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CORRESPONDENCE
Year : 2013  |  Volume : 9  |  Issue : 2  |  Page : 314-316

Low to intermediate grade salivary duct carcinoma associated with osteoclast like-giant cell tumor of parotid gland: A rare case with distinct pathological features


1 Department of Histopathology, Rajiv Gandhi Cancer Institute and Research Centre, Rohini, Delhi, India
2 Department of Surgical Oncology, Rajiv Gandhi Cancer Institute and Research Centre, Rohini, Delhi, India

Date of Web Publication13-Jun-2013

Correspondence Address:
Jatin S Gandhi
Department of Histopathology, Rajiv Gandhi Cancer Institute and Research Center, Rohini, New Delhi-110085
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0973-1482.113406

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 > Abstract 

Osteoclast like-giant cell tumor of the salivary gland is an extremely rare tumor with distinct pathological features and unknown histogenesis. The neoplastic nature of these tumors in itself is questionable. We present the twentieth case in English literature of primary osteoclast like-giant cell tumor with accompanying low to intermediate grade salivary duct carcinoma of parotid gland, metastasizing to the ipsilateral cervical lymph node. As far as we know this is the second case with lymph node metastasis. Due to the rarity of the tumor its exact biological course is uncertain. We present and discuss this rare case with special emphasis on the histology, immunohistochemistry, and histogenesis.

Keywords: Giant cell tumor, osteoclast, parotid gland, salivary duct carcinoma


How to cite this article:
Pasricha S, Gandhi JS, Mehta A, Gupta G, Tapaswini ­Pradhan. Low to intermediate grade salivary duct carcinoma associated with osteoclast like-giant cell tumor of parotid gland: A rare case with distinct pathological features. J Can Res Ther 2013;9:314-6

How to cite this URL:
Pasricha S, Gandhi JS, Mehta A, Gupta G, Tapaswini ­Pradhan. Low to intermediate grade salivary duct carcinoma associated with osteoclast like-giant cell tumor of parotid gland: A rare case with distinct pathological features. J Can Res Ther [serial online] 2013 [cited 2019 Sep 17];9:314-6. Available from: http://www.cancerjournal.net/text.asp?2013/9/2/314/113406


 > Introduction Top


Osteoclast like-giant cell tumor (OGCT) of salivary glands is an exceedingly rare tumor which was first described by Eusebi et al. in 1984. [1],[2],[3] OGCT comprises of osteoclast like-giant cells (OGC) and mononuclear stromal cells similar to those seen in giant cell tumor (GCT) of the bone. The histogenesis is incompletely understood and the precise origin of the mononuclear cells is largely unexplained. To the best of our knowledge only 19 cases have been published in the world literature but none from India. [2],[3],[4],[5] Approximately half of these are associated with carcinoma of salivary gland. We present a case of OGCT of parotid gland associated with salivary duct carcinoma with emphasis on the cytological, histological, and immunohistochemical aspects.


 > Case Report Top


A 62-year-old hypertensive male presented with a gradually increasing swelling over the right parotid region with associated pain for past 2 months. There was no history suggestive of facial nerve weakness. Local examination revealed a firm, multilobulated parotid swelling measuring 8 × 8 cm. Fine needle aspiration cytology (FNAC) revealed cellular aspirates comprising of biphasic cell population. One population showed mononuclear cell plump to spindle, having bland nuclei with significant mitosis. Many osteoclastic giant cells were seen. The other population comprised of few epithelial cells with significant atypia with occasional cell showing intracytoplasmic mucin [Figure 1]a. A diagnosis of OGCT of parotid was offered with a high index of suspicion for an underlying carcinoma. Subsequently the right parotid was excised along with selective neck dissection. Specimen grossly measured 6 × 5.5 × 2 cm. On external aspect it had a gray white and lobulated appearance. The cut surface was gray brown with tumor measuring 5.2 × 4.9 × 1.8 cm with few cystic areas [Figure 1]b. The tumor was grossly infiltrating the surrounding parotid gland. The routine hematoxylin and eosin (H&E) stained sections showed predominantly intraductal carcinoma having a cribriform, fenestrated, and solid architecture. The nuclear grade was low to intermediate with conspicuous nucleoli. The invasive epithelial component had cells of mild to moderate pleomorphism with few of them exhibiting conspicuous nucleoli. Mitotic activity was evident within the epithelial component (3-4/10 hpf). The second component comprised of OGCT. The stroma exhibited monomorphic plump, spindle to oval mononuclear cells with significant mitosis (8/10 hpf). The number of nuclei in giant cells ranged from 3 to 30 (average 15). These two tumor components showed area of transition and occasional area showed intermingling. The tumor was seen infiltrating the surrounding normal salivary gland and adjacent soft tissues with perineurial and intraneurial invasion with involvement of the inked resected margin [Figure 2]. Out of 19 lymph nodes isolated, 1 showed metastasis of salivary duct carcinoma with no evidence of OGCT component. The patient underwent intensity-modulated radiation therapy (IMRT) as adjuvant therapy and is doing well after 9 months of follow up.
Figure 1: (a) FNAC Smears showing osteoclastic giant cells and atypical epithelial cells (Giemsa; × 40) with intracytoplasmic mucin vacuoles (black arrow); inset (Giemsa; × 200) (b) Section showing a solid-cystic gray white tumor with infiltrating margins

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Figure 2: (a) section showing intraductal component of the salivary duct carcinoma, (H and E, × 40) (b) section showing intraductal component as well as invasive component of the salivary duct carcinoma, (H and E, × 200) (c) section showing transition zone of invasive salivary duct carcinoma and OGCT. Also observed is intraneurial invasion of salivary duct carcinoma, (H and E, × 200) (d) section showing transition zone of intraductal salivary duct carcinoma and OGCT, (H and E, × 200)

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The detailed immunohistochemistry (IHC) findings are mentioned in [Table 1]. The salivary duct carcinoma cells were positive for cytokeratin (CK), CK7, CK8, p53, and carcinoembryonic antigen (CEA) and negative for CD68, vimentin and CK5. The mononuclear cells were positive for vimentin, epithelial membrane antigen (EMA), and negative for CK, p-63, CEA, and CD68. The osteoclastic giant cells were positive for CD68 and vimentin only [Figure 3]. The intraductal component showed presence of intact myoepithelial cell layer which was highlighted by p-63; however, it was fractured in areas where the invasive component was amalgamating with the giant cell component. In view of the morphology and IHC features a diagnosis of OGCT with salivary duct carcinoma, low to intermediate grade was rendered.
Figure 3: (a) Immunostains with EMA showing positivity in salivary duct carcinoma and also in few mononuclear cells. (DAB; × 100) (b) Immunostains with pan CK showing positivity only in the salivary duct carcinoma component (DAB; × 100) (c) Immunostains with CEA showing positivity only in the salivary duct carcinoma component (DAB; × 100) (d) Immunostains with CD68 showing positivity in the osteoclastic giant cells and in few mononuclear cells only. (DAB; × 100)

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Table 1: Shows the panel of antibodies used for immunohistochemistry with there expression in the carcinoma cells, mononuclear cell and OGC

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 > Discussion Top


Multinucleated giant cells histologically similar to osteoclasts of GCT of bone have been described in neoplasms of parenchymal organs like thyroid, breast, colon, lung, ovary, kidney, and bladder. However, OGCT of the salivary gland is exceedingly rare. [6],[7],[8] The most common symptoms were a rapidly enlarging mass with/without pain with rare involvement of facial nerve. Lymph nodal metastasis at the time of diagnosis has been found previously in only one case. [3] In the presented case the mass was gradually increasing with recent history of pain and a lymph node metastasis at the time of diagnosis. The age ranges from 28 to 92 years with a male predominance (M:F : 5:1).

OGCT of salivary gland can be pure or accompanied with carcinoma. Among the 19 previously reported cases in parotid gland, 10 (52.6%) cases showed associated carcinoma consisting of salivary duct carcinoma in majority of cases followed by carcinoma ex-pleomorphic adenoma in remaining cases. The presence of the carcinomatous component constitutes significant risk factor for metastasis. [3] The salivary duct carcinoma even when unaccompanied by OGCT has predominantly high grade cytological atypia with very few cases reported exhibiting low to intermediate grade cytological features. [9] In the presented case the tumor had low to intermediate grade cytological atypia, which makes it even rarer. The tumor was extensively sampled for the histomorphological assessment of salivary duct carcinoma and to rule out the presence of foci showing high grade nuclear atypia, brisk mitosis, or comedonecrosis but none was evident [Figure 2]. Hence, with a consensus opinion the grade of salivary duct carcinoma was low to intermediate grade.

Although the OGCT of salivary gland and GCT of bone shows many morphological similarities but following features of OGCT of salivary gland contrasts with the GCT of bone: (1) OGCT of salivary gland has a substantial association of with carcinoma and is biologically more aggressive. (2) OGCT of salivary gland lacks the reactive bone formation at the periphery. Although Tse et al. stated the significant difference between the nuclei of OGC and stromal cells in OGCT-salivary gland, but we could not establish any significant difference except in the area where OGCT was intermingling with the salivary duct carcinoma. [2]

The histogenesis of OGCT in salivary gland and their association with carcinoma has been a matter of debate. Eusebi et al. examined the GCT of salivary gland ultrastructurally and the features of OGC were similar to those of osteoclasts in bone, however, the features of the mononuclear cells were not distinctive enough to determine the histogenesis. [2],[3] According to Tse et al., OGCT salivary gland is more akin to carcinoma and the interspersed osteoclasts may be nonneoplastic component. [1] Moreover, OGCT and associated salivary duct carcinoma shared the similar mutation of allele on chromosome 17p13 and few mononuclear cells were positive for epithelial markers (CK, EMA). The analysis performed demonstrated that OGCT of salivary gland is neoplastic rather than reactive lesion and the author preferred the term osteoclast type giant cell carcinoma.

In conclusion, the identification of an OGCT like neoplasm of salivary gland on FNAC or biopsy specimen should warrant an immediate excision biopsy with extensive sampling to rule out the presence of associated carcinoma, which could be a minor component within the tumor, but a major factor in the prognostic outcome of the patient. Hence, early diagnosis and timely intervention can prevent metastasis and improve the long-term survival of the patient. The purpose of this article is to contribute to the accumulated experience related to the OGCT of salivary gland and its biological behavior.


 > Acknowledgement Top


. Anila Sharma, Department of Histopathology, Rajiv Gandhi Cancer Institute and Research Centre, Rohini, Delhi-110085, India.

 
 > References Top

1.Eusebi V, Martin SA, Govoni E, Rosai J. Giant cell tumor of major salivary glands: Report of three cases, one occurring in association with a malignant mixed tumor. Am J Clin Pathol 1984;81:666-75.  Back to cited text no. 1
    
2.Tse LL, Finkelstein SD, Siegler RW, Barnes L. Osteoclast-type giant cell neoplasm of salivary gland. A microdissection-based comparative genotyping assay and literature review: Extraskeletal "giant cell tumor of bone" or osteoclast-type giant cell "carcinoma"? Am J Surg Pathol 2004;28:953-61.  Back to cited text no. 2
    
3.Kadivar M, Nilipour Y, Sadeghipour A. Osteoclast-like giant-cell tumor of the parotid with salivary duct carcinoma: Case report and cytologic, histologic, and immunohistochemical findings. Ear Nose Throat J 2007;86:628-30.  Back to cited text no. 3
    
4.Kwon MJ, Nam ES, Cho SJ, Shin HS, Kwon JH, Rho YS. Osteoclast-like giant cell tumor of the parotid gland accompanied with carcinoma ex pleomorphic adenoma. Korean J Pathol 2011;45:S84-8.  Back to cited text no. 4
    
5.Fang X, Hicks DG, Hicks W Jr, Zhang S. Osteoclastlike giant cell tumor of the salivary gland. Ann Diagn Pathol 2009;13:114-8.  Back to cited text no. 5
    
6.Torabinezad S, Kumar PV, Hashemi SB, Rahimi A. Osteoclastomalike giant cell tumor of the parotid gland: Report of a case with fine needle aspiration diagnosis. Acta Cytol 2006;50:80-3.  Back to cited text no. 6
    
7.Balogh K, Wolbarsht RL, Federman M, O'Hara CJ. Carcinoma of the parotid gland with osteoclast-like giant cells. Immunohistochemical and ultrastructural observations. Arch Pathol Lab Med 1985;109:756-61.  Back to cited text no. 7
    
8.Batsakis JG, Ordonez NG, Sevidal PA, Baker JR. Osteoclast-type giant cell neoplasms of the parotid gland. J Laryngol Otol 1988 ; 102:901-4.  Back to cited text no. 8
    
9.Delgado R, Klimstra D, Albores-Saavedra J. Low grade salivary duct carcinoma. A distinctive variant with a low grade histology and a predominant intraductal growth pattern. Cancer 1996;78:958-67.  Back to cited text no. 9
    


    Figures

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

  [Table 1]


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