Uncommon occurrence of pleomorphic adenoma in submandibular salivary gland in old age males: case series

Pleomorphic adenoma is the most common tumor of the benign salivary gland neoplasms, the submandibular gland is the second most common site of PA after the parotid gland. Authors present 3 series case of pleomorphic adenoma in submandibular salivary gland in institution which were admitted in institution within a month interval. Fine needle aspiration cytology (FNAC) of all 3 cases proved to be benign lesion arising from submandibular salivary gland. All 3 cases underwent excision in to and the postoperative period was uneventful. DT removed on 3rd POD and discharged in POD 10. Biopsy report proved to be pleomorphic adenoma in all cases. past studies showed pleomorphic adenoma most commonly occurs in the parotid gland and its occurrence in the submandibular salivary gland is uncommon. Also, age occurrence involves 30s-50s and is more common in females. But all this case was male and occurred in older age group. Early intervention with surgical excision in toto after definite confirmation with FNAC is the treatment of choice in preventing its malignant transformation.

Giant pleomorphic adenoma of submandibular salivary gland: a case report

The most common benign salivary gland tumor is the pleomorphic adenoma (PA). They can attain grotesque proportions and weigh several kilograms. They can cause facial disfigurement and, if untreated, could lead to airway compromise. Authors report a case of a large PA arising from the right submandibular salivary gland in a 48-year-old male. The lesion measured 9cmx8cmx5cm.

DNA Aneuploidy in Malignant Salivary Gland Neoplasms is Independent of USP44 Protein Expression

Chromosomal instability, leading to aneuploidy, is one of the hallmarks of human cancers. USP44 (ubiquitin specific peptidase 44) is an important molecule that plays a regulatory role in the mitotic checkpoint and USP44 loss causes chromosome mis-segregation, aneuploidy and tumorigenesis in vivo. In this study, it was investigated the immunoexpression of USP44 in 28 malignant salivary gland neoplasms and associated the results with DNA ploidy status assessed by image cytometry. USP44 protein was widely expressed in most of the tumor samples and no clear association could be established between its expression and DNA ploidy status or tumor size. On this basis, it may be concluded that the aneuploidy of the salivary gland cancers included in this study was not driven by loss of USP44 protein expression.

Resumo Instabilidade cromossômica acarretando aneuploidia é um dos fatores marcantes de neoplasias malignas humanas. USP44 (peptidase específica de ubiquitina 44) é uma importante molécula que exerce um papel regulador no ciclo celular e sua perda pode acarretar em segregação cromossômica deficiente, aneuploidia e desenvolvimento de tumores in vivo. Neste estudo, investigou-se a expressão imuno-histoquímica da proteína USP44 em 28 neoplasias malignas de glândulas salivares, associando-se os resultados com o estado de ploidia do DNA avaliado por citometria de fluxo. A proteína USP44 apresentou ampla expressão na maioria das amostras avaliadas e não foi observada associação entre a expressão protéica e o estado de ploidia do DNA ou extensão do tumor. Baseando-se nos resultados, concluiu-se que a aneuploidia das neoplasias malignas de glândulas de salivares incluídas neste estudo não foi influenciada pela perda de expressão da proteína USP44.

Carcinoma ex pleomorphic adenoma composed of three malignant components in parotid gland:A case report and literature review / 吉林大学学报(医学版)

Objective:To investigate the diagnosis and prognosis of one patient with carcinoma ex pleomorphic adenoma (CXPA)composed of three malignant components in parotid gland,and to raise the clinicians'awareness of the disease.Methods:A patient was presented to hospital because of the mass in left parotid gland region for more than 30 years and the accompanied pain lasted for one month.After color ultrasonography,the left parotid gland tumor resection was performed. Results: The operation was successful. The postoperative pathological diagnosis results comfirmed as CXPA,it was composed of three malignant components,including non-specific adenocarcinoma,ductal carcinoma,and myoepithelial carcinoma.Conclusion:CXPA composed of three malignant components at the same time are extremely rare.CXPA is difficult to diagnose and its prognosis is poor.The clinicians are supposed to improve the understanding of CXPA.

Carcinoma ex-pleomorphic adenoma derived from recurrent pleomorphic adenoma shows important difference by array CGH compared to recurrent pleomorphic adenoma without malignant transformation / Carcinoma ex-adenoma pleomórfico derivado de adenoma pleomórfico recorrente mostra diferença importante por array CGH em comparação com adenoma pleomórfico recorrente sem transformação maligna

Abstract Introduction: A key step of cancer development is the progressive accumulation of genomic changes resulting in disruption of several biological mechanisms. Carcinoma ex-pleomorphic adenoma (CXPA) is an aggressive neoplasm that arises from a pleomorphic adenoma. CXPA derived from a recurrent PA (RPA) has been rarely reported, and the genomic changes associated with these tumors have not yet been studied. Objective: We analyzed CXPA from RPAs and RPAs without malignant transformation using array-comparative genomic hybridization (array-CGH) to identify somatic copy number alterations and affected genes. Methods: DNA samples extracted from FFPE tumors were submitted to array-CGH investigation, and data was analyzed by Nexus Copy Number Discovery Edition v.7. Results: No somatic copy number alterations were found in RPAs without malignant transformation. As for CXPA from RPA, although genomic profiles were unique for each case, we detected some chromosomal regions that appear to be preferentially affected by copy number alterations. The first case of CXPA-RPA (frankly invasive myoepithelial carcinoma) showed copy number alterations affecting 1p36.33p13, 5p and chromosomes 3 and 8. The second case of CXPA-RPA (frankly invasive epithelial-myoepithelial carcinoma) showed several alterations at chromosomes 3, 8, and 16, with two amplifications at 8p12p11.21 and 12q14.3q21.2. The third case of CXPA-RPA (minimally invasive epithelial-myoepithelial carcinoma) exhibited amplifications at 12q13.3q14.1, 12q14.3, and 12q15. Conclusion: The occurrence of gains at chromosomes 3 and 8 and genomic amplifications at 8p and 12q, mainly those encompassing the HMGA2, MDM2, WIF1, WHSC1L1, LIRG3, CDK4 in CXAP from RPA can be a significant promotional factor in malignant transformation.

Resumo Introdução: Uma etapa fundamental do desenvolvimento do câncer é o acúmulo progressivo de alterações genômicas, resultando na ruptura de vários mecanismos biológicos. Carcinoma ex-adenoma pleomórfico (CXAP) é uma neoplasia agressiva que surge a partir de um adenoma pleomórfico. O CXAP derivado de um AP recorrente (APR) foi raramente relatado e, até o momento, as alterações genômicas associadas a esses tumores não foram estudados. Objetivo: Avaliar as diferenças entre os CXAPs decorrentes de APRs e os APRs sem transformações malignas usando hibridização genômica comparativa em microarrays (array Comparative Genomic Hibridization - aCGH) a fim de identificar alterações no número de cópias somáticas e os genes afetados. Método: Amostras de DNA extraídas de tumores provenientes de tecido emblocado em parafina foram submetidos à investigação com a técnica aCGH, e os dados foram analisados com o Nexus Copy Number Discovery Edition v.7. Resultados: Não observamos alterações no numero de cópias somáticas nos APRs sem transformação maligna. Quanto ao CXAP de APR, embora os perfis genômicos sejam exclusivos para cada caso, detectamos algumas regiões cromossômicas que pareciam ser preferencialmente afetadas por alterações no número de cópias. O primeiro caso de CXAP-APR (carcinoma mioepitelial francamente invasivo) apresentou alterações no numero de cópias afetando 1p36.33p13, 5p e cromossomos 3 e 8. O segundo caso de CXAP-APR (carcinoma epitelialmioepitelial francamente invasivo) apresentou várias alterações nos cromossomos 3, 8 e 16, com duas amplificações em 8p12p11.21 e 12q14.3q21.2. O terceiro caso de CXAP-APR (carcinoma epitelial-mioepitelial minimamente invasivo) apresentou amplificações em 12q13.3q14.1, 12q14.3, e 12q15. Conclusão: A ocorrência de ganhos de cromossomos 3 e 8, e as amplificações genômicas em 8p e 12q, principalmente aquelas que englobam os HMGA2, MDM2, WIF1, WHSC1L1, RG3, CDK4 no CXAP decorrente de APR podem ser fatores promocionais significativos para a transformação maligna.

A Case of Adenosquamous Carcinoma Ex Pleomorphic Adenoma of Submandibular Gland / 대한이비인후과학회지

We experienced a case of carcinoma ex pleomorphic adenoma arising in the submandibular gland of a 47-year-old male patient. The patient underwent submandibular gland ressection and supraomohyoid neck dissection. Histologic examination revealed that the malignant component of carcinoma ex pleomorphic adenoma was adenosquamous carcinoma. The patient refused postoperative radiation therapy and tumor recurred at the neck and lung about 18 month later. Modified radical neck dissection was carried out, and additional postoperative radiotherapy and palliative chemotherapy were initiated. To the best of our knowledge, this is the first case of carcinoma ex pleomorphic adenoma, of which the malignant component is adenosquamous carcinoma in the submandibular gland. Therefore the authors report this rare case with a literature review.

Cellular Proliferation Index between Carcinoma Ex-Pleomorphic Adenoma and Pleomorphic Adenoma

Carcinoma ex pleomorphic adenoma (CXPA) has been considered an interesting model of carcinogenesis, presenting various histological subtypes and invasiveness phase. The objective was to determine the proliferative index of CXPA and comparing to pleomorphic adenoma (PA). Thirty six cases of CXPA (36 PA) and 22 areas of PA in CXPA (residual PA) were studied by Ki-67 expression. All CXPA cases were classified according to invasiveness phase (intracapsular, minimally and frankly invasive) and histopathological subtypes. Data was statistically analyzed by Wilcoxon, Mann-Whitney and Kruskal-Wallis tests. CXPA included 5 intracapsular, 9 minimally invasive and 22 frankly invasive cases. Fifteen cases corresponded to salivary duct carcinoma, 7 to adenocarcinoma NOS, 7 myoepithelial, 5 epithelial-myoepithelial, one case of squamous cell and one case of sarcomatoid carcinoma. The Ki-67 index of PA and residual PA were significantly lower than CXPA. Intracapsular and minimally invasive showed smaller proliferative index than frankly invasive. Considering the subtypes of CXPA, there was not a statistic difference among them. Ki-67 is a useful marker in the differential diagnosis of PA and CXPA, even when in the early invasive phase.

Carcinoma ex adenoma pleomorfo (CXAP) tem sido considerado um interessante modelo de carcinogênese, apresentando vários subtipos histológicos e fases de invasividade. Determinar o índice proliferativo de CXAP e compará-lo ao adenoma pleomorfo (AP). e seis casos de CXAP, 36 AP, e 22 áreas de AP em CXAP (AP residual) foram estudadas através da expressão de Ki-67. Todos os casos de CXAP foram classificados de acordo com a fase de invasividade (intracapsular, minimamente invasivo e francamente invasivo) e de acordo com os diversos subtipos histopatológicos. Os dados foram estatisticamente analisados através dos testes Wilcoxon, Mann-Whitney e Kruskal-Wallis. O grupo de CXAP era formado por 5 intracapsulares, 9 minimamente invasivos e 22 francamente invasivos. Quinze casos corresponderam a carcinoma de ducto salivar, 7 a adenocarcinoma nos, 7 a carcinoma mioepitelial, 5 a carcinoma epitelial-mioepitelial, 1 a carcinoma epidermoide e 1 a carcinoma sarcomatóide. Os índices de Ki-67 de AP e AP residual foram significativamente menores que o encontrado em CXAP. Os casos intracapsulares e minimamente invasivos mostraram índices proliferativos menores que os francamente invasivos. Considerando os subtipos histológicos de CXAP, não houve diferença estatística entre eles. Ki-67 é um marcador útil no diagnóstico diferencial de AP e CXAP, mesmo quando o carcinoma está em fase precoce de invasividade.

A Case of Carcinoma Ex Pleomorphic Adenoma at 1st Operation

PURPOSE: To report a case of carcinoma ex pleomorphic adenoma observed during the patient's first operation. CASE SUMMARY: A 63-year-old female presented with proptosis and ptosis that was aggravated 1 year prior. On preoperative CT image, a 32 x 20 x 21 mm-sized well demarcated mass (suspected as pleomorphic adenoma) was observed and was removed entirely by anterolateral orbitotomy. The excised mass surface was uneven but the capsule appeared intact on gross examination. Hard, yellow-colored and soft, dark-colored materials were found concurrently on cross section. The histological examination showed malignant cells as part of the soft material and was diagnosed as carcinoma ex pleomorphic adenoma. CONCLUSIONS: We report a case of carcinoma ex pleomorphic adenoma of the lacrimal gland that presented with malignant change during the patient's first operation. Supposedly, during the process of mass growth, minimal rupture occurred causing malignant transformation. Clinically, although a mass is believed benign based on imaging, the possibility of malignant transformation of a tumor increasing rapidly or enlargement causing development of rapid proptosis should be considered.

Mucoepidermoid carcinoma ex pleomorphic adenoma of the lacrimal gland: A rare presentation.

Carcinoma ex pleomorphic adenoma in lacrimal gland is a rare entity unlike its salivary gland counterpart. This rare tumor poses a diagnostic challenge to clinicians as pre‑operative diagnosis is difficult and diagnosis is only by careful pathological assessment. We report this uncommon lesion in a 62‑year‑old lady, wherein the malignant component was mucoepidermoid carcinoma. The elderly patient remained clinically and radiologically free of the tumor for two years after complete excision of the tumor but computed tomography at the end of two and a half years showed a recurrent lesion in the region of the lacrimal gland. This makes long term follow up of patients with these rare lacrimal tumors imperative with a minimum period of at least five years.

Carcinoma ex Pleomorphic Adenoma of the Salivary Glands: Distinct Clinicopathologic Features and Immunoprofiles Between Subgroups According to Cellular Differentiation

In carcinoma ex pleomorphic adenoma (CXPA), pleomorphic adenoma (PA) and diverse carcinoma components showing luminal (ductal) or non-luminal (myoepithelial) differentiation coexist. To elucidate the clinicopathological implications of cellular differentiation in CXPA and the potential role of p53, vascular endothelial growth factor (VEGF), c-erbB-2, c-kit, and glucose transporter 1 (Glut-1) in carcinogenesis, we analyzed 11 CXPAs with luminal differentiation (CXPAs-LD) and 6 CXPAs with non-luminal differentiation (CXPAs-NLD) and compared protein expressions in residual PAs and carcinomas by immunohistochemistry. Among the CXPAs-LD, 5 were invasive and 8 were histologically high-grade tumors. The 5-year survival rate was 72.7%. P53, c-erbB-2, VEGF, and Glut-1 were more immunoreactive in carcinoma components than in PAs (P = 0.008, 0.004, 0.002, and 0.024, respectively); c-erbB-2 overexpression was associated with high histological grade (P = 0.024). Carcinoma components frequently lacked c-kit expression (P = 0.009). CXPAs-NLD were all low-grade and invasive with a larger mean tumor size (5.2 cm) than CXPAs-LD (3.3 cm) (P = 0.040). The patients remained disease-free without significant immunohistochemical expression. The immunoprofiles and clinical course of CXPA differed according to cellular differentiation. Therefore, it is important to report the histological subtype and to assess potential biomarkers in diagnostic and therapeutic trials.

Carcinoma ex pleomorphic adenoma of the parotid gland: Case report

Carcinoma ex pleomorphic adenoma is transformed at the incidence of 1-20% in pleomorphic adenoma and frequently recurred. It accounts for 10% of all malignant salivary tumors and its average age of occurrence is 60s. It will present in a large, long-standing pleomorphic adenoma or in one that was previously treated but has recurred. According to cell composition in malignant cell carcinoma, and clear cell adenocarcinoma. Most (75%) occur in parotid gland, while about 20% occur in the minor gland of the oral mucosa. The metastasis rate to regional lymph node is about 25%, and to distant organs about 33% and the 5-year survival rates are 40%. Though the treatment of the carcinoma ex pleomorphic adenoma is not established, it is treated ideally with and extensive resection, neck dissection, postoperative radiotherapy, and chemotherapy. When occurred in parotid gland, facial paralysis is reported. With a review of literatures, we report a case of carcinoma ex pleomorphic adenoma which operated with total parotidectomy and supraomohyoid neck dissection.

Carcinoma Ex Pleomorphic Adenoma Including Malignant Component of Adenoid Cystic Carcinoma in the Parotid Gland / 대한이비인후과학회지

Carcinoma ex pleomorphic adenoma is rare, aggressive, poorly understood malignancy that occurs in the salivary glands. In carcinoma ex pleomorphic adenoma, an epithelial malignancy develops in association with primary or recurrent benign pleomorphic adenoma. The patient was a 37-year-old female with slow-growing preauricular mass that has been present for 2 years. She visited the hospital because of sudden increase of mass size. The initial cytologic finding by fine needle aspiration biopsy showed what was probably a benign tumor and the radiologic finding revealed a 2.0x1.9 cm sized mass without cervical lymphadenopathy. After right superficial parotidectomy, the histologic examination revealed that tumor was composed of epithelial and mesenchymal component of pleomorphic adenoma and cribriform areas mimicking adenoid cystic carcinoma. Additionally, we did right near-total parotidectomy and postoperative radiotheraphy for 6 weeks. We present a rare case of carcinoma ex pleomorphic adenoma including adenoid cystic component in the parotid gland.

A Case of Carcinoma Ex Pleomorphic Adenoma in the Maxillary Sinus

Carcinoma ex pleomorphic adenoma is one of the rare malignant tumors arising in the salivary glands. Parotid gland is the most frequently affected site, while the other minor salivary glands have much lower incidence rates. There has been no clinical report of the carcinoma ex pleomorphic adenoma developed in the maxillary sinus. Carcinoma ex pleomorphic adenoma is very malignant and its prognosis is rarest among those of parotid gland tumors. We experienced a 24-year-old male patient who had a bulging mass on his left infraorbital area for 18 months. A carcinoma ex pleomorphic adenoma arising in the left maxillary sinus was diagnosed and medial maxillectomy, postoperative chemotherapy and concurrent radiotherapy were done. So far we have followed up the patient for 15 months and there is no sign or symptom of recurrence or metastasis.

A CASE OF CARCINOMA EX PLEOMORPHIC ADENOMA OF PALATE

Carcinoma ex pleomorphic adenoma is a very rare malignant tumor arising in the salivary gland and mainly in the parotid gland. It is highly malignant but asymptomatic, painless and has space occupying lesion sometimes. The metastasis is common(70%) and the prognosis is relatively poor. Although there is no definite treatment modality, the combined surgical intervention and the postoperative radiation therapy has been recommended. We treated a case of carcinoma ex pleomorphic adenoma of the palate with local excision, skin graft and postoperative radiation therapy