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Year : 2015  |  Volume : 11  |  Issue : 4  |  Page : 1031

Tumor of the maxilla-odontogenic or glandular? A diagnostic challenge and the role of immunohistochemical markers

1 Department of Oral Pathology and Microbiology, College of Dentistry, Government Dental College, Indore, Madhya Pradesh, India
2 Faculty of Dentistry, Basic Dental Sciences, Al-Hawaiyah, Taif University, Kingdom of Saudi Arabia

Date of Web Publication15-Feb-2016

Correspondence Address:
B S Manjunatha
Faculty of Dentistry, Basic Dental Sciences, Al-Hawaiyah, Taif University, Taif
Kingdom of Saudi Arabia
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/0973-1482.147387

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 > Abstract 

Today's practice in medicine has reached remarkable change mainly due to the advances in the field. Odontogenic tumors represent a spectrum of lesions ranging from hamartomas to benign and malignant neoplasms. Rarely, odontogenic tumors pose a challenge due to varied histological features. But appropriate and accurate diagnosis is crucial for further treatment and follow-up as these have an influence on the prognosis. In such situations, immunohistochemical. (IHC) markers play a significant role in the differentiating various lesions. Within its palette of histology, there are multiple histopathological presentations, many a times these features come in an intermixed pattern simulating different origin. We here, report such a case presented in a 70-year-old female came with a complaint of swelling in the posterior maxilla. The microscopic findings were indicative of a benign neoplasm. To know the nature of the lesion and arrive at a diagnosis, many IHC markers were used. Based on all these findings, a final diagnosis of unicystic ameloblastoma was arrived.

Keywords: Ameloblastoma, immunohistochemical, odontogenic tumor, salivary gland neoplasm, swelling of jaw, tumor of maxilla

How to cite this article:
Joshi J, Manjunatha B S, Kumar H, Kumar P. Tumor of the maxilla-odontogenic or glandular? A diagnostic challenge and the role of immunohistochemical markers. J Can Res Ther 2015;11:1031

How to cite this URL:
Joshi J, Manjunatha B S, Kumar H, Kumar P. Tumor of the maxilla-odontogenic or glandular? A diagnostic challenge and the role of immunohistochemical markers. J Can Res Ther [serial online] 2015 [cited 2021 Dec 9];11:1031. Available from: https://www.cancerjournal.net/text.asp?2015/11/4/1031/147387

 > Introduction Top

Pathology encountered in the posterior maxilla could arise from dentition and spread to involve the sinus or vice versa. Large, aggressive lesions of the maxillary posterior area may invade, destroy the walls of maxillary sinus, floor of the orbit and nasal wall. Numerous diagnostic markers are used to assess the cellular lineage and histogenic origin of various neoplasms. Thus, these markers can be used for the confirmatory diagnosis of various tumors.

Odontogenic tumors comprise a complex group of lesions with diverse histopathologic types and clinical behaviors.[1] We present an unusual neoplasm in a 70-year-old female who presented with a hard swelling located in the posterior maxilla. Histopathological findings were indicative of a benign tumor of odontogenic or glandular origin. Various immunohistochemical (IHC) markers were used, and results concluded as plexiform variety of unicystic ameloblastoma.

 > Case Report Top

A 70-year-old female patient reported with a slow growing swelling of the middle face since last 18 months. Facial asymmetry was noted in the left maxilla due to the swelling and was nontender and firm with normal overlying skin [Figure 1] and [Figure 2]. There was no tenderness, history of epistaxis, nasal obstruction, headache, or visual disturbance. Intraoral examination revealed a palpable mass in the left maxillary premolar-molar region and left side of the hard palate measuring about 5 cm × 6 cm. Obliteration of the buccal vestibule on the same area was noted [Figure 3].
Figure 1: Extraoral photograph of the lesion in the left maxilla

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Figure 2: Clinical photograph of the swelling in the left palate extending posterior to pharyngeal part and buccal vestibule

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Figure 3: Contrast-enhanced computed tomography of paranasal air sinuses showing a large well defined nonhomogenous enhancing expansile mass of left maxillary sinus with erosion of walls

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Contrast-enhanced computed tomography of paranasal air sinuses view revealed a large well-defined nonhomogenous enhancing expansile mass of left maxillary sinus with thinning of walls and associated with destruction of left upper alveolus and base of pterygoid plates. Erosion of the left half of the hard palate with displacement of the nasal septum toward the right side was also noted. Based on clinical and radiographic findings, benign odontogenic tumor, salivary gland neoplasms were thought. An incisional biopsy was taken, and a diagnosis of benign salivary gland tumor of maxilla was given. Entire lesion was removed surgically under general anesthesia and sent for histopathological examination [Figure 4]. The healing was uneventful without any complications and was disease free after 6 months follow-up
Figure 4: Photograph showing surgical specimen along with involved teeth

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Microscopically, tumor cells were arranged in irregular islands network of interconnecting strands of cells [Figure 5]. Peripheral cells in strands and islands were slightly columnar without any reverse polarity of the nuclei having dense collagenized connective tissue stroma. Myxoid changes of the stroma were found adjacent to epithelial islands. No diagnosis could be drawn and to know the origin and nature of the lesion, a series of IHC staining containing cytokeratin (CK8 and 18, CK5, CK6) and pancytokeratin for odontogenic and other markers indicated for salivary gland origin such as calretinin, Ki-67, laminin, smooth muscle actin (SMA), S-100, vimentin were done. The results [Figure 6] and [Figure 7] are enumerated in [Table 1]. After correlating the clinical, radiological and IHC findings, a final diagnosis of “plexiform type of unicystic ameloblastoma” was made.
Figure 5: Photomicrograph showing sheets of spindle-shaped cells arranged in a network or interconnecting strands of cells (H and E, ×100)

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Figure 6: Photomicrograph showing positive immunostaining with cytokeratin (CK8 and 18) (a and b), CK5 and 6 (c and d) and pancytokeratin (e and f) in most of the cells of tumor islands with little connective tissue stroma (blue color)

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Figure 7: Photomicrograph showed most tumor cells negative for calretinin (a), Ki-67 (b), laminin (c), smooth muscle actin (d), S-100 (e), and vimentin (f)

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Table 1: Results of immunohistochemical staining

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 > Discussion Top

The differential diagnosis of lesions in the posterior maxilla includes sinonasal, odontogenic, and minor salivary gland origins. Odontogenic cysts and tumors are high in the list when a lesion is encountered in the tooth-bearing area of the jaws. Ameloblastoma arises from the epithelium of the dental lamina affecting mainly the posterior mandible in about 80% and to a lesser extent the posterior maxilla in about 20%.[2]

In the present case, the lesion comprised of uniform, bland cells with oval and vesicular nuclei and discreet or small nucleoli. The cytoplasm was abundant and appeared eosinophilic. The cells were arranged in various configurations/growth patterns like solid, lobular, tubular, and small papillary areas. The connective tissue stroma showed normal hyaline areas. Hence, we carried out panel of IHC for origin and nature of the lesion. The results are shown in [Table 1] and suggestive of odontogenic origin.

Cytokeratins are a group of intermediate filaments expressed in epithelial cells and are of 20 different types.[3] The patterns of expression of CKs vary with the type of epithelial cells, thus they are used in the differential diagnosis of tumors as a marker of epithelial tumors derived from stratified squamous epithelia, glandular epithelia, and transitional epithelia.[4] Neoplasms related to the odontogenic apparatus may show only the epithelial tissue or associated with odontogenic ectomesenchyme. The IHC detection of different CKs used to know the histogenesis of many epithelial diseases.[5]

Cytokeratin 8 and 19 are normally found in the odontogenic epithelium and react positively to the constituting cells in all types of ameloblastomas.[6] The plexiform type of ameloblastoma is the most primitive form and the CK8 and 19 expressions possibly signify recapitulation of the odontogenic epithelium to the original oral epithelium. Hence, these ameloblastomas are not able to induce hard tissue formation.[4]

Calretinin has been determined as a sensitive marker to discriminate ameloblastoma from other odontogenic tumors.[7] The immunoreactivity of ameloblastoma was restricted to stellate reticulum-like neoplastic epithelial cells and widely expressed throughout the tumor. p63 expression was found in a variety of ameloblastomas.[8] Topographical differences of p63 expression were noted compared to solid odontogenic neoplasms and cystic lesions.[9]

Odontogenic epithelium shows positivity for CK14, but it is gradually replaced by CK19 in preameloblasts and secreting ameloblasts. Odontogenic tumors with epithelial component frequently express CK14 and 19. Numerous studies have shown that ameloblastomas and adenomatoid odontogenic tumors.[10]

According to Fukumashi et al., CK8 and 19 are normally found in the odontogenic epithelium and show positivity in all types of ameloblastomas.[4]

The IHC characteristics were investigated in the present case in an attempt to elucidate the histogenesis. The results showed that the epithelial component of this neoplasm tended to show its distinct odontogenic character and strongly expressed keratin profile for various CKs including CK8 and 18, CK5 and 6 and pancytokeratin shown in [Figure 6]. Other IHC markers indicated for salivary gland origin such as calretinin, Ki-67, laminin, SMA, S-100, and vimentin were negative [Figure 7]. Thus, the lesion was confirmed as odontogenic in nature.[11]

 > Summary and Conclusion Top

Ameloblastoma of maxilla is rare odontogenic neoplasms which are histologically benign and originate from epithelial remnants of the odontogenic apparatus within the jaw bone. Sometimes, IHC markers play a very vital role in diagnosing cases showing combined pattern mimicking lesions of different origin. The present report shows an unusual case in a 70-year-old female who presented with a hard swelling located in the posterior maxilla.

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.

 > References Top

Neville BW, Damm DD, Allen CM, Bouquot JE. Oral and Maxillofacial Pathology. 3rd ed. Philadelphia: Saunders; 2002. p. 701-15.  Back to cited text no. 1
Philipsen HP, Reichart PA, Nikai H, Takata T, Kudo Y. Peripheral ameloblastoma: Biological profile based on 160 cases from the literature. Oral Oncol 2001;37:17-27.  Back to cited text no. 2
Gale N, Westra W, Pilch BZ et al. Epithelial precursor lesions. In: Barnes L, Everson JW, Reichart P, Sindransky D, eds. World Health Organization Classification of Tumours: Pathology and Genetics of Head and Neck Tumours. Lyon: IARC Press, 2005; 177-9.  Back to cited text no. 3
Fukumashi K, Enokiya Y, Inoue T. Cytokeratins expression of constituting cells in ameloblastoma. Bull Tokyo Dent Coll 2002;43:13-21.  Back to cited text no. 4
Wato M, Yu C, Fang YR, He ZH, Wu LY, Bamba Y, et al. Immunohistochemical expression of various cytokeratins in ameloblastoma. Oral Med Pathol 2006;11:67-74.  Back to cited text no. 5
Crivelini MM, de Araújo VC, de Sousa SO, de Araújo NS. Cytokeratins in epithelia of odontogenic neoplasms. Oral Dis 2003;9:1-6.  Back to cited text no. 6
Matsuo A, Ueno S. Immunohistochemical demonstration of keratin in ameloblastoma as an indication of tumor differentiation. J Oral Maxillofac Surg 1991;49:282-8.  Back to cited text no. 7
Alaeddini M, Etemad-Moghadam S, Baghaii F. Comparative expression of calretinin in selected odontogenic tumours: A possible relationship to histogenesis. Histopathology 2008;52:299-304.  Back to cited text no. 8
Gratzinger D, Salama ME, Poh CF, Rouse RV. Ameloblastoma, calcifying epithelial odontogenic tumor, and glandular odontogenic cyst show a distinctive immunophenotype with some myoepithelial antigen expression. J Oral Pathol Med 2008;37:177-84.  Back to cited text no. 9
Kumamoto H, Ohki K, Ooya K. Expression of p63 and p73 in ameloblastomas. J Oral Pathol Med 2005;34:220-6.  Back to cited text no. 10
Premalatha BR, Patil S, Rao RS, Reddy NP, Indu M. Odontogenic tumor markers-An overview. J Int Oral Health 2013;5:59-69.  Back to cited text no. 11


  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]

  [Table 1]


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