am Dent Assoc 1984;108:231-2.  Back to cited text no. 7
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  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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