Home About us Editorial board Ahead of print Current issue Archives Submit article Instructions Subscribe Search Contacts Login 
  • Users Online: 615
  • Home
  • Print this page
  • Email this page


 
 Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 28  |  Issue : 4  |  Page : 433-436

Immunohistochemical analysis of plasmacytoid myoepithelioma of minor salivary glands: A case report with review of literature


1 Department of Oral Medicine and Radiology, Faculty of Dental Sciences, Institute of Medical Sciences, Banaras Hindu University, Varanasi, Uttar Pradesh, India
2 Department of Oral and Maxillofacial Pathology, Faculty of Dental Sciences, Institute of Medical Sciences, Banaras Hindu University, Varanasi, Uttar Pradesh, India
3 Department of Oral Medicine and Radiology, Babu Banarasi Das College of Dental Sciences, Lucknow, Uttar Pradesh, India
4 Department of Oral Pathology and Microbiology, Vananchal Dental College and Hospital, Garhwa, Jharkhand, India

Date of Submission13-Feb-2016
Date of Acceptance20-Dec-2016
Date of Web Publication21-Feb-2017

Correspondence Address:
Dr. Rahul Agarwal
Department of Oral and Maxillofacial Pathology, Faculty of Dental Sciences, Institute of Medical Sciences, Banaras Hindu University, Varanasi, Uttar Pradesh
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-1363.200625

Rights and Permissions
   Abstract 

Myoepithelioma (ME) is a rare benign neoplasm of salivary gland, which commonly originates from parotid gland. Limited number of cases originating from the minor salivary gland tissue have been reported. MEs have characteristic histopathologic and immunohistochemical features, helping in its definitive diagnosis. Here, we present a case of plasmacytoid ME of the palate with review of literature. To the best of our knowledge, only 19 such cases have been reported in literature till date.

Keywords: Myoepithelioma, palatal tumors, plasmacytoid, salivary gland tumors


How to cite this article:
Srivastava A, Agarwal R, Channaiah SG, Chandra A. Immunohistochemical analysis of plasmacytoid myoepithelioma of minor salivary glands: A case report with review of literature. J Indian Acad Oral Med Radiol 2016;28:433-6

How to cite this URL:
Srivastava A, Agarwal R, Channaiah SG, Chandra A. Immunohistochemical analysis of plasmacytoid myoepithelioma of minor salivary glands: A case report with review of literature. J Indian Acad Oral Med Radiol [serial online] 2016 [cited 2019 Apr 18];28:433-6. Available from: http://www.jiaomr.in/text.asp?2016/28/4/433/200625


   Introduction Top


Myoepitheliomas (MEs) are rare benign neoplasms of salivary glands that represent 1.5% of all salivary glands neoplasms.[1] Their most frequent location is parotid glands (40% of all cases). The less common locations include the submandibular gland or the minor salivary glands that are distributed throughout the oral cavity. MEs of the soft or hard palate account for 21% of all cases of MEs and for 93% of intraoral MEs.[2],[3] The term “myoepithelioma” was first introduced by Sheldon in 1943. ME can be defined as a benign, solid tumor composed predominantly or entirely of neoplastic cells of myoepithelial differentiation.[4]Several authors now consider this tumor as being a distinct pathological entity with a biological behavior different from that of mixed tumors, even though ME was once considered to be a variant of pleomorphic adenoma (PA) with exclusive myoepithelial differentiation.[5] The stroma of these tumors is frequently composed of fibrohyalinized or myxoid connective tissue, similar to that seen in some PAs; however, in contrast to the later, ME do not present chondroid or osteoid formation. Moreover, ductal/luminal differentiation is not normally expected in ME and, when present, it constitutes less than 5% of the tumor parenchyma; this is quite useful for distinguishing this lesion from a mixed tumor.[6]

In international literature, the tumor is reported to appear at any age between 8 and 85 years, with an average age of 40 years and a peak in the third decade.[5],[7] Till date, only 19 cases of plasmacytoid MEs of the palate have been reported in scientific literature [Table 1]. With the objective of better understanding this neoplasm, we present a case of plasmacytoid ME of the palate with focus on the clinical, histological and immunohistochemical features along with a review of literature.
Table 1: Reported cases of plasmacytoid myoepithelioma of the palate

Click here to view



   Case Report Top


A 23-year-old female presented with an asymptomatic, slow growing, well-circumscribed, large swelling located on the right side of the hard palate. The medical history of the patient was noncontributory. There was no lymphadenopathy. The lesion was oval, approximately 3.5 × 2 cm in diameter and covered by normal appearing mucosa. It extended anteriorly from the canine region till the junction of the hard and soft palate posteriorly [Figure 1]. Pulp vitality check of the adjoining teeth revealed no non-vital teeth. The occlusal radiograph showed no destruction of the adjacent palatal bone. A provisional diagnosis of PA was made and total excision of the lesion was carried out under local anesthesia.
Figure 1: Large intraoral swelling on the right side of hard palate

Click here to view


The specimen was fixed in 10% buffered formalin and paraffin embedded block was prepared. Sections were taken from the block and stained with Hematoxylin and Eosin [H and E] stain. Microscopic examination revealed large cells with plasmacytoid characteristics presenting round or ovoid eccentric nuclei and a prominent eosinophilic cytoplasm [Figure 2] and [Figure 3]. The plasmacytoid cells were arranged in sheets and lobular pattern, separated by connective tissue septae, with abundant myxoid stroma. The mass was surrounded by a thin, fibrous capsule outside of which small salivary gland acini were found. Ductal and acinar differentiation was absent. There was no evidence of malignancy such as mitotic figures, necrosis, interstitial hemorrhage or infiltration of the adjacent tissues. The histopathological diagnosis was consistent with benign ME of plasmacytoid type.
Figure 2: H and E section showing plasmacytoid myoepithelial cells arranged in sheets and clusters and separated by connective tissue septae (×10)

Click here to view
Figure 3: Plasmacytoid cells with round or ovoid eccentric nuclei and a prominent eosinophilic cytoplasm (×40)

Click here to view


To corroborate the myoepithelial nature of the neoplastic cells and to better understand this neoplasm, the sample was analyzed with immunohistochemical technique. The antibodies used were S-100 protein and α-smooth muscle actin (α-SMA). The nuclei of plasmacytoid myoepithelial neoplastic cells showed strong positive immunoreactions against S-100 protein [Figure 4] while the immunoreactivity of myoepithelial cells against α-SMA was negative [Figure 5]. These findings also confirmed the diagnosis of ME. No evidence of tumor recurrence has been observed on 6-month follow up.
Figure 4: S-100 stained section showing strong immunopositivity (×40)

Click here to view
Figure 5: α-SMA stained section showing negative immunoexpression (×40)

Click here to view



   Discussion Top


The essential component of MEs are myoepithelial cells, which are contractile cells located between the basal lamina and the acinar and ductal cells in exocrine glands including salivary glands. They are ectodermal in origin but function as mesodermal cells.[8] According to some reports, these cells participate in some neoplastic processes such as tumor cell differentiation, invasion, angiogenesis and synthesis of basal membrane.[9]

Clinically, the majority of cases of MEs present as asymptomatic, slow growing, painless, firm, submucosal masses, usually of small size.[10],[11] In our case, the tumor presented as a large swelling which was unusual. Despite no reports of gender predilection, the analysis of plasmacytoid ME of palate cases reported in scientific literature [Table 1] shows a male:female ratio of 1:2 (7 males: 13 females), and the highest prevalence is observed in the third decade of life.[1] However, the average of age of all the reported cases is 37.5 years, and their highest prevalence is ubiquitous in the second decade of life.[5] Our patient was a female of 23 years, which was consistent with previous reports.

In the present case, the neoplastic cells were round-to-oval in shape with eccentric nuclei and eosinophilic hyalinized cytoplasm, thus resembling plasma cells. No ductal/luminal cellular differentiation was seen. These findings were in agreement with the reports in literature and are consistent with the diagnosis of plasmacytoid ME.[4],[12],[13] The immunohistochemical studies have revealed that myoepithelial neoplastic cells show variable immunoreactivity related to cell phenotype. It has been reported to show positive immunoreactivity against S-100 protein, cytokeratin-14, actin muscle specific, vimentin, and fibrillar glycoprotein. On the other hand, negative immunoreactivity has been reported against actin muscle specific.[5] The immunohistochemical study of the current case demonstrated positivity for S-100 protein but not for α-SMA.[10]

The negativity for myogenic markers is expected in the plasmacytoid ME as it frequently lacks myogenic differentiation, even though it is positive for pan-cytokeratin.[11],[12] It has been demonstrated that cultured cell lines derived from plasmacytoid MEs express α-SMA, however, this is not so for the tumor cells of paraffin embedded tissue. These findings suggest that plasmacytoid cells show full myoepithelial differentiation in vitro. Thus, they should be considered myoepithelial-like cells, and the lack of myogenic differentiation in vivo could be due to an inhibitory process mediated by the extracellular matrix.[14],[15] This supports the negative immunoexpression in the neoplastic cells for α-SMA in the present case. Immunoreactivity for S-100 protein is currently considered an important characteristic of this morphologic variant of ME.[5] Similar to the findings of the present case, many studies have reported strong S-100 positivity in this kind of salivary gland tumor.[2],[4],[5],[15]

World Health Organization classifies ME as an independent entity.[1] It has been proposed that, if the neoplasm contains less than 5% of ductal and acinar components, it must be named ME.[1],[13] Establishing specific percentages such as 5% or 10% of determinate histological component, could be subjective for each observer.[5] Finally, epithelial-myoepithelial carcinoma, polymorphous low-grade adenocarcinoma and adenoid cystic carcinoma, as well as inflammatory conditions, benign mesenchymal soft tissue tumors, such as leiomyoma and schwannoma and metastatic tumors, must be excluded.[2],[12] Till now, only 20 cases of plasmacytoid ME of the palate have been reported in literature including the present case [Table 1]. It explains the rarity of this lesion. The choice of treatment for plasmacytoid ME is surgical excision with margin of healthy borders and the potential for recurrence of ME is low.[12] Prognosis is quite favorable, however, patients should undergo regular follow-up examinations to rule out local recurrence.[13]


   Conclusion Top


The key features that helped in diagnosing the case are round-to-oval neoplastic cells with eccentric nuclei and eosinophilic hyalinized cytoplasm resembling plasma cells; no ductal/luminal cellular differentiation was seen; and immunohistochemical study demonstrated positivity for S-100 protein. On the basis of our findings, we support that plasmacytoid ME is an independent and rare entity that should be considered in the differential diagnosis of neoplasms of palatal salivary glands.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Cardesa A, Alos L. Myoepithelioma. In: Barnes L, Everson JW, Reichert P, Sidransky D, editors. Pathology and Genetics of Head and Neck Tumours. Tumours of the Salivary Glands. World Health Organization. Lyon, France: IARC Press; 2005. p. 259-60.  Back to cited text no. 1
    
2.
Ferri E, Pavon I, Armato E, Cavaleri S, Capuzzo P, Ianniello F. Myoepithelioma of a minor salivary gland of the cheek: Case report. Acta Otorhinolaryngol Ital 2006;26:43-6.  Back to cited text no. 2
    
3.
Cruz Perez DE, Lopes MA, Almeida OP, Jorge J, Kowalski LP. Plasmacytoid myoepithelioma of the palate in a child. Int J Paediatr Dent 2007;17:223-7.  Back to cited text no. 3
    
4.
Cuadra ZF, Quezada RD, Tapia VJL, Paez VC, Gaitán CLA. Plasmacytoid myoepithelioma of the palate. Report of one case and review of the literature. Med Oral Patol Oral Cir Bucal 2007;12:552-5.  Back to cited text no. 4
    
5.
Astarci HM, Celik A, Sungu N. Cystic clear cell myoepithelioma of the parotid gland. A case report. Oral Maxillofac Surg 2009;13:45-8.  Back to cited text no. 5
    
6.
Bakshi J, Parida PK, Mahesha V, Radotra BD. Plasmacytoid myoepithelioma of palate: Three rare cases and literature review. J Laryngol Otol 2007;121:13-7.  Back to cited text no. 6
    
7.
Dantas da Silveira EJ, Pereira AL, Fontora MC, de Souza LB, de Almeida Freitas R. Myoepithelioma of minor salivary gland- An immunohistochemical analysis of four cases. Braz J Otorhinolaryngol 2006;72:528-32.  Back to cited text no. 7
    
8.
Santos EP, Cavalcante DR, Melo AU, Pereira JC, Gomes MZ, Albuquerque RL Jr. Plasmacytoid myoepithelioma of minor salivary glands: Report of case with emphasis in the immunohistochemical findings. Head Face Med 2011;7:24-30.  Back to cited text no. 8
    
9.
Zormpa MT, Sarigelou AS, Eleftheriou AN, Assimaki AS, Kolokotronis AE. Plasmacytoid myoepithelioma of the palate: Case report. Head Neck Pathol 2011;5:154-8.  Back to cited text no. 9
    
10.
Kim H-S, Lee WM, Choi SM. Myoepitheliomas of the soft palate: Helical CT findings in two patients. Korean J Radiol 2007;8:552-5.  Back to cited text no. 10
    
11.
Darvishian F, Lin O. Myoepithelial cell-rich neoplasms: Cytologic features of benign and malignant lesions. Cancer 2004;102:355-61.  Back to cited text no. 11
    
12.
Lopez JI, Ugalde A, Arostegui J, Bilbao FJ. Plasmacytoid myoepithelioma of the soft palate. Report of a case with cytologic, immunohistochemical and electron microscopic studies. Acta Cytol 2000;44:647-52.  Back to cited text no. 12
    
13.
Dardick I, Cavell S, Boivin M, Hoppe D, Parks WR, Stinson J, et al. Salivary gland myoepithelioma variants. Histological, ultrastructural, and immunocytological features. Virchows Arch A Pathol Anat Histopathol 1989;416:25-42.  Back to cited text no. 13
    
14.
Açikalin MF, Paşaoğlu O, Cakli H, Ciftçi E. Plasmacytoid myoepithelioma of the soft palate: A review of the literature and report of a case with immunohistochemical findings. Kulak Burun Bogaz Ihtis Derg 2005;14:127-30.  Back to cited text no. 14
    
15.
Raitz R, Araujo VC. Estudo do estado da diferenciação da célula mioepitelial nas neoplasias de glândulas salivares. Acta Scient Heath Sci 2004;26:345-50.  Back to cited text no. 15
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]
 
 
    Tables

  [Table 1]



 

Top
 
 
  Search
 
Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
Access Statistics
Email Alert *
Add to My List *
* Registration required (free)

   Abstract Introduction Case Report Discussion Conclusion Article Figures Article Tables
  In this article
 References

 Article Access Statistics
    Viewed296    
    Printed7    
    Emailed0    
    PDF Downloaded95    
    Comments [Add]    

Recommend this journal


[TAG2]
[TAG3]
[TAG4]