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Pleomorphic adenoma of the lachrymal gland: presentation of a clinical case of relapse.

Pleomorphic adenoma affecting the lachrymal gland requires a well-grounded clinical and therapeutic protocol to avoid the risk of malignant transformation or disease recurrence, which is quite dangerous at this site. The authors present a clinical case of pleomorphic adenoma recurrence affecting the right lachrymal gland in a patient who first underwent a biopsy, with subsequent resection of the neoplasm. The authors also review the clinical and radiological features enabling a differential diagnosis and describe the radical "exenteratio orbitae" surgery for pleomorphic adenoma recurrence 7 years after the first operation, which was performed because of total derangement of the orbital cavity. The patient remains disease-free 5 years after this surgical treatment. Therefore, it turns out that an incisional biopsy carries a higher biological cost for the patient.

Adenoma, Pleomorphic↗

Ossifying parotid carcinoma ex pleomorphic adenoma: CT findings.

The CT appearance of a large necrotic carcinoma ex pleomorphic adenoma of the parotid gland is described. The tumour contained trabecular bone, which was correctly identified on a preoperative CT examination and which indicated the origin of the tumour to be a benign pleomorphic adenoma. This degree of macroscopic bone formation is rare in pleomorphic adenoma, and its demonstration by CT has not been previously reported.

Adenoma, Pleomorphic↗

Role of radiotherapy for pleomorphic adenoma.

The purpose of this article was to evaluate the role of radiotherapy (RT) in the treatment of pleomorphic adenoma. This is a retrospective analysis of 17 patients treated with RT alone (2 patients) or combined with surgery (15 patients). Follow up ranged from 1.8 to 27.1 years (median, 9.6 years). Local control was obtained in 8 of 10 patients (80%) with subclinical disease and 3 of 7 patients (43%) with gross disease. Overall local control was obtained in 11 of 17 patients (65%). The 5- and 10-year overall local control rates were 69% and 61%, respectively. One patient died secondary to tumor extension into the central nervous system, 1 patient had recurrence of carcinoma ex-pleomorphic adenoma and died of cancer at the primary site, 5 patients died free of disease, and 10 patients were alive at last follow up. RT is relatively effective at controlling subclinical disease in patients at high risk for developing recurrence after surgery for pleomorphic adenoma. Limited data suggest that RT may also occasionally control gross disease.

Adenoma, Pleomorphic↗

Mucoepidermoid carcinoma arising from pleomorphic adenoma of the soft palate.

Carcinoma arising in pleomorphic adenoma is a rare entity. A case of mucoepidermoid carcinoma in pleomorphic adenoma occurring in the soft palate of a 40-year-old woman is reported. An intraoral excision of the tumor was performed. Histopathological examination revealed high-grade mucoepidermoid carcinoma proliferated in pleomorphic adenoma with free surgical margin. The patient received adjuvant neck radiotherapy. She remains free of disease 16 months post-treatment.

Adenoma, Pleomorphic↗

Ultrastructural similarities of adenoid cystic carcinoma and pleomorphic adenoma.

Ultrastructural examination of five adenoid cystic carcinomas, three breast and two salivary gland, reveals identical patterns of tumour cell differentiation, organization and distribution of cellular products (Zaloudek, Oertel & Orenstein 1984). In both sites, there is proliferation of two populations of cells, one with characteristics and organization of duct-type luminal epithelial cells and a second that forms the principal proliferating component and has the overall organization and appearance that would suggest that they represent modified myoepithelial cells. Recent ultrastructural studies also indicate that tumour cell types and histological organization similar to those described for adenoid cystic carcinoma occur during histodifferentiation of salivary gland pleomorphic adenoma (Dardick et al. 1983a, b). The characteristic histological pattern of adenoid cystic carcinoma is dependent on the formation of pseudolumina containing proteoglycans and reduplicated basal lamina. Similar, but smaller, lumina of like organization and contents are evident in some cases of pleomorphic adenoma. Both the ultrastructural similarities of the tumour cell types and their organization, in adenoid cystic carcinoma and pleomorphic adenoma, suggest that these tumours have a similar histogenetic basis. The fact that one lesion is malignant and the other benign does not preclude common types of tumour cells and developmental processes.

Adenoma, Pleomorphic↗

In situ adenocarcinoma ex pleomorphic adenoma of the lacrimal gland.

A case of an in situ adenocarcinoma of the lacrimal gland that developed in a recurring pleomorphic adenoma is presented. Six years after incomplete resection of a pleomorphic adenoma, the patient complained of a progressive swelling of her right upper eyelid without pain or diplopia. An orbital computerized tomography scan showed an inhomogeneous mass in the right lacrimal gland region without bone involvement. After local excision of the in situ adenocarcinoma ex pleomorphic adenoma, the tumour has been in control for over 6 years.

Adenoma, Pleomorphic↗

Basal cell carcinoma occurring in scar tissue following excision of a parotid gland pleomorphic adenoma.

BACKGROUND: A 49-year-old patient presented with a basal cell carcinoma growing on scar tissue left following excision of a parotid gland pleomorphic adenoma. OBJECTIVE: The objective was to determine whether the basal cell carcinoma had resulted from tumor spillage at the time of the pleomorphic adenoma excision. METHODS: Substantial evidence is presented linking the two tumors. RESULTS: The basal cell carcinoma most likely occurred as the result of tumor spillage at the time of the parotid gland pleomorphic adenoma excision. CONCLUSIONS: Cutaneous connective tissue stroma plays a significant role in determining the differentiation of basal cell carcinomas.

Adenoma, Pleomorphic↗

Nucleolar organizing regions (NORs) in pleomorphic adenomas of the salivary glands.

The number of nucleolar organizing regions (NORs) can be used to reveal the degree of cell activity or metabolism in histopathology specimens. Forty-one cases of pleomorphic adenomas of the salivary glands were studied with a silver nitrate colloid staining method to demonstrate the NORs in the cells of the solid and duct pattern areas and in chondroid areas. Acinic and intercalated ducts cells of the surrounding normal salivary glands were also examined. The data was analyzed statistically and it was found that the cells in the solid/ductal areas had a higher cellular activity than the chondroid cells. This difference is probably directly related with the possibility of malignant transformation and the incidence of the different types of malignant pleomorphic adenomas. Furthermore, it is concluded that the number of NORs in pleomorphic adenomas to define malignancy should be done discriminating between the different histologic patterns because of their different metabolic activities.

Adenoma, Pleomorphic↗

Synthesis of elastin by pleomorphic adenomas.

Previous morphologic and histochemical studies have documented extracellular elastin and elastic fibers within the matrix of pleomorphic adenomas of the salivary glands. By morphologic criteria, the elastin appeared to be synthesized by the tumor rather than being a consequence of stroma produced in response to the tumor cells. To examine this issue, nine pleomorphic adenomas were studied by immunohistochemistry and in situ hybridization with tropoelastin-specific antibodies and complementary DNAs. Corroborating the previous morphologic studies, immunohistochemistry demonstrated abundant extracellular elastin within the matrix of pleomorphic adenomas. Additionally, both immunohistochemistry and in situ hybridization demonstrated continued synthesis of tropoelastin by the neoplastic cells. Tropoelastin production was seen in neoplastic cells with all morphologies.

Adenoma, Pleomorphic↗

Quantitation and localization of cycling tumor cells in pleomorphic adenomas and myoepitheliomas: an immunohistochemical analysis.

Previous investigations have suggested that in certain salivary gland tumors only nonluminal (myoepithelial-like) tumor cells proliferate and that this may have histogenetic implications; however, no quantitative assessment of the cycling component is available. We decided, therefore, to enumerate the cycling luminal and nonluminal cells in 15 pleomorphic adenomas and six myoepitheliomas by using an antibody to proliferating cell nuclear antigen. The mean percentages of cycling luminal and nonluminal cells in pleomorphic adenomas were 2.3 +/- 1.4 and 2.4 +/- 2.3, respectively, and 2.1 +/- 0.7 of the tumor cells in myoepitheliomas. Even though duct-like structures in pleomorphic adenomas are considerably separated by more numerous nonluminal cells and, therefore, cycling cells may seem fewer, both cell types proliferate at similar rates. The results indicate that nonluminal cells are not the sole proliferative component in salivary gland tumors.

3,3'-Diaminobenzidine↗

Cytokeratins, smooth muscle actin and vimentin in human normal salivary gland and pleomorphic adenomas. Immunohistochemical studies with particular reference to myoepithelial and basal cells.

The distribution of immunostaining in normal major salivary gland and in 12 pleomorphic adenomas was studied using monospecific monoclonal antibodies to a number of cytokeratins, including cytokeratin 14, to smooth muscle actin and vimentin. A number of these antibodies enabled a distinction to be made between structural components of the normal gland, and to relate this to the different structures of pleomorphic adenomas. In the normal gland, the luminal duct cells expressed cytokeratins 7, 8, 18 and 19. Three antibodies were of particular value for the characterization of normal myoepithelial and basal cells; while the antibody to smooth muscle actin and the cytokeratin antibody Ks8.12 mutually exclusively stained the myoepithelial (basket) cells and the basal duct (light) cells, respectively, the recently established monospecific antibodies to cytokeratin 14 showed specific immunostaining with both cell types. These three antibodies left luminal cells virtually unstained. Ck 13 was found occasionally in single luminal excretory duct cells. Antibodies to cytokeratins 1/2, 10 and 10/11 did not show any staining in the normal gland. In the pleomorphic adenomas, the staining pattern of the two-layered tubular formation resembled that of the normal gland ducts: tumour luminal cells showed the characteristic, although more irregular, expression of cytokeratins 7, 8, 18 and 19; the outer cells resembled normal ductal basal cells with their anti-cytokeratin 14/Ks8.12-epitope staining and in that they virtually lacked staining for smooth muscle actin. Trabecular formations and cells in myxoid areas were reactive with Ks8.12 and for cytokeratin 14, occasionally also for cytokeratins 7, 18 and 19. Epidermoid cell islets expressed mainly cytokeratin 14 and inconsistently the squamous epithelial cytokeratin 13 and the epidermal cytokeratin 10/11. Vimentin was found in cells of myxoid areas. The results support the postulate that some of the normal duct basal cells act as reserve cells and can give rise to tumour formation with a primitive myxoid or trabecular pattern and a more differentiated tubular or epidermoid configuration.

Actins↗

Myoepithelial carcinoma (malignant myoepithelioma) of the parotid gland arising in a pleomorphic adenoma.

A myoepithelial carcinoma, a rare malignant salivary gland neoplasm, arose in a pleomorphic adenoma of the parotid gland. The initial tumour was a pleomorphic adenoma with epithelial and myoepithelial elements. Subsequently the tumour recurred twice and was characterised by invasion of the mandible. Histological examination of the second recurrence showed a malignant spindle cell neoplasm with an infiltrative growth pattern and a high mitotic rate. There was involvement of local lymph nodes. The immunophenotype was characteristic of myoepithelial differentiation: tumour cells stained positively with anticytokeratin antibodies, S-100 protein, alpha smooth muscle actin, and vimentin. Electron microscopy confirmed myoepithelial differentiation, with small foci of keratinocytic phenotype. Large numbers of tumour cell nuclei were reactive with the anti-p53 antibody, DO-7, in contrast to the two previous resections. Thus malignant transformation of a pleomorphic adenoma may involve myoepithelial as well as epithelial elements. Accumulation of p53 protein, perhaps through mutational events, may have played a role in this malignant transformation.

Adenoma, Pleomorphic↗

Myoepithelial carcinoma arising in a pleomorphic adenoma of the parotid gland: report of a case with cytopathologic findings.

BACKGROUND: Carcinoma arising in a mixed tumor, or carcinoma ex pleomorphic adenoma (CEPA), is an uncommon primary salivary gland neoplasm. Among the various types of carcinomas that can be seen histologically in a CEPA, myoepithelial carcinoma is one of the rarest forms. CASE: A 76-year-old woman presented with an incidental parotid/parapharyngeal mass. Computed tomography-guide fine needle aspiration (FNA) showed a biphasic neoplasm with epithelial and stromal components consistent with pleomorphic adenoma (PA). However, in addition, a distinct population of discohesive atypical and pleomorphic cells with high nuclear/cytoplasmic ratio was noted in the background. In the cytopathologic diagnosis a suspicion was raised about a possible CEPA. Subsequent resection of the parotid mass confirmed the presence of low grade myoepithelial carcinoma arising in a PA. CONCLUSION: Although uncommon, CEPA should be suspected on FNA when atypical cytomorphologic characteristics are observed. In rare cases a myoepithelial carcinoma also arises in a preexisting PA, necessitating an accurate interpretation for more definitive therapy.

Adenoma, Pleomorphic↗

Cytomorphologic spectrum of carcinoma ex pleomorphic adenoma.

OBJECTIVE: To delineate the cytomorphologic features of carcinoma ex pleomorphic adenoma (CPA) and identify the diagnostic pitfalls. STUDY DESIGN: Smears of 14 cases suspected as CPA on fine needle aspiration over a period of 15 years were reviewed. Cytohistologic correlation was done in 10 cases. RESULTS: All cases had a salivary gland mass of 1-16 years' duration, with a rapid increase in size in 10 cases. Epithelial cells predominated over stroma in 11 of 14 cases. Group I showed unequivocal malignant cells admixed with benign epithelial and stromal components of pleomorphic adenoma (PA), which were considered diagnostic of CPA on review. The cytologic differential diagnosis in these cases included mucoepidermoid carcinoma, carcinosarcoma and metastatic adenocarcinoma. Group II comprised 7 cases suspected to be cellular PA with atypia or CPA. These showed mild to moderate degrees of pleomorphism, absence of unequivocal malignant cells, and a variable proportion of benign epithelial and stromal components. Four of them were histologically confirmed as CPA. CONCLUSION: Sampling error is an important cause of diagnostic pitfalls. Correlation with clinical data is essential in diagnosis of CPA on cytology. In a proper clinical setting, extensive fine needle aspiration sampling should be done initially. Any degree of nuclear atypia in PA should be documented, alerting the clinician and histopathologist to the possibility of CPA.

Adenocarcinoma↗

Fine needle aspiration diagnosis of malignant mixed tumor (carcinosarcoma) arising in pleomorphic adenoma of the salivary gland. A case report.

BACKGROUND: True malignant mixed tumor (carcinosarcoma), composed of carcinoma and sarcoma components, is rare in salivary gland neoplasms. Even rarer is a true malignant mixed tumor arising in the pleomorphic adenoma of the salivary gland. CASE: A 64-year-old male was admitted with left pharyngeal pain. Head and neck magnetic resonance imaging revealed a left parapharyngeal mass; fine needle aspiration (FNA) cytology through the oral cavity was performed. CONCLUSION: The aspirate presented a mixture of large, pleomorphic, vacuolated, single or multinucleated cells in a mucoid matrix and clusters of adenocarcinoma cells. Additionally, occasional benign glandular cells were noted. A cytologic diagnosis of malignant mixed tumor arising in the pleomorphic adenoma of the deep lobe of the parotid gland was made and confirmed by the surgically resected specimen.

Adenoma, Pleomorphic↗

Immunohistochemical localization of carcinoembryonic antigen in carcinoma in pleomorphic adenoma of salivary gland: use in the diagnosis of benign and malignant lesions.

The localization of carcinoembryonic antigen (CEA) and lysozyme (LZM) was immunohistochemically studied in 34 carcinomas arising in benign pleomorphic adenomas and 25 normal salivary glands in order to assess its potential diagnostic value. CEA in the normal salivary gland was located in luminal cell membranes of intercalated duct cells and serous acinar cells. Strongly positive cell surface and intraluminal staining of CEA appeared in the areas of gland-forming pattern in pleomorphic adenoma. CEA activity was detected in 7/9 cases (78%) of adenocarcinoma, 10/11 cases (91%) of epidermoid carcinoma, 3/8 cases (38%) of anaplastic carcinoma, 5/5 cases (100%) of mucoepidermoid carcinoma, and 1/1 case (100%) of adenoid cystic carcinoma. CEA was always present in the cytoplasm of epithelial cells and luminal contents of neoplastic glands. CEA in epidermoid carcinoma may occasionally react strongly in the cytoplasm. Lysozyme-immunoreactivity was detected in the cytoplasm of intercalated duct cells and serous acinar cells of the normal salivary gland but little or no LZM was observed in any of the tumors. These results suggest that the presence of CEA could be a useful marker that provides valuable information for the differential diagnosis between benign and malignant areas of carcinoma in pleomorphic adenoma of the salivary gland. Moreover, LZM could be of valuable use for discriminating neoplastic from non-neoplastic tissue of salivary glands.

Adenoma, Pleomorphic↗

Unusual progression of pleomorphic adenoma of the lacrimal gland: case report.

A 72-year-old female complained of acute pain on left eye movement followed by progressive exophthalmos. Neuroimaging revealed a large well-demarcated lesion consisting of solid and cystic parts, as well as bone destruction and hemorrhage, within the left orbital cavity. The preoperative diagnosis was pleomorphic adenoma with or without malignant transformation, or cavernous angioma. En bloc excision including adjacent tissues was planned to resolve the progressive symptoms and to obtain a histological diagnosis. The transcranial route was chosen since tumor invasion to the cranial base was possible. The histological diagnosis was pleomorphic adenoma. Pathological and preoperative radiological examinations indicated that repeated intratumoral hemorrhage had caused the orbital bone destruction and acute orbital pain. Neoplasms should be differentiated from a wide spectrum of other possible pathologies. Accurate clinical diagnosis of neoplasm in the orbital cavity is important for correct therapeutic management. Malignancy is generally suspected if painful and progressive signs and symptoms are associated with an orbital mass lesion. The present case suggests that pleomorphic adenoma should also be considered in the differential diagnosis. The therapeutic strategy for lacrimal gland tumors remains controversial, so a flexible management approach is required.

Adenoma, Pleomorphic↗

Expression of ras-P21 and ras gene alteration in pleomorphic adenomas.

The expression of ras-P21 protein and gene alteration of the K-ras gene were examined in 12 cases of pleomorphic adenoma of the salivary gland. The tumor cells in all 12 cases were strongly stained for ras-P21 protein by the indirect immunoperoxidase method, suggesting the enhanced expression of ras-P21 protein in pleomorphic adenomas. Analysis of the PCR products for the K-ras gene by temperature-gradient gel electrophoresis (TGGE) revealed that gene alterations such as point mutations occurred in 4 out of 12 pleomorphic adenoma tissues examined.

Adenoma, Pleomorphic↗