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  Table of Contents    
CASE REPORT  
Year : 2014  |  Volume : 57  |  Issue : 1  |  Page : 136-138
Suprasellar salivary gland-like pleomorphic adenoma


1 Department of Pathology, Beijing San Bo Brain Hospital, Capital Medical University, Haidian, Beijng, P. R., China
2 Department of Neurology, Beijing San Bo Brain Hospital, Capital Medical University, Haidian, Beijng, P. R., China

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Date of Web Publication17-Apr-2014
 

   Abstract 

Suprasellar salivary gland-Like pleomorphic adenoma is not a common disease and seldom reported so far. We are reporting a case of a 23-year-old man with recurrent suprasellar salivary gland-like pleomorphic adenoma, who underwent an operation of subtotal, subfrontal resection under the wrong pathology diagnosis of benign teratoma in another hospital 4-year-ago. Four years later, he was admitted to our hospital for additional visual loss of the right eye (left, 1.0; right, 0.4) resulting from tumor regrowth. Magnetic resonance imaging revealed that suprasellar extension and compressed optic chiasm resulted in visual disturbance of the patient. The tumor was totally excised and histological examination evidenced the pathological features of intrasellar salivary gland-like pleomorphic adenoma. The patient did not receive any further treatment and he is free from tumor recurrence for 30 months after the operation. From this point of view, clinical prognosis of intrasellar salivary gland-like pleomorphic adenoma was good after total surgical resection.

Keywords: Central nervous system, pathologic findings, pleomorphic adenoma, suprasellar region tumor

How to cite this article:
Yao K, Duan Z, Bian Y, Wang M, Qi X. Suprasellar salivary gland-like pleomorphic adenoma. Indian J Pathol Microbiol 2014;57:136-8

How to cite this URL:
Yao K, Duan Z, Bian Y, Wang M, Qi X. Suprasellar salivary gland-like pleomorphic adenoma. Indian J Pathol Microbiol [serial online] 2014 [cited 2019 Dec 7];57:136-8. Available from: http://www.ijpmonline.org/text.asp?2014/57/1/136/130926



   Introduction Top


The suprasellar salivary gland-like pleomorphic adenoma is not a common disease and seldom reported so far. [1],[2],[3] We experienced a case of suprasellar salivary gland-like pleomorphic adenoma, which was misdiagnosed as pituitary adenoma in pre-operation.


   Case Report Top


Four years ago, a 23-year-old man visited a hospital with polydipsia, polyuria and signs of asymmetrical breast development and visual acuity loss of the right eye (left, 1.0; right, 0.6.) 4-year-ago. Radiographically, a large neoplasm with suprasellar extension was identified and thought to compress the optic nerves and chiasm. He had decreased cortisol, luteinizing hormone (LH) and follicle-stimulating hormone (FSH), and replacement therapy was initiated. His prolactin (PRL) was elevated; adrenocorticotropic hormone (ACTH), growth hormone (GH) and thyroid-stimulating hormone (TSH) were normal. Pre-operative diagnosis of pituitary adenoma was made on the basis of clinical features. Then he underwent an operation of subfrontal, subtotal resection under the pathology diagnosis of benign teratoma in that hospital.

At 4 years later, he complained of additional visual loss of the right eye (left, 1.0; right, 0.4) and intermittent fever for 3 weeks resulting from tumor regrowth. He was admitted to our hospital for a second operation. The serum TSH decreased this time. PRL, GH, LH, ACTH and FSH were normal. Magnetic resonance imaging (MRI) revealed a large mass in the suprasellar region [Figure 1]a. The pre-operative diagnosis was either pituitary adenoma or benign teratoma this time. Craniotomy for tumor removal was performed through a right pterional approach. [1] The tumor was gently removed completely from the hypothalamus and optic nerve.

The patient experienced uneventful recovery. He was well on replacement hormone therapy. MRI obtained 6 days after surgery showed the tumor was removed completely [Figure 1]b. The vision condition improved during the patient's hospitalization period and he was discharged from our hospital 39 days after the operation.
Figure 1: Magnetic resonance images obtained before (a) and 6 days after surgery (b)

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We obtained histological sections at the time of operation which were composed of both epithelial and mesenchymal-like tissues [Figure 2]a. Hyaline-like stroma [Figure 2]a and b], and lots of myxoid stroma in the tumor [Figure 2]a and b] were observed. We also found an element of vague tubule formation in the cellular portion [Figure 2]b], occasionally some were composed of two layers of cells. The cells of tumor were uniform and cuboidal. Most of them were immunohistochemically positive for actin [Figure 2]c, cytokeratin (CK) [Figure 2]d], epithelial membrane antigen (EMA) [Figure 2]e] and glial fibrillary acidic protein (GFAP), meanwhile negative for GH, ACTH, TSH, FSH, LH, PRL, synaptophysin (Syn) [Figure 2]f]. The MIB-1 index of the tumor was 1.2%. From these findings, we made the histological diagnosis of suprasellar salivary gland-like pleomorphic adenoma.
Figure 2: Histological picture of tumor. Both epithelial and mesenchymallike tissues were shown (a) H and E, 100. An element of tubule formation was evident in the tumor (b) H and E, 400. Hyaline like stroma was also observed (a) 100, (b) 400. Cells that made up the ductal structures were immunohistochemically positive for actin (c) 400, cytokeratin (d) 400, epithelial membrane antigen (e) 400. Cells that made up the ductal structures were immunohistochemically negative for synaptophysin (f) 400. Both epithelial and mesenchymallike tissues were shown in the first operation H and E, (g) 200, (h) 400

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   Discussion Top


Salivary gland-like tumor was an extremely rare non-adenomatous tumor specific to the sellar and suprasellar region. It had close relationship to the salivary glands resting in human posterior pituitary. Salivary gland rests have been reported in autopsy specimens. [1] It was supported that the origin of salivary cells in the pituitary was from the epithelium of Rathke's pouch. [2] However, these tissues were considered as clinically silent. Occasionally, large lesions could cause hormonal abnormality, [2],[3],[4] and might rarely give rise to salivary gland-like tumors in the sellar and suprasellar regions.

This tumor was easy to be misdiagnosed as pituitary adenoma in the pre-operation period. We knew many pituitary adenomas were either nonfunctioning or secreting hormones at such low levels that they became symptomatic only when they had grown large enough to produce visual symptoms resulting from compression of the optic chiasm and the normal pituitary. [5] There were also many other tumors enter into the differential diagnosis of a sellar mass. These included craniopharyngioma, pituicytoma, granular cell tumor, meningioma, germcelltumors, malignant lymphoma, gangliocytoma, paraganglioma, hemangiopericytoma, metastatic tumor. Because pituitary adenomas were by far the most frequently occurring lesion, pituitary adenoma was always the first provisional diagnosis in pre-operation.

Many patients with sellar and suprasellar salivary gland-like tumors usually had similar clinical features such as visual disturbances, disorder of hormones levels and cranial nerve involvement. [6] In this case, the clinical features included polydipsia, polyuria, disorder of hormones levels and visual acuity loss. On MRI, large masses of suprasellar region were found. Diagnoses were both pituitary adenoma at two times before operation. It was difficult to make completely correct pre-operation diagnosis merely by clinical history and MRI findings.

We thought the differential diagnosis relayed mainly on the basis of histopathological and immunohistochemical findings. The diagnosis of suprasellar salivary gland-like pleomorphic adenoma was made on the basis of positive immunostaining for CK, EMA, GFAP, the well-differentiated cells with a moderate MIB-1 labeling and the glandular structure. [2],[3],[4],[7] In our case, the tumor cells were positive for CK, EMA and GFAP, but negative for GH, ACTH, TSH, FSH, LH, PRL and Syn. In combination with the hyaline-like stroma structures, the tumor was diagnosed as salivary gland-like monomorphic adenoma of the suprasellar region. We got embedded paraffin sections from his first operation 4-year-ago. Both ductal structures and mesenchymal-like tissues were found in the first specimen [Figure 2]g and h]. Hence, microscopic structures indicating the diagnosis of teratoma was indeed wrong 4-year-ago.

In this case, the tumor was not totally resected in his first operation due to a wrong diagnosis of benign teratoma in other hospital 4-year-ago. We thought incomplete removal was the important reason for relapse. Recurrence of pulmonary artery following surgery was recorded in some reports rising to 44%. [7] Incomplete excision of the tumor was considered as the only proven reason contributing to recurrent disease. [3] This is why a complete resection of the mass was performed in the present case. Options for pleomorphic adenoma of salivary gland treatment included both operation and radiotherapy. [8] Whereas some studies had reported enhanced local control with post-operative radiation therapy compared with surgery alone, others had found no significant improvement in outcome. [9],[10] We found Gamma knife radio surgery not only failed to control the tumor but also changed a prominent portion into degenerated tissue. [4] Hence in the present case, we did not advise the patient to have radiotherapy. The patient is free from tumor recurrence after operation for at least 30 months.

 
   References Top

1.Schochet SS Jr, McCormick WF, Halmi NS. Salivary gland rests in the human pituitary. Light and electron microscopical study. Arch Pathol 1974;98:193-200.  Back to cited text no. 1
[PUBMED]    
2.Chimelli L, Gadelha MR, Une K, Carlos S, Pereira PJ, Santos JL, et al. Intra-sellar salivary gland-like pleomorphic adenoma arising within the wall of a Rathke's cleft cyst. Pituitary 2000;3:257-61.  Back to cited text no. 2
    
3.Hampton TA, Scheithauer BW, Rojiani AM, Kovacs K, Horvath E, Vogt P. Salivary gland-like tumors of the sellar region. Am J Surg Pathol 1997;21:424-34.  Back to cited text no. 3
    
4.Takahashi S, Mikami S, Akiyama T, Kawase T. Intrasellar salivary gland-like pleomorphic adenoma: Case report. Neurosurgery 2011;68:E562-5.  Back to cited text no. 4
    
5.Mangiardi JR. The pathology of non-functioning pituitary adenomas. Semin Diagn Pathol 1986;3:83-94.  Back to cited text no. 5
    
6.van Furth W, Smyth HS, Horvath E, Kovacs K, Salehi F, Cusimano MD. Salivary gland-like tumor of the sella. Can J Neurol Sci 2007;34:478-82.  Back to cited text no. 6
[PUBMED]    
7.Niparko JK, Beauchamp ML, Krause CJ, Baker SR, Work WP. Surgical treatment of recurrent pleomorphic adenoma of the parotid gland. Arch Otolaryngol Head Neck Surg 1986;112:1180-4.  Back to cited text no. 7
[PUBMED]    
8.Lingam RK, Daghir AA, Nigar E, Abbas SA, Kumar M. Pleomorphic adenoma (benign mixed tumour) of the salivary glands: Its diverse clinical, radiological, and histopathological presentation. Br J Oral Maxillofac Surg 2011;49:14-20.  Back to cited text no. 8
    
9.Friedrich RE, Li L, Knop J, Giese M, Schmelzle R. Pleomorphic adenoma of the salivary glands: Analysis of 94 patients. Anticancer Res 2005;25:1703-5.  Back to cited text no. 9
    
10.Liu FF, Rotstein L, Davison AJ, Pintilie M, O'Sullivan B, Payne DG, et al. Benign parotid adenomas: A review of the Princess Margaret Hospital experience. Head Neck 1995;17:177-83.  Back to cited text no. 10
    

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Correspondence Address:
Xueling Qi
Department of Pathology, Beijing San Bo Brain Hospital, Capital Medical University, Haidian District, Beijng, P. R.
China
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Source of Support: This work was supported by Open Research Fund of the Beijing Key Lab of Epilepsy Research (Grant No. 2013DXBL03), Joint Fund for Basic and Clinical Research Cooperation project of Capital Medical University (Grant No. 12JL87), Capital Health Research and Development of special (Grant No. 2011-1020-01), Beijing Municipal Natural Science Foundation (Grant No.7122088) and Beijing Postdoctoral Research Foundation (Grant No. 2013ZZ-22),, Conflict of Interest: None


DOI: 10.4103/0377-4929.130926

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