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Frequent apocrine changes in pleomorphic adenoma with malignant transformation: a possible pre-malignant step in ductal carcinoma ex pleomorphic adenoma
Joon Seon Song, Yeseul Kim, Yoon-Se Lee, Seung-Ho Choi, Soon Yuhl Nam, Sang Yoon Kim, Kyung-Ja Cho
J Pathol Transl Med. 2023;57(3):158-165.   Published online May 10, 2023
DOI: https://doi.org/10.4132/jptm.2023.03.13
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  • 1 Web of Science
  • 1 Crossref
AbstractAbstract PDF
Background
The most common type of carcinoma ex pleomorphic adenoma (CPA) is histologically equivalent to salivary duct carcinoma, which has an apocrine phenotype. Invasive CPA is often accompanied by non-invasive or in situ carcinoma, an observation that suggests the presence of precursor lesions. The aim of this study was to identify candidate precursor lesions of CPA within pleomorphic adenoma (PA).
Methods
Eleven resected cases of CPA with residual PA and 17 cases of PA with atypical changes were subjected to immunohistochemistry (IHC) for p53, human epidermal growth factor receptor 2 (HER2), androgen receptor (AR), pleomorphic adenoma gene 1, gross cystic disease fluid protein-15 (GCDFP-15), and anti-mitochondrial antibody.
Results
Invasive or in situ carcinoma cells in all CPAs were positive for AR, GCDFP-15, and HER2. Atypical foci in PAs corresponded to either apocrine or oncocytic changes on the basis of their reactivity to AR, GCDFP-15, and anti-mitochondrial antibody. Atypical cells in PAs surrounding CPAs had an apocrine phenotype without HER2 expression.
Conclusions
Our study identified frequent apocrine changes in residual PAs in CPA cases, suggesting a possible precursor role of apocrine changes. We recommend the use of HER2 IHC in atypical PAs, and that clinicians take HER2 positivity into serious consideration.

Citations

Citations to this article as recorded by  
  • Characterization of a Molecularly Distinct Subset of Oncocytic Pleomorphic Adenomas/Myoepitheliomas Harboring Recurrent ZBTB47-AS1::PLAG1 Gene Fusion
    Ziyad Alsugair, Jimmy Perrot, Françoise Descotes, Jonathan Lopez, Anne Champagnac, Daniel Pissaloux, Claire Castain, Mihaela Onea, Philippe Céruse, Pierre Philouze, Charles Lépine, Marie-Delphine Lanic, Marick Laé, Valérie Costes-Martineau, Nazim Benzerdj
    American Journal of Surgical Pathology.2024; 48(5): 551.     CrossRef
PLAG1, SOX10, and Myb Expression in Benign and Malignant Salivary Gland Neoplasms
Ji Hyun Lee, Hye Ju Kang, Chong Woo Yoo, Weon Seo Park, Jun Sun Ryu, Yuh-Seog Jung, Sung Weon Choi, Joo Yong Park, Nayoung Han
J Pathol Transl Med. 2019;53(1):23-30.   Published online November 14, 2018
DOI: https://doi.org/10.4132/jptm.2018.10.12
  • 8,554 View
  • 352 Download
  • 24 Web of Science
  • 28 Crossref
AbstractAbstract PDF
Background
Recent findings in molecular pathology suggest that genetic translocation and/oroverexpression of oncoproteins is important in salivary gland tumorigenesis and diagnosis. Weinvestigated PLAG1, SOX10, and Myb protein expression in various salivary gland neoplasm tissues.
Methods
A total of 113 cases of surgically resected salivary gland neoplasms at the NationalCancer Center from January 2007 to March 2017 were identified. Immunohistochemical stainingof PLAG1, SOX10, and Myb in tissue samples was performed using tissue microarrays.
Results
Among the 113 cases, 82 (72.6%) were benign and 31 (27.4%) were malignant. PLAG1 showednuclear staining and normal parotid gland was not stained. Among 48 cases of pleomorphicadenoma, 29 (60.4%) were positive for PLAG1. All other benign and malignant salivary glandneoplasms were PLAG1-negative. SOX10 showed nuclear staining. In normal salivary gland tissuesSOX10 was expressed in cells of acinus and intercalated ducts. In benign tumors, SOX10 expressionwas observed in all pleomorphic adenoma (48/48), and basal cell adenoma (3/3), but not inother benign tumors. SOX10 positivity was observed in nine of 31 (29.0%) malignant tumors.Myb showed nuclear staining but was not detected in normal parotid glands. Four of 31 (12.9%)malignant tumors showed Myb positivity: three adenoid cystic carcinomas (AdCC) and onemyoepithelial carcinoma with focal AdCC-like histology.
Conclusions
PLAG1 expression is specificto pleomorphic adenoma. SOX10 expression is helpful to rule out excretory duct origin tumor,but its diagnostic value is relatively low. Myb is useful for diagnosing AdCC when histology isunclear in the surgical specimen.

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Case Study
Squamous Metaplasia in Pleomorphic Adenoma: A Diagnostic and Prognostic Enigma
Swati Sharma, Monica Mehendiratta, Nivedita Chaudhary, Vineet Gupta, Maulshree Kohli, Anjana Arora
J Pathol Transl Med. 2018;52(6):411-415.   Published online October 1, 2018
DOI: https://doi.org/10.4132/jptm.2018.07.15
  • 6,722 View
  • 133 Download
  • 7 Web of Science
  • 14 Crossref
AbstractAbstract PDF
Pleomorphic adenoma (PA) is the most common benign salivary gland tumor. Histologically, squamous metaplasia has been reported in PA, but has rarely been documented as being extensive enough to cause significant misdiagnosis. Here, we present an unusual case of PA in a 50-year-old female patient presenting with swelling on the postero-lateral aspect of the palate for a week. Histopathologically, the tumor exhibited the features of conventional PA with extensive squamous metaplasia and giant keratotic lamellae in cyst-like areas. Such exuberant squamous metaplasia and keratin can be a diagnostic and prognostic pitfall and lead to overtreatment of the patient.

Citations

Citations to this article as recorded by  
  • Effective Management of a Giant Deforming Pleomorphic Adenoma With Airway Displacement in a 93-Year-Old Patient: A Case Report
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  • ECTOPIC PLEOMORPHIC ADENOMA OF BUCCAL SPACE: CASE REPORT WITH REVIEW OF LITERATURE
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Brief Case Report
Cytokeratin-Positive Gastrointestinal Stromal Tumor of Biphasic Morphology: A Case Report
Sung Sun Kim, Yoo Duk Choi, Jae Hyuk Lee, Chan Choi
Korean J Pathol. 2014;48(5):375-378.   Published online October 27, 2014
DOI: https://doi.org/10.4132/KoreanJPathol.2014.48.5.375
  • 6,877 View
  • 38 Download
  • 2 Crossref
PDF

Citations

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  • CYTOKERATINS: NOT AN EPITHELIAL ENTITY ANYMORE?
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Original Article
Diagnostic Features of Fine Needle Aspiration Cytology of Pleomorphic Adenoma, Adenoid Cystic Carcinoma, and Mucoepidermoid Carcinoma of Salivary Gland.
Eun Sook Nam, Won Bo Jo, Jung Ho Han, Insun Kim
Korean J Cytopathol. 1990;1(1):60-67.
  • 6,123 View
  • 293 Download
AbstractAbstract PDF
The evaluate the diagnostic findings of salivary gland tumors, we reexamined aspiration cytology smears of 7 cases of pleomorphic adenoma, 3 cases of adenoid cystic carcinoma, and 3 cases of mucoepidermoid carcinoma, performed during April 1986 to March 1990, which were comfurmed by surgical excision and histologic diagnosis. The results obtained are summarized as follows : 1. All cases of pleomorphic adenoma showed branching cellular clusters of epithelial and myoepithelial cells. Acellular elements including myxomatous and chondroid components were observed. There were no cellular pleomorphism and nucleoli. Keratinizing squamous epithelial cells and keratin pearls were noted. 2. The smears of adenoid cystic carcinoma showed cell balls or cell cords containing a central hyaline core. Nuclear atypism and the nucleoli were frequently observed. There were no keratinizing squamous epithelial cells. 3. The smears of mucoepidermoid carcinoma showed mainly sheets or clusters of intermediate cells and some mucin-producing cells. Some nuclear pleomorphism was observed. Mucinous material and many inflammatory cells were present in the background.
Case Report
Cytologic Features of Epithelial-Myoepithelial Carcinoma Occurring in External Auditory Canal: A Case Report.
Ho chang Lee, Hyung Geun Song, Young Seok Choi, Ok Jun Lee
Korean J Cytopathol. 2008;19(1):52-56.
DOI: https://doi.org/10.3338/kjc.2008.19.1.52
  • 2,202 View
  • 16 Download
AbstractAbstract PDF
Epithelial-myoepithelial carcinoma (EMC) is a low grade malignant neoplasm that commonly occurs in the parotid gland. Recently, we investigated a case of EMC that occurred in the external auditory canal (EAC) in a 35-year-old male, and this tumor was initially diagnosed as pleomorphic adenoma. The difficulty associated with diagnosing EMC by aspiration cytology arises from both the rare incidence of this tumor and the overlapping spectrum of cytological features found in various salivary gland tumors, such as pleomorphic adenoma, basal cell adenoma and adenoid cystic carcinoma.
Original Article
p53 Expression in the Head and Neck Tumor.
Chae Hong Suh, Mi Sook Lee, Sin Eui Yoon
Korean J Pathol. 1996;30(7):576-586.
  • 1,538 View
  • 21 Download
AbstractAbstract PDF
Mutations in the p53 gene seem to be the most common genetic changes in human malignancies. Mutation or altered p53 expression is a common occurrence in many solid neoplasms, including head and neck carcinomas. Recent studies have also shown p53 alterations in several premalignant conditions of the colon, esophagus, lung, and brain. Preliminary data have suggested that p53 mutations may be involved in tumor progression. This study was performed to determine the incidence of p53 mutations in histologically 27 squamous cell carcinomas, 19 basal cell carcinomas, 18 Schneiderian papillomas, 3 Schneiderian papillomas with malignant transformation, and 15 pleomorphic adenomas of the head and neck region. The degree of p53 gene overexpression was also evaluated according to differentiation, histologic type of tumor, and tumor progression in the head and neck carcinomas. The results were as follows; 1) Eighteen of 27 squamous cell carcinomas, and 4 of 27 dysplasias adjacent to the squamous cell carcinoma of the head and neck expressed p53 protein, but none of the normal control specimens expressed detectable p53 protein. There was no relationship between differentiation of squamous cell carcinoma and p53 protein expression. 2) Twelve of 19 basal cell carcinomas expressed p53 protein; the adenoid type especially overexpressed p53 protein. 3) Nine of 15 pleomorphic adenomas expressed p53 protein especially in the epithelial components. 4) Thirteen of 18 Schneiderian papillomas and all Schneiderian papillomas with malignant transformation expressed p53 protein. The above results indicate that the p53 protein expression is a useful tool for the prediction of tumor progression in the head and neck tumor, but there was no relationship between the differentiation of the tumor and p53 protein expression.
Case Reports
Malignant Myoepithelioma Arising in a Recurrent Pleomorphic Adenoma: A case report.
Jeong Yu Kyung, Yeon Lim Suh
Korean J Pathol. 1999;33(7):517-520.
  • 1,590 View
  • 13 Download
AbstractAbstract PDF
Malignant myoepithelioma is a rare neoplasm of salivary gland which may either arise de novo or develop in a pre-existing pleomorphic adenoma. The malignant myoepithelioma occurs in 0.45% of major salivary gland tumors. Malignant myoepitheliomas arising in the pleomorphic adenoma number less than 20 in English literature and 1 in Korea. We describe a case of malignant myoepithelioma arising in a recurrent pleomorphic adenoma of the left parotid gland of a 61-year-old man. The tumor was ill-defined and composed of polygonal or plasmacytoid myoepithelial cells. Infiltration to surrounding tissue, hemorrhage, necrosis, increased mitotic activity and vascular tumor emboli indicated its malignant nature. There were several satellite nodules with histologic features of typical pleomorphic adenoma. Immunohistochemically, tumor cells were reactive for S-100 protein, AE1/AE3, vimentin, smooth muscle actin and glial fibrillary acid protein.
Epithelial-Myoepithelial Carcinoma of the Parotid Gland: Report of a Case Misinterpreted as Pleomorphic denoma on Fine Needle Aspiration Cytology.
Dong Chul Kim, Ahwon Lee, Kyo Young Lee, Cang Suk Kang, Sang In Shim
Korean J Cytopathol. 2002;13(1):42-46.
  • 1,764 View
  • 15 Download
AbstractAbstract PDF
Epithelial-myoepithelial carcinoma(EMC) is a rare, low grade malignant tumor of the salivary glands. The EMC has a distinctive histological appearance comprising ductal structures with an inner epithelial cell component and an outer layer of myoepithelial cells which show plump clear cytoplasm. The cytologic features of the EMC have been rarely described. A correct cytological diagnosis to this rare tumor is difficult with high false negative rate. We report a case of EMC in which fine needle aspiration cytologic findings were misinterpreted as a pleomorphic adenoma.
Original Article
Histopathologic Study of the Myoepithelial Cells in the Pleomorphic Adenoma.
Jong Boum Choi, Sung Churl Lim, Ho Jong Jeon, Chae Hong Suh
Korean J Pathol. 1992;26(2):125-136.
  • 1,649 View
  • 17 Download
AbstractAbstract PDF
A total of 51 cases of pleomorphic adenoma was obtained from the pathologic files of Chosun University hospital from january, 1980 December, 1989. The studies of clinical, microscopic, immunohistochemical, and electronmicroscopic examination showed the following results. The peak age incidence of the pleomorphic adenoma in this study was in the 5th decade and no significant difference in the incidence between male and female patients was observed. The most common histologic type of myoepithelial data showed positive reactions of the myoepithelial cells to cytokeratin and S-100 protein in the solid, myxoid, and chondroid area. The electronmicroscopic examination of the myoepithelial cells revealed myofilaments, dense bodies, desmosomes, and basement membrane suggesting epithelial origin of these cells. In conclusion, the pleomorphic adenoma seems to be derived from epithelial component and the variable stromal change may be derived from modified myoepithelial cells considering the results of immunohistochemical and electron microscopic study.
Case Report
Pleomorphic Adenoma of the Bronchus: A case report.
Eun Sun Park, Mi Kyung Jee, Seok Jin Gang, Byung Kee Kim, Sun Moo Kim
Korean J Pathol. 1989;23(1):136-140.
  • 1,506 View
  • 15 Download
AbstractAbstract PDF
Pleomorphic adenoma presenting as primary lesion of the bronchus is very rarely encountered, and in our knowledge only 6 cases have been reported in the literature of the western world, and no case report has been published in Korea. Currently, we experienced a case of bronchial pleomorphic adenoma occuring in a 38 years old woman. On X-ray examination, hazy density in the right upper lobe and emphysematous change in the right lower lobe were noted. A right pneumonectomy was done under bronchoscopic diagnosis of bronchial adeoma. The pathologic examination of the present case showed a polypoid endobronchial tumor, 1.4 x 1.1 cm (with extraluminal portion, 2.2 x 1.7 cm) in the right upper bronchus. The microscopic examination revealed a pleomorphic adenoma showing same morphology as those found in the salivary gland. This case, therefore, was believed to be a genuine example of bronchial pleomorphic adenoma of salivary gland type. We compared this case with 6 cases in the literature.

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