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  Table of Contents  
Year : 2015  |  Volume : 52  |  Issue : 2  |  Page : 216-217

Pneumocytic adenomyoepithelioma in a case of myoepithelial carcinoma of the submandibular gland

1 Department of Surgical Oncology, Cancer Institute, Adyar, Chennai, India
2 Department of Pathology, Cancer Institute, Adyar, Chennai, India

Date of Web Publication5-Feb-2016

Correspondence Address:
A Krishnamurthy
Department of Surgical Oncology, Cancer Institute, Adyar, Chennai
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/0019-509X.175813

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How to cite this article:
Krishnamurthy A, Vaidhyanathan A, Majhi U. Pneumocytic adenomyoepithelioma in a case of myoepithelial carcinoma of the submandibular gland. Indian J Cancer 2015;52:216-7

How to cite this URL:
Krishnamurthy A, Vaidhyanathan A, Majhi U. Pneumocytic adenomyoepithelioma in a case of myoepithelial carcinoma of the submandibular gland. Indian J Cancer [serial online] 2015 [cited 2021 Aug 5];52:216-7. Available from: https://www.indianjcancer.com/text.asp?2015/52/2/216/175813


A 54-year-old lady was referred to our institution with what was believed to be a second local recurrence of a pleomorphic adenoma (PA) arising in the right submandibular gland; since its initial onset in the year 1994. She presented to us in March 2007 with slowly progressing painless nodular swellings (four in number, the largest being 3 × 3 cm) in the right submandibular region. There was no significant neck adenopathy. Fine needle aspiration cytology (FNAC) of the submandibular lesion was suggestive of an adenoma. A chest radiograph showed a nodular opacity in the right upper zone. Further evaluation with a chest CT showed a well defined 14.0 × 12.0 mm cavitating nodule in the right upper zone; a guided FNAC from the same [Figure 1]a was suggestive of an epithelial neoplasm. She then underwent wide excision of all the nodular lesions along the overlying scarred skin in the submandibular region and a pulmonary metastatectomy in a same setting [Figure 1]b.
Figure 1: (a) Chest CT showing guided FNAC from right lung lesion, (b) Post operative clinical photograph of the patient

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Microscopic examination of the submandibular lesions revealed the tumor amidst compressed glands; immmunohistochemistry stained positive for vimentin, S100, smooth muscle actin, keratin and weak positivity for glial fibrillary acidic protein. About 30% of cells showed strong nuclear positivity for Ki-67; the final impression was that of a PA undergoing malignant transformation to low grade myoepithelail carcinoma (MC) [Figure 2]a,[Figure 2]b.
Figure 2: (a) H and E, ×40: Section of salivary gland tumor showing plump spindle and myoepithelial cells, (b) H and E, ×40: Salivary gland tumor- Many myoepithelial cells show strong nuclear positivity for Ki-6, (c) H and E, ×20: Pneumocytic Adenomyoepithelioma (PAM) of lung showing both glandular and myoepithelial cell component, (d) H and E, ×40: Pulmonary tumor. Ki 67 does not show any proliferative activity by tumor cells

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Examination of the pulmonary specimen revealed the cells arranged as glandular structures with features suggestive of pneumocytic adenomyoepithelioma (PAM) [Figure 2]c,[Figure 2]d. The patient is on follow up for the past three and a half years and remains asymptomatic.

MCs account for less than 1% of salivary gland tumors.[1] Histiogenesis of a MC has been explained by two separate mechanisms; a de novo formation in normal salivary gland and development from a recurrent PA.[2] Recently, it has been suggested that assessment of cell proliferative activity may be helpful in the differential diagnosis between malignant and benign myoepitheliomas, and that more than seven mitoses per 10 HPFs or a Ki 67 labeling index of more than 10% is diagnostic as a myoepithelial carcinoma.[2]

PAMs are distinctive lung tumors with epithelial, myoepithelial and pneumocytic differentiation and are generally low grade tumors.[3] Surgery is the mainstay of treatment for MC and PAM; which consists of meticulous resection with adequate margins taking care to avoid intraoperative tumor spillage as was done in our case for both the primary as well as the lung lesion.[4] The roles of radiotherapy and chemotherapy remain yet to be established. Malignant transformation should be suspected in a long standing benign tumor with a history of rapid growth and/or multiple recurrences in a preexisting pleomorphic adenoma with or without lymph node metastasis.[4] The association of PAM in a case of MC of submandibular salivary gland needs to be further studied and this to the best of our knowledge is the first report in English language literature.

  References Top

Seifert G, Sobin LH. The World Health Organization's Histological Classification of Salivary Gland Tumors. A commentary on the second edition. Cancer 1992;70:379-85.  Back to cited text no. 1
Nagao T, Sugano I, Ishida Y, Tajima Y, Matsuzaki O, Konno A, et al. Salivary gland malignant myoepithelioma: A clinicopathologic and immunohistochemical study of ten cases. Cancer 1998;83:1292-9.  Back to cited text no. 2
Chang T, Husain AN, Colby T, Taxy JB, Welch WR, Cheung OY, et al. Pneumocytic adenomyoepithelioma: A distinctive lung tumor with epithelial, myoepithelial and pneumocytic differentiation. Am J Surg Pathol 2007;31:562-8.  Back to cited text no. 3
Kane SV, Bagwan IN. Myoepithelial carcinoma of the salivary glands: A clinicopathologic Study of 51 cases in a tertiary cancer center. Arch Otolaryngol Head Neck Surg 2010;136:702-12.  Back to cited text no. 4


  [Figure 1], [Figure 2]

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