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An Official Publication of the Indian Association of Oral and Maxillofacial Pathologists


 
  Table of Contents    
CASE REPORT  
Year : 2022  |  Volume : 26  |  Issue : 5  |  Page : 116-118
 

The clinical challenges and dilemma in the management of uncommon maxillary sinus tumors − A report of two cases


1 Department of Head and Neck Surgical Oncology, Tata Memorial Center, Homi Bhabha National Institute, Mumbai, Maharashtra, India
2 Department of Pathology, Tata Memorial Center, Homi Bhabha National Institute, Mumbai, Maharashtra, India

Date of Submission15-Jul-2021
Date of Decision24-Nov-2021
Date of Acceptance24-Nov-2021
Date of Web Publication28-Feb-2022

Correspondence Address:
Shivakumar Thiagarajan
Department of Head and Neck Surgical Oncology, Tata Memorial Center, Homi Bhabha National Institute, Mumbai - 400 012, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jomfp.jomfp_236_21

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   Abstract 


Maxillary sinus is the common site for the nose and paranasal sinus tumors with diverse histopathological types and the treatment for each may differ. Making a histopathological diagnosis on occasion can be challenging. We had two patients presenting with upper alveolus growth in whom establishing the histopathological diagnosis was challenging. Through clinical evaluation, imaging (computed tomography and/or magnetic resonance imaging) and identification of key histopathological features helped in the management of these patients.


Keywords: Giant cell tumor, management, maxillary sinus, myofibroblastic tumor, tumor


How to cite this article:
Jain S, Thiagarajan S, Panjwani P, Sathe P, Ramadwar M. The clinical challenges and dilemma in the management of uncommon maxillary sinus tumors − A report of two cases. J Oral Maxillofac Pathol 2022;26:116-8

How to cite this URL:
Jain S, Thiagarajan S, Panjwani P, Sathe P, Ramadwar M. The clinical challenges and dilemma in the management of uncommon maxillary sinus tumors − A report of two cases. J Oral Maxillofac Pathol [serial online] 2022 [cited 2022 Nov 29];26:116-8. Available from: https://www.jomfp.in/text.asp?2022/26/5/116/338747





   Introduction Top


The maxillary sinus is the most common site of the nasal cavity and paranasal sinus tumors.[1] There are a plethora of histopathological types arising from this site which include both benign and malignant.[1] It is very important to establish the histopathological diagnosis for these tumors as this could dictate their further management.


   Case Report Top


We had two patients presenting to us with upper alveolus growth of a few months' duration following a dental procedure. Both patients were evaluated with imaging. In Case 1 [Figure 1]a, magnetic resonance imaging (MRI) revealed an ill-defined heterogeneously enhancing soft-tissue lesion with central scarring and calcifications involving the maxillary sinus, retro-maxillary space and infratemporal fossa suggestive of reparative giant cell granuloma [Figure 1]b. Biopsy was suggestive of giant cell rich lesion, with no evidence of malignancy. Brown's tumor was one of the differential diagnoses which were ruled out after appropriate investigations. A decision to perform an excisional biopsy was taken in the tumor board; however, the procedure could not be completed due to profuse bleeding during the procedure for which angioembolization was necessitated. The report of the biopsy done during this procedure did not add to the diagnosis. Hence, a complete surgical excision of the tumor with appropriate reconstruction was done. Histopathologically, it was a giant cell-rich lesion with evenly distributed multinucleated osteoclasts. The stroma showed areas of hemorrhage and fibroblastic proliferation. No stromal atypia or abnormal mitotic figures were seen [Figure 1]c. The overall features were favoring a giant cell tumor (GCT) [Figure 1]d.
Figure 1: (a) Clinical image of right upper alveolus growth in case.1, (b) MRI of case.1 showing the extensive involvement of the right maxilla, (c) Microphotograph of the tumor with hematoxylin and eosin stains at 400× showing evenly distributed multi nucleated osteoclasts, (d) Microphotograph of the tumor with hematoxylin and eosin stains at 200x showing areas of hemorrhage and fibroblastic proliferation

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In case. 2 [Figure 2]a MRI showed an ill-defined mass in the left maxillary alveolar region [Figure 2]b. The maxilla appeared eroded on imaging and the biopsy was suggestive of spindle cell lesion (low grade) with myofibroblastic proliferation. A decision to perform surgery was taken in the tumor board, and an infrastructure maxillectomy with reconstruction was performed. Microscopically, it was moderately cellular spindle cell tumor, composed of stellate/spindle cells situated in a myxoid stroma [Figure 2]d. There was no necrosis with very occasional mitotic activity and no atypical mitoses. In immunohistochemistry, the cells showed focal expression for desmin [Figure 2]c. The overall features were consistent with a low-grade myofibroblastic tumor, FNCLCC grade 1.
Figure 2: (a) Clinical image of the left upper alveolus growth in case.2, (b) MRI picture of case.2 showing the upper alveolus growth with bone erosion, (c) Microphotograph of the tumor with the cells showing focal expression for Desmin on IHC, (d) Microphotograph of the tumor with hematoxylin and eosin stain at 400× showing stellate/spindle cells situated in a myxoid stroma

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   Discussion Top


Some of the differential diagnoses for GCT are giant cell granuloma, Brown's tumor, aneurysmal bone cyst, phosphaturic mesenchymal tumor and tenosynovial GCT (TSGCT).

A giant cell granuloma shows unevenly distributed giant cells with areas of hemosiderin deposition and reactive bone.[2],[3],[4],[5] The giant cells are usually seen arranged around the hemorrhagic areas. It is a benign process, and the common sites are the jaw and craniofacial bones. These tumors are usually unencapsulated.

Brown's tumor of hyperparathyroidism shows multiple bony lesions with similar histologic features such as giant cell granuloma.[6] It is essential to perform serum PTH and serum alkaline phosphatase in these patients.

An aneurysmal bone cyst is also known to occur in the facial bone and shows air-fluid levels on radiology. Histological features are similar to giant reparative granuloma.[6]

Phosphaturic mesenchymal tumor has a typical clinical association with rickets and osteomalacia.[6] These tumors also show osteoclastic giant cells and a characteristic grungy type of calcification, areas of hemorrhage and ossification. These are usually benign and associated with low levels of serum phosphate and Vitamin D.

TSGCT, also considered to be a fibro-histiocytic tumor is known to affect the temporomandibular joint.[6],[7] Histologic features include osteoclastic giant cells mixed with macrophages, and histiocytic cells containing hemosiderin pigment.

The histopathological diagnosis in our second case was low-grade myofibroblastic tumor. Myofibroblastic tumors are usually seen as deep intramuscular tumors in the extremities and heads-and-neck region. These are tumors having myofibroblastic differentiation, with at least one marker of myogenic differentiation positive.[8] These may mimic other benign fibroblastic proliferation like fibromatosis and nodular fasciitis.[8] There were no features suggestive of a high grade sarcoma.

The histologic features among the various nasal cavity and peripheral nervous system tumors may overlap. A thorough clinical evaluation along with appropriate imaging, which includes computed tomography and/or MRI are essential. The correlation of the clinical, imaging findings and key histologic findings are important to arrive at a definite diagnosis or at least have a few.differential diagnoses based on which treatment for these patients can be planned.

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 initial s 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.
Barnes L. WHO histological classification of tumors of the nasal cavity and paranasal sinuses. In: Barnes EL, Eveson JW, Reichart P, Sidransky D, editors. World Background Documentation Head and Neck Nasal Cavity, Paranasal Sinuses Nasal Cavity Paranasal Sinus 3.0.0.0 22 Health Organization Classification of Tumours: Pathology and Genetics of Head and Neck Tumours. Lyon, France: IARC Press; 2005.  Back to cited text no. 1
    
2.
Murphey MD, Nomikos GC, Flemming DJ, Gannon FH, Temple HT, Kransdorf MJ. From the archives of AFIP. Imaging of giant cell tumor and giant cell reparative granuloma of bone: Radiologic-pathologic correlation. Radiographics 2001;21:1283-309.  Back to cited text no. 2
    
3.
Chrcanovic BR, Gomes CC, Gomez RS. Central giant cell lesion of the jaws: An updated analysis of 2270 cases reported in the literature. J Oral Pathol Med 2018;47:731-9.  Back to cited text no. 3
    
4.
Lee JC, Huang HY. Soft tissue special issue: Giant cell-rich lesions of the head and neck region. Head Neck Pathol 2020;14:97-108.  Back to cited text no. 4
    
5.
Borges BB, Fornazieri MA, Bezerra AP, Martins LA, Pinna Fde R, Voegels RL. Giant cell bone lesions in the craniofacial region: A diagnostic and therapeutic challenge. Int Forum Allergy Rhinol 2012;2:501-6.  Back to cited text no. 5
    
6.
Fisher C. Myofibrosarcoma. Virchows Arch 2004;445:215-23.  Back to cited text no. 6
    
7.
Bisceglia M, Tricarico N, Minenna P, Magro G, Pasquinelli G. Myofibrosarcoma of the upper jawbones: A clinicopathologic and ultrastructural study of two cases. Ultrastruct Pathol 2001;25:385-97.  Back to cited text no. 7
    
8.
Qiu X, Montgomery E, Sun B. Inflammatory myofibroblastic tumor and low-grade myofibroblastic sarcoma: A comparative study of clinicopathologic features and further observations on the immunohistochemical profile of myofibroblasts. Hum Pathol 2008;39:846-56.  Back to cited text no. 8
    


    Figures

  [Figure 1], [Figure 2]


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Surgeries. 2022; 3(3): 153
[Pubmed] | [DOI]



 

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