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PubMed2006

Pleomorphic adenoma arising in an accessory lacrimal gland of Wolfring.

PURPOSE: To describe a patient with pleomorphic adenoma arising in an accessory lacrimal gland of Wolfring in the lower lid and to illustrate the immunohistochemical and molecular cytogenetics. DESIGN: Single interventional case report. METHODS: A 62-year-old man presented with a 20-year history of a painless slowly growing mass at the temporal part of the right lower eyelid. Histological, immunohistochemical, and fluorescence in situ hybridization studies of the excised tumor were performed. RESULTS: Histological evaluation showed many glandular elements embedded in a myxoid stroma. The tumor was situated beneath an area of a normal accessory lacrimal gland of Wolfring and in close association with normal meibomian glands. Myoepithelial tumor cells in the myxoid stroma reacted strongly with an antibody against glial fibrillary acidic protein, which did not bind to normal lacrimal gland tissue. Tumor cells with both epithelial and myoepithelial morphologies reacted positively for both pleomorphic adenoma gene-1 and high-mobility group A2 proteins. Fluorescence in situ hybridization analysis showed no evidence of clonal translocations or numerical abnormalities involving chromosome 8 or 12. CONCLUSIONS: Pleomorphic adenoma of the accessory lacrimal gland is an exceedingly rare tumor of the ocular adnexa. Glial fibrillary acidic protein seems to be a tumor-associated antigen. Genetically, this case of pleomorphic adenoma arising from an accessory lacrimal gland of Wolfring is identical with those originating from salivary glands.

Adenoma, Pleomorphic↗
PubMed2004

Expression of cyclooxygenase-2, Bcl-2 and Ki-67 in pleomorphic adenoma with special reference to tumor proliferation and apoptosis.

The present report is of immunohistochemical and cell biological studies on cyclooxygenase(COX)-2, Bcl-2 and Ki-67 in pleomorphic adenoma (n = 35). Pleomorphic adenomas from oral minor glands (n = 15), parotid gland (n = 11), Submandibular gland (n = 8) were studied and sublingual gland (n = 1). Expression of COX-2, Bcl-2 and Ki-67 was examined immunohistologically. Immunostaining intensity of COX-2 and Bcl-2 was classified into expression levels from +3 to 0. The positivity to Ki-67 was evaluated by counting the number of positive cells per 1000 cells, and the values were expressed in as percentage. Apoptotic cells were detected using the modified TUNEL method. Expression of COX-2 mRNA was analyzed by real time PCR using fresh tissues of 4 cases. The relationship between morphological findings and the level of COX-2 mRNA expression was analyzed using a laser capture microdissection (LCM). The results of immunohistochemistry and gene analysis using LCM revealed expression of COX-2 mainly in luminar tumor cells. Expression of COX-2 in pleomorphic adenoma was correlated with expression of Bcl-2 statistically (p = 0.044 < 0.05, r = 0.342). There was only a small number of apoptotic cells cells, and intensity of expression of TUNEL was not correlated with the expression of COX-2 (p = 0.463 > 0.05, r = -0.128). There was no statistical correlation between COX-2 and Ki-67 (p = 0.97 > 0.05, r = -0.07). It is suggested that COX-2 may inhibit apoptosis mediating Bcl-2 expression in pleomorphic adenoma, rather than playing a role in cell proliferation.

Adenoma, Pleomorphic↗
PubMed2005

Recurrent pleomorphic adenoma of the parotid gland.

OBJECTIVES: To assess the long-term results in the management of 42 patients with recurrent pleomorphic adenoma of the parotid gland. STUDY DESIGN: A retrospective analysis of 42 patients who underwent parotidectomy for recurrent pleomorphic adenoma was performed to study presenting clinicoradiographic features, surgical technique, facial nerve management, and the long-term risk of recurrence. RESULTS: All 42 patients had multi-focal, nontender recurrent nodules following one to four prior surgical procedures and 6 patients underwent prior radiotherapy. Surgical procedures included subtotal parotidectomy in 12 patients, total parotidectomy in 18 patients, parotidectomy with facial nerve resection in 7 cases, and subtotal petrosectomy with facial nerve resection in 5 individuals. The 2 patients with malignant transformation died of disseminated lung and bone metastasis. Twenty-nine of the remaining 40 patients had no recurrent disease. Seven patients developed local parotid bed or cutaneous recurrent disease, 2 patients died of unrelated causes, and 2 patients were lost to follow-up. CONCLUSIONS: All 7 patients with recurrent disease underwent subtotal parotidectomy with "negative" surgical margins. Total parotidectomy or subtotal petrosectomy with facial nerve resection in selected cases may reduce the risk of multiple episodes of pleomorphic adenoma recurrence. Two of 42 patients were found to have carcinoma ex-pleomorphic adenoma, both of these patients underwent prior radiotherapy, and both died of metastatic disease.

Adenoma, Pleomorphic↗
PubMed2005

Pleomorphic adenoma (benign mixed tumor) of the breast: a case report and review of the literature.

We report a case of pleomorphic adenoma (benign mixed tumor) of the breast, which is an extremely rare location for this tumor. Examination of a 55-year-old woman unexpectedly revealed a mass measuring 0.8 cm in diameter in the subareolar region of the right breast. Excisional biopsy was performed, and the tumor histologically showed pleomorphic adenoma composed of duct epithelial cells, myoepithelial cells, and a myxochondroid matrix. Immunohistochemically, duct epithelial cells were positive for the estrogen receptor, but negative for the progesterone receptor. The nuclei of the spindle and myoepithelial cells were immunoreactive for HMGI-C and HMGI(Y) proteins, indicating a histogenesis similar to pleomorphic adenoma of the salivary glands. Interphase fluorescence in situ hybridization performed on paraffin-embedded tissue sections with 12q15 probes and a 6p21 probe demonstrated no chromosomal rearrangement. Sixty-nine cases of this type of tumor arising in the breast have been described previously. Using imaging procedures, the tumor has occasionally been misdiagnosed as malignant clinically and even pathologically in frozen section diagnosis. Careful diagnosis based on paraffin sections is required to avoid unnecessary aggressive surgery, and pathologists should include pleomorphic adenoma in the differential diagnosis of a demarcated, juxtaareolar, small hard mass.

Adenoma, Pleomorphic↗
PubMed2004

Synchronous occurrence of ipsilateral synovial chondromatosis of the temporomandibular joint and pleomorphic adenoma of the parotid gland.

We present a unique case characterized by true coexistence of synovial chondromatosis (SC) of the temporomandibular joint (TMJ) with a salivary gland tumor, specifically pleomorphic adenoma, of the ipsilateral parotid gland. The possibility that the condition was the result of a single pathologic entity, ie, SC with extracapsular involvement or pleomorphic adenoma affecting the TMJ, received strong consideration. Nonetheless, careful assessment of the clinical, radiographic, MRI, and surgical findings strongly suggested the synchronous occurrence of 2 separate pathologic processes, despite their close proximity. Further, histopathologic examination confirmed the presence of both SC of the TMJ and pleomorphic adenoma of the superficial lobe of the ipsilateral parotid gland. Previous reports have pointed out the possibility of misdiagnosing extracapsular spread of SC as a salivary gland tumor. Vice versa, it has been noted that parotid gland tumors, especially malignant, may cause TMJ symptoms similar to those of SC. However, to the best of our knowledge, this is the first time that true synchronous occurrence of ipsilateral SC and pleomorphic adenoma, or parotid tumor of any kind, is reported.

Adenoma, Pleomorphic↗
PubMed1996

Benign mixed tumor (pleomorphic adenoma) of the lacrimal gland in a 6-year-old boy.

PURPOSE: To report a 6-year-old boy who had benign mixed tumor (pleomorphic adenoma) of the lacrimal gland. METHODS: A 6-year-old boy was referred for evaluation of a lacrimal fossa mass. We performed a lateral orbitotomy and excised the mass. RESULTS: Histopathologic evaluation of the excised orbital mass disclosed a benign mixed tumor (pleomorphic adenoma) of the lacrimal gland. CONCLUSION: Benign mixed tumors (pleomorphic adenomas) of the lacrimal gland are rare in children. However, they must be considered in the differential diagnosis of pediatric orbital masses because of the prognostic importance of removing a pleomorphic adenoma with the capsule intact.

Adenoma, Pleomorphic↗
PubMed1994

Characteristic bright signal of parotid pleomorphic adenomas on T2-weighted MR images with pathological correlation.

The purpose of this study was to correlate the MR features and histological findings of pleomorphic adenomas, which are the most common tumour of the parotid gland, and to distinguish them from other types of tumour in the parotid gland. The materials studied consisted of 20 pleomorphic adenomas, including 12 untreated and eight recurrent cases, 17 other benign parotid tumours, and 12 malignant tumours. T1- and T2-weighted images and Gd-DTPA enhanced T1-weighted images were obtained. Characteristic bright signals with a higher intensity than that of CSF were seen in all of the pleomorphic adenomas on T2-weighted images, and were found to represent myxoid areas histologically. Although the cystic spaces in some benign tumours produced bright signals, these areas were distinguished from myxoid areas due to nonenhancement with Gd-DTPA. No bright signals were seen in the malignant tumours. In conclusion, bright signals on T2-weighted images represent myxoid tissue and are useful findings for diagnosing pleomorphic adenomas.

Adenolymphoma↗
PubMed2005

[Adenocarcinoma in a pleomorphic adenoma of the lacrimal gland: a case study].

PURPOSE: To report a patient with an adenocarcinoma in a pleomorphic adenoma of the lacrimal gland. Adenocarcinoma constitutes a distinct group of epithelial malignancies of the lacrimal gland. METHODS: The clinical presentation, workup, surgical approach, and pathological findings were reviewed. RESULTS: A 56-year-old man presented with a 3-month history of a lacrimal fossa mass. This patient presented a painless mass in the upper outer eyelid with significant displacement of the globe. The tumor was localized to the lacrimal gland. Radiological investigations showed a round, well-defined lesion in the fossa of the lacrimal gland. We performed a lateral orbitotomy; en bloc resection was accomplished. The final specimen showed an adenocarcinoma in a pleomorphic adenoma. No recurrences were detected during follow-up. CONCLUSION: Although adenocarcinoma has rarely been reported in association with a pleomorphic adenoma of the lacrimal gland, this combination can exist. If the malignant tumor is limited in the pleomorphic adenoma, the prognosis appears to be better than in cases of local extension. Adjuvant therapy, radiation or others, are not useful.

Adenocarcinoma↗
PubMed2002

Malignant myoepithelioma arising from recurrent pleomorphic adenoma of minor salivary gland.

Malignant myoepitheliomas of the salivary gland are very rare tumors which may either arise de novo or develop in a pre-existing pleomorphic adenoma. We report a case of malignant myoepithelioma of the minor salivary gland that progressed from benign pleomorphic adenoma. The original pleomorphic adenoma was resected in 1979 (the '79 tumor). The first recurrent tumor was operated in 1995 (the '95 tumor). The '95 tumor was diagnosed as pleomorphic adenoma. Although the myoepithelial tumor component was more prevalent in the '95 tumor, histological features of the first recurrent tumor were similar to the '79 tumor. The second recurrent tumor showed more aggressive clinical features (the '98 tumor). It also showed massive growth of myoepithelial tumor cells with bone invasion, which led to the diagnosis of the '98 tumor as malignant myoepithelioma. With adequate surgical margin, the patient has been free from tumor recurrence for 20 months.

Adenoma, Pleomorphic↗
PubMed2003

Assessment of pleomorphic adenomas using MRI and dynamic contrast enhanced MRI.

We evaluated magnetic resonance images (MRI) and the value of dynamic contrast enhanced MRI (DCE-MRI) of pleomorphic adenomas retrospectively. MRI was performed for 18 pleomorphic adenomas, including 11 cases with DCE-MRI. We obtained the following results on the MRI and DCE-MRI. (a). Pleomorphic adenomas showed a predilection for homogeneous intermediate signal intensity on T1-weighted images (T1WI), heterogeneous high signal intensity on T2-weighted images, and heterogeneous enhancement on Gd-T1WI. (b). Of 11 contrast index (CI) curves of pleomorphic adenomas, nine CI curves (81.8%) increased gradually to 600 s or increased gradually, reached a plateau, and sustained the plateau to 600 s. The remaining two (18.2%) increased gradually and decreased gradually thereafter. (c). CI curves reached the maximum CI index at 135-300 s.

Adenoma, Pleomorphic↗
PubMed1991

Differential diagnosis of pleomorphic adenoma by immunohistochemical means.

Immunohistochemical study of major salivary gland tumours was performed on 60 pleomorphic adenomas, five basal cell adenomas and 10 adenoid cystic carcinomas to determine the diagnostic value of each antigen. Immunoreactivity examined were intermediate filaments (keratin, vimentin, desmin and glial fibrillary acidic protein [GFAP]) and related substances (actin, S-100 protein and secretory component). In pleomorphic adenomas, there was positive immunoreactivity for GFAP which was not observed in normal tissue or other neoplastic tissues. Immunoreactivity of GFAP was closely related to myxomatous and early chondromatous differentiation in pleomorphic adenoma. It is considered that GFAP immunoreactivity should be assessed in the occasional differential diagnostic dilemma of pleomorphic adenoma versus adenoid cystic carcinoma and basal cell adenoma, because of its ability to show potential and definite myxochondromatous differentiation.

Adenoma↗
PubMed1995

Carcinoma ex pleomorphic adenoma of the soft palate.

A case of carcinoma ex pleomorphic adenoma arising in the soft palate is reported. The tumour presented had enlarged gradually over 10 years and finally occupied the oral cavity. The patient was admitted to our hospital due to disturbance of her speech and swallowing, and a sudden haemorrhage from the tumour. The initial pathological diagnosis by open biopsy was benign pleomorphic adenoma. After total resection, histological examination revealed that the tumour was composed partly of benign pleomorphic adenoma and partly of an adenocarcinomatous component. The carcinoma cells with prominent nucleoli were spheroid or polygonal in shape, and frequently formed ductal structures with areas of necrosis. Mitoses were also found. These findings showed that this tumour was a secondary carcinoma which had developed in a preexisting pleomorphic adenoma.

Adenocarcinoma↗
PubMed2001

Immunolocalisation of cartilage-derived retinoic acid-sensitive protein in pleomorphic adenoma of the parotid salivary gland.

Pleomorphic adenoma of the parotid salivary glands often contains chondroid elements and may exhibit cartilaginous and osseous differentiation, although the latter is extremely rare. Twenty-nine pleomorphic adenomas (PAs) of the parotid gland were examined immunohistochemically for the distribution of cartilage-derived retinoic acid-sensitive protein (CD-RAP), a recently described marker of chondrocytes, which may be important in the morphogenesis and development of the salivary gland. In the normal parotid gland, the ductal cells expressed CD-RAP, but not the myoepithelial cells. In the pleomorphic salivary adenomas, the duct-like cells, but not the myoepithelial cells, expressed CD-RAP. Since many authorities consider myoepithelial cells to be the source of the chondroid matrix, it is surprising that these cells do not express the chondrocytic marker, CD-RAP. Putative neoplastic myoepithelium in the pleomorphic adenoma and some cells in the myxochondroid areas expressed S-100 and calponin.

Adenoma, Pleomorphic↗
PubMed2002

Precancerous foci in pleomorphic adenoma of the salivary gland: recognition of focal carcinoma and atypical tumor cells by P53 immunohistochemistry.

BACKGROUND: It is still controversial if atypical tumor cells scattered in salivary pleomorphic adenomas are precancerous and how carcinoma arises in pleomorphic adenomas. METHODS: We studied clinicopathologically the frequency and variation of cellular atypia among tumor cells and examined the expression status of p53 gene products as well as proliferating cell nuclear antigen (PCNA) in 101 surgical materials of pleomorphic adenomas. RESULTS: Histopathologically, atypical tumor cells were found in 51% of the cases examined. Their mode of distribution was classified into three groups: focal (six cases, 6%) which could be identified as focal carcinoma, measuring less than 1 mm in diameter; sporadic (15 cases, 15%) and singular (30 cases, 30%). These atypical cells were located mainly within sheet-like nests of tumor cells but not in chondroid or fibro-hyaline foci. Immunohistochemically, most of the atypical cells were positive for p53 gene products and PCNA. CONCLUSION: The results indicated that atypical cells with p53 protein accumulation in their nuclei could be regarded as cells in a precancerous state not yet forming an apparent carcinomatous nest. Some cell population with these atypical cells are likely to form focal carcinomas and then to an apparent form of carcinoma in pleomorphic adenoma.

Adenocarcinoma↗
PubMed1999

Intravascular tumour in intra-oral pleomorphic adenomas: a diagnostic and therapeutic dilemma.

AIMS: Intravascular tumour has been described very rarely in pleomorphic adenomas. The aim of this study was to establish the frequency of intravascular tumour in pleomorphic adenomas arising in minor salivary glands and to determine the biological significance of this phenomenon. METHODS AND RESULTS: Representative sections of 67 widely excised pleomorphic adenomas were examined for the presence of intravascular tumour. Sixty-two cases were derived from the palate while the remaining five were from the cheeks and lips. In instances where intravascular tumour was identified, multiple serial sections were assessed and immunohistochemical stains were performed. None of these cases showed cytological evidence of malignancy. Solid cords of intravascular tumour were present in six palatal tumours (8.9%) and consisted of plasmacytoid myoepithelial cells permeating muscular walled blood vessels and capillaries both within the tumour and capsule. Immunoperoxidase staining confirmed that the intravascular cells were phenotypically identical to those of the tumour being S100- and smooth muscle actin (SMA) positive. There is some evidence that this phenomenon represents true vascular invasion although artefactual spillage cannot be excluded. CONCLUSIONS: Although the biological significance of intravascular tumour in pleomorphic adenomas of minor salivary glands remains unknown, the occurrence of metastatic disease has not been demonstrated nor have aggressive behaviour or recurrences.

Actins↗
PubMed2007

[Multiple primary pleomorphic adenomas of the unilateral parotid gland].

BACKGROUND: Classical lateral parotidectomy is the standard surgical procedure for the management of lateral lobe parotid pleomorphic adenomas in most clinical centres. Extracapsular dissection and subtotal lateral parotidectomy are alternatively discussed in current literature and preferred by many authors. CASE: We report on a 20-year-old patient with multiple primary pleomorphic adenomas in the unilateral parotid gland, which preoperatively presented as a solitary tumour node. CONCLUSION: Considering this case we focus on the distinctive surgical procedures for the management of parotid pleomorphic adenomas. We work out, why lateral or even total parotidectomy are justified in the treatment of solitary palpable tumours and why we account lateral parotidectomy as the standard surgical procedure for the management of lateral lobe parotid pleomorphic adenomas.

Adenoma, Pleomorphic↗
PubMedDate not supplied

Benign and malignant parotid pleomorphic adenomas: CT and MR studies.

Parotid pleomorphic adenomas are usually benign, solitary, ovoid masses. Occasionally, these tumors may be highly lobulated and clinically mimic multiple lesions. Actual multiple benign mixed tumors are rare as are malignancies that occur in these pleomorphic adenomas. Of these malignancies, the most common type is the carcinoma ex pleomorphic adenoma. Two patients are presented, one of whom had a markedly lobulated benign mixed tumor and the other patient had an ex pleomorphic malignancy associated with multiple benign mixed tumors. Both cases are documented with CT and magnetic resonance studies. The relative merits of these modalities are discussed as they apply to these parotid masses.

Adenoma, Pleomorphic↗
PubMed2003

Adenocarcinoma ex-pleomorphic adenoma of the lacrimal sac and nasolacrimal duct: a case report.

OBJECTIVES: To present the clinical presentation, workup, surgical approach, and pathological findings of the first case report of a patient with adenocarcinoma ex-pleomorphic adenoma of the lacrimal sac and nasolacrimal duct. STUDY DESIGN: Retrospective review of the records of a case of adenocarcinoma ex-pleomorphic adenoma of the lacrimal sac and nasolacrimal duct. METHODS: The clinical presentation, workup, surgical approach, and pathological findings were reviewed. RESULTS: A 51-year-old man presented with a 10-year history of recurrent epiphora of the right eye. At dacryocystorhinostomy a small lesion was visualized within the lumen of the lacrimal sac. A biopsy specimen was consistent with adenocarcinoma. En bloc resection was accomplished using a lateral rhinotomy and medial maxillectomy. The final specimen showed adenocarcinoma ex-pleomorphic adenoma. The patient was given postoperative radiation therapy. He was free of disease 16 months after treatment. CONCLUSIONS: Lacrimal sac tumors should be considered in the differential diagnosis of chronic epiphora. Management of nasolacrimal adenocarcinoma requires complete surgical resection. Radiation treatment in and of itself is not curative but may be useful as adjuvant therapy. Carcinoma ex-pleomorphic adenoma can develop in the lacrimal sac and nasolacrimal duct.

Adenoma, Pleomorphic↗