EZH2 immunoexpression in pleomorphic adenoma and adenoid cystic carcinoma and clinicopathological features

Abstract The aim of this study was to evaluate the expression of the EZH2 protein and describe the clinical and microscopic characteristics of adenoid cystic carcinoma (ACC) and pleomorphic adenoma (PA). The study included 16 ACC cases and 12 PA. All ACC and PA cases were positive for EZH2 and the ACC samples showed significantly higher EZH2 expression. The clinical and microscopic covariates were described in relation to EZH2 staining in ACC samples. The highest mean values of EZH2 were observed in cases with local metastasis, recurrence, perineural invasion, and predominantly cribriform growth pattern without solid areas. EZH2 is a potential marker of malignancy.

Clinical behavior of recurrent pleomorphic adenoma in the palate: a systematic review

Purpose: This systematic review analyzed the clinical behavior and odds of malignancy of the palatal recurrent pleomorphic adenomas. Methods: Systematic review of patients with recurrent pleomorphic adenoma arising in the palate. Database search: MEDLINE, Scopus, Web of Science, Cochrane, EMBASE, Virtual Health Library, Google Scholar, and OpenGrey. A binomial logistic regression was performed to assess the odds of detecting recurrence five, 10 and 20 years after the treatment of primary tumor. Results: Thirteen studies (n = 18 patients) out of 336 were included. The recurrent pleomorphic adenoma in palate was more common in females (61.6%), average age was 49 years old (range 9-73 years old). Four patients progressed to malignant transformation. The odds ratio (OR) of detecting a recurrence at 10 (OR = 5.57; 95% confidence interval - 95%CI 1.13-27.52), and 20 years (OR = 18.78; 95%CI 3.18-110.84) after treatment of primary pleomorphic adenoma was significantly higher than at one-year follow-up. Conclusions: The recurrence of pleomorphic adenoma in palate remains a rare event of late occurrence. It mainly affects middle-aged female and carries a risk of malignant transformation. Although uncommon, patients with palatal pleomorphic adenoma should be warned about the possibility of recurrence or malignant transformation of tumor at advanced ages.

Lacrimal gland pleomorphic adenoma: a narrative review / Adenoma pleomórfico de glândula lacrimal: uma revisão narrativa

ABSTRACT We present a literature review of 57 publications describing this pathology, published from the year 2012. In all these studies patients were reported to depict a slow-growing, motionless mass, which is painless at most times. All cases were managed by total excision, except for one report where adjuvant radiotherapy was applied. Among the several therapeutic strategies, the total tumor resection, preserving the tumor pseudocapsule intact, appears to be a consensus in treating the disease efficiently. Furthermore, fine-needle aspiration biopsy, including the assessment of genetic alterations, has proved to be a valuable tool in the diagnosis of challenging cases. Our literature survey also suggests that an incisional biopsy before the surgery may lead to the pseudocapsule disruption, thus considerably increasing the chances of adenoma recurrence, enabling its malignization. At present, genetics studies indicate that the molecular aberrations involved in the adenoma are similar to those represented in the salivary gland tumor pathogenesis. Further, in the recurrent cases, the pathology becomes difficult to treat and multiple surgeries may be required, occasionally, leading to radical surgery treatment.

RESUMO Uma revisão narrativa da literatura de 57 publicações que descrevem esta patologia, publicada a partir de 2012. Os pacientes têm uma massa de crescimento lento e imóvel, que na maioria das vezes é indolor. Todos os casos foram tratados por excisão total, com exceção de um relatório de radioterapia adjuvante. Entre as estratégias terapêuticas encontradas, a ressecção total do tumor, preservando a pseudocápsula tumoral intacta, parece ser um consenso. Alternativamente, a biópsia por aspiração de agulha fina incluindo a avaliação de alterações genéticas pode representar uma ferramenta valiosa nos casos diagnósticos desafiadores. Uma biópsia incisional antes da cirurgia não é recomendada, pois a ruptura da pseudocápsula aumenta consideravelmente a recorrência do adenoma, permitindo até mesmo sua malignização. Com relação à genética, estudos atuais indicam que as aberrações moleculares envolvidas no adenoma são semelhantes às da patogênese do tumor da glândula salivar. Para casos de recorrência, a patologia torna-se difícil de tratar e múltiplas cirurgias podem ser necessárias, às vezes levando a um tratamento cirúrgico radical.

Oral rehabilitation after partial maxilectomy for removal of pleomorphic adenoma: case report / Reabilitação oral após maxilectomia parcial para remoção de adenoma pleomórfico: relato de caso

ABSTRACT Salivary gland tumors account for only 3%-5% of all tumors in the head and neck, 10%-15% of which originate from minor salivary glands. Pleomorphic adenoma is a benign lesion of the salivary gland, most commonly occurring in the region of the hard and soft palates. The treatment of choice for pleomorphic adenomas is with the partial or total function of the extension of the lesion, and placing palatal obturators are one method of reestablishing masticatory function and facial esthetics. This study aimed to rehabilitate a patient using a palatal implant following partial maxillectomy for the removal of a pleomorphic adenoma. A young patient with pleomorphic adenoma of the hard palate underwent a partial right-sided maxillectomy procedure which removed the hard palate and alveolar regions of the molars. Prior to surgery, the patient was assessed to make a surgical guide for resection of the tumor, as well as a provisional obturator plate using orthodontic wire clasps. After healing, the patient was rehabilitated using a palatal obturator which had been incorporated into a removable partial denture. The clinical sequence used to fabricate the final prosthesis was as follows: initial molding after surgery, prosthesis design, preparation of the mouth, work molding, structure testing and orientation planning, teeth testing and installation, and periodic maintenance. Thus, we can conclude that the palatal obturator is an excellent means of restoring a patient's oral function, facial esthetics, and overall quality of life.

RESUMO Os tumores de glândulas salivares representam apenas 3%-5% de todos os tumores em região de cabeça e pescoço, dentre eles 10%-15% se originam de glândulas salivares menores. O adenoma pleomórfico é uma lesão benigna de glândula salivar, tendo uma maior prevalência em região de palato. O tratamento dessas lesões se dá por método excisional ou maxilectomia dependendo da extensão da lesão e, uma das formas de reestabelecimento da estética e função do paciente é com um obturador palatino. O objetivo deste trabalho foi relatar uma reabilitação oral através da utilização de um obturador palatino em um paciente submetido a maxilectomia parcial para remoção de adenoma pleomórfico. Paciente de 22 anos, com adenoma pleomórfico em região de palato, foi submetido a maxilectomia parcial, removendo região de palato duro e região alveolar dos molares do lado direito. Foi realizada moldagem da maxila previamente a cirurgia para confecção de um guia cirúrgico, o guia foi utilizado na ressecção do tumor e, serviu também, para confecção de uma placa obturadora provisória associada a fios ortodônticos. Após cicatrização completa, foi acordado que a reabilitação do paciente seria com uma prótese parcial removível do tipo obturador palatino. A sequência clínica para a realização da prótese foi: moldagem inicial após a cirurgia, delineamento, preparo de boca, moldagem de trabalho, prova da infraestrutura e plano de orientação, prova dos dentes, instalação e manutenções periódicas. Com isso, podemos concluir que o obturador palatino é um excelente meio de reestabelecer função e estética do paciente, melhorando sua qualidade de vida.

Siringoma condroide: apresentação incomum de tumor raro / Chondroid syringoma: unusual presentation of a rare tumor

O siringoma condroide, também conhecido como tumor misto cutâneo, é uma neoplasia benigna rara, originada das glândulas sudoríparas, composta por estruturas epiteliais imersas em um estroma mixocondroide. Geralmente, apresenta-se como tumor sólido, único, localizado em face ou pescoço, com evolução crônica e assintomática. Relata-se caso de mulher, 75 anos, com lesão discretamente elevada na fronte, cujo diagnóstico foi definido pela análise histopatológica.

Chondroid syringoma, also known as a cutaneous mixed tumor, is a rare benign neoplasm originating from the sweat glands, composed of epithelial structures immersed in a myxochondroid stroma. It usually presents as a solid, single tumor located on the face or neck with a chronic and asymptomatic course. We report the case of a 75-year-old woman with a slightly elevated lesion on the forehead, whose diagnosis was defined by histopathological analysis.

Pleomorphic adenoma of the hard palate with calcifications: an unusual presentation / Adenoma pleomórfico del paladar duro con calcificaciones: una presentación inusual

Pleomorphic Adenoma (PA) is the most common benign salivary gland tumor. The most common sites for minor salivary gland from which PA arises are the palate followed by the lips and buccal mucosa. Calcifications are a common finding in major salivary glands with chronic inflammatory disorders. Major salivary gland tumors rarely show calcifications and it is less common to find them in minor salivary gland tumors. We report a case of pleomorphic adenoma of the hard palate in a 67-year-old female patient with intra-tumoral, irregular and scattered calcifications visible on computed tomography (CT). The treatment was complete surgical excision of the lesion. The diagnosis was confirmed with the histopathological study.

El adenoma pleomórfico (AP) es el tumor benigno de las glándulas salivales más común. Los sitios de mayor frecuencia donde surge el AP en glándulas salivales menores es el paladar seguido de los labios y la mucosa bucal. Las calcificaciones son un hallazgo común en las glándulas salivales mayores con trastornos inflamatorios crónicos, pero en el caso de tumores rara vez muestran calcificaciones y es menos común encontrarlos en tumores de las glándulas salivales menores. Presentamos un caso de adenoma pleomórfico del paladar duro en una paciente de 67 años con calcificaciones intratumorales, irregulares y dispersas visibles en la tomografía computarizada. El tratamiento fue la extirpación quirúrgica completa de la lesión. El diagnóstico se confirmó con el estudio histopatológico

Differentiating parotid gland pleomorphic adenoma from adenolymphoma based on T1WI and IDEAL-T2WI radiomics models / 中国医学影像技术

Objective: To explore the value of radiomics features and machine learning models based on T1WI and IDEAL-T2WI in differential diagnosis of parotid gland pleomorphic adenoma (PA) from adenolymphoma (AL). Methods: Clinical and imaging data of 58 patients with parotid tumors, including 33 with PA and 25 with AL were retrospectively analyzed. Axial T1WI and IDEAL-T2WI were manually segmented, and radiomics features were extracted using the radcloud software. Effective radiomics features were selected with the variance threshold method, SelectKBest and Lasso algorithm. The machine learning models were established by using random forest and Logistic regression algorithm, and the ROC curves were drawn to analyze the diagnostic performance. The ability of T1WI, IDEAL-T2WI and image combination in diagnosis of PA from AL were analyzed. Results: T1WI, IDEAL-T2WI and IDEAL-T2WI combined with T1WI obtained 6, 9 and 12 effective features. The random forest model based on IDEAL-T2WI combined with T1WI sequences had the highest diagnostic performance, with AUC of 0.87 (95%CI[0.59,1.00]) and accuracy of 0.83. Conclusion: Radiomics features and machine learning models based on T1WI and IDEAL-T2WI can provide important references for differentiation of PA and AL.

Enhanced CT combined with histogram analysis for differential diagnosis of Warthin tumor and polymorphic adenoma of parotid gland / 中国医学影像技术

Objective: To investigate the value of enhanced CT combined with histogram analysis in differential diagnosis of Warthin tumor and pleomorphic adenoma(PA) of parotid. Methods: Maxillofacial plain and enhanced CT were performed on 37 patients with Warthin tumor (Warthin tumor group) and 28 patients with PA (PA group). CT values of plain, arterial phase (AP) and venous phase (VP) CT of lesions were measured, and the enhanced patterns and pure enhanced values were recorded. Then the parameters of histogram analysis, including the mean, standard deviation (StdDev), minimum (Min), maximum (Max), Median, skewness (Skew) and kurtosis (Kurt) were calculated. The differences of all parameters were compared between groups. ROC curves of parameters being statistically different between groups were drawn for differential diagnosis of Warthin tumor and PA of parotid gland, and the corresponding diagnostic efficacy were analyzed. Results: There were statistically differences of CT values in each phase, AP pure enhanced values and enhanced patterns between groups (all P<0.05), and MeanAP, MinAP, MaxAP, MedianAP, SkewAP, MeanVP, StdDevVP, MinVP, MedianVP and SkewVP also showed significant differences (all P<0.05). AUC and Youden indexes of enhanced pattern, AP pure enhanced value, MeanAP, MinAP, MaxAP, MedianAP and SkewAP were all higher than those of VP histogram parameters in differential diagnosis of PA and Warthin tumor. SkewAP had the highest Youden index (0.71), with AUC of 0.88. The diagnostic efficacy enhanced pattern+SkewAP was the most effective, with AUC, Youden index, sensitivity and specificity of 0.94, 0.80, 83.58% and 96.43%, respectively. Conclusion: Enhanced CT combined with histogram analysis based on AP images were helpful to differential diagnosis of Warthin tumor and polymorphic adenoma of parotid. The diagnosis performance of combined parameters was better than that of single parameter.

Clinicopathological analysis of salivary gland tumors over a 15-year period

Abstract Salivary gland tumors (SGT) are rare neoplasms that generate interest due to their histopathological diversity and clinical behavior. The aims of the present study were to investigate clinicopathological aspects of SGTs diagnosed at a tertiary health center and compare the findings with epidemiological data from different geographic locations. Cases of tumor in the head and neck region at a single health center in the period between 1995 and 2010 were reviewed. Patient gender, age and ethnic group as well as anatomic location, histological type and clinical behavior of the tumor were recorded. Availability of complete information about these aspects was considered the inclusion criteria. Descriptive statistical analysis of the data was performed using the frequencies of categorical variables. Among the 2168 cases of tumors in the head and neck region, 243 (11.20%) cases were diagnosed in the salivary glands, 109 of which met the inclusion criteria: 85 (78%) benign tumors and 24 (22%) malignant tumors. Mean patient age was 46.47 years. The female gender accounted for 56 cases (51.4%) and the male gender accounted for 53 (48.3%). The major salivary glands were affected more (75.2%) than the minor glands. The most frequent benign and malignant SGTs were pleomorphic adenoma (81.2%) and adenoid cystic carcinoma (58.3%), respectively. In conclusion, pleomorphic adenoma and adenoid cystic carcinoma are the most frequent benign and malignant lesions, respectively. Comparing the present data with previous studies on SGTs, one may infer that some demographic characteristics and the predominance of malignant tumors vary in different geographic regions.

Nodular Fasciitis of the Parotid Gland, Masquerading as Pleomorphic Adenoma

It is difficult to distinguish nodular fasciitis (NF) from other neoplasm of the parotid gland, especially pleomorphic adenoma (PA) by fine needle aspiration cytology. A 39-year-old female noticed a mass in the parotid region. The aspirate material showed cohesive parts composed of the cells that had oval or spindle-shaped nuclei and relatively abundant cytoplasm and some cells with plasmacytoid features. The background substance was fibromyxoid. PA was diagnosed based on the cytologic findings. Subsequently, parotidectomy was performed and NF was diagnosed based on histologic and immunohistochemical findings. NF in the parotid region is rare and may be misdiagnosed as other benign or malignant tumors of the parotid gland. The clinical history of rapid growth and the presence of mitoses and inflammatory cells help to distinguish NF from PA. In addition, immunohistochemical stains for smooth muscle actin and CD68 are useful to confirm the diagnosis of NF.

Adenoma pleomórfico benigno del paladar: presentación de un caso / Benign pleomorphic adenoma of the palate: a case presentation

Fundamento: el tumor más común de las glándulas salivales es el adenoma pleomórfico. Es un tumor mixto que se presenta con frecuencia en la glándula parótida. Objetivo: presentar las características clínicas e histopatológicas de un adenoma pleomórfico localizado en el paladar. Se describe también el tratamiento realizado. Caso clínico: se presenta el caso de una paciente con un adenoma pleomórfico localizado en el paladar duro. El tumor de 7 cm por 5 cm se encontraba cubierto de mucosa normal firme, en algunos sitios con poca resiliencia, gomosa, no dolorosa; además los dientes de la zona presentaron movilidad. De acuerdo con la radiografía se observó una zona esclerótica difusa y desde el punto de vista histopatológico se detectó la presencia de nidos, cordones y estructuras ductales, con una secreción luminal eosinofílica. A la paciente se le realizó una maxilectomía parcial y colocación de férula intraquirúrgica. Conclusiones: aunque el adenoma pleomórfico es un tumor benigno requiere un tratamiento quirúrgico con resección de un margen de tejido sano debido a que tiende a infiltrar la cápsula. En el presente caso su dimensión fue tal que requirió una maxilectomía parcial.

Background: benign pleomorphic adenoma is the most common tumor in the salivary glands. It is a mixed tumor that frequently appears in the parotid gland. Objective: to present the clinical and histopathologic characteristics of a benign pleomorphic adenoma located in the palate. Clinical case: the case of a patient with a pleomorphic adenoma in the hard palate is presented. The tumor, that was 7 cm by 5 cm, was covered with a normal firm mucosa which had not much resilience and was rubbery and non-painful. Besides, the teeth in the area presented mobility. According to the radiography, there was a diffuse sclerotic region and from the histopathologic point of view, nests, cords, and ductal structures were detected, along with a luminal eosinophilic secretion. The patient underwent a partial maxillectomy and the placing of an intrasurgical splint. Conclusions: in spite of the fact that the pleomorphic adenoma is a benign tumor, it requires a surgical treatment with removal of a part of healthy tissue because the adenoma tens to infiltrate the capsule. The present case, due to its dimensions, required a partial maxillectomy.

Synchronous pleomorphic adenoma and periapical cyst: clinical case report / Adenoma pleomorfo e cisto periapical sincrônicos: relato de caso clínico

The pleomorphic adenoma or mixed benign tumor is the most common benign neoplasia of the salivary gland in human beings. It preferentially occurs in the superficial lobe of the parotid gland. In the oral cavity, associated with the minor salivary glands, it has a greater predilection for the palate region, with slight predisposition in women between the 3rd and 4th decades of life. The radicular cyst is an odontogenic inflammatory cyst resulting from pulp lesions caused by traumas or caries. In spite of being relatively common, there are no reports of association with salivary gland tumors. The aim of this work is to report a case of a 36-year-old woman patient, who presented with synchronous development of a pleomorphic adenoma and periapical cyst within the same region. The option taken was to perform enucleation of both lesions,followed by local curettage. The patient has been followed-up clinically and radiographically for around 18 months without signs of recurrence.

O adenoma pleomórfico ou tumor misto benigno é a neoplasia benigna mais comum primária de glândula salivar. Ocorre preferencialmente no lobo superficial da glândula parótida. Na cavidade oral, associado com as glândulas salivares menores, tem uma maior predileção para a região do palato, com ligeira predisposição em mulheres entre a 3 ª e 4 ª décadas de vida. O cisto radicular é um cisto odontogênico inflamatório decorrente de lesões causadas por traumas ou cárie. Apesar de ser relativamente comum, não há relatos de cistos periapicais associados com tumores de glândula salivar. O objetivo deste artigo é relatar um caso de uma paciente de 36 anos de idade com desenvolvimento sincrônico de adenoma pleomorfo e cisto periapical em uma mesma região. A opção tomada foi a de realizar a enucleação de ambas as lesões, seguido de curetagem local. A paciente está sendo acompanhada clínica e radiograficamente por cerca de 18 meses sem sinais de recidiva.

Adenoma pleomórfico em palato mole / Pleomorphic adenoma of the soft palate

Adenoma pleomórfico (AP) é a neoplasia de glândula salivar mais comum. A lesão apresenta-se como uma massa firme de crescimento lento e indolor. Pode ocorrer em qualquer idade, sendo mais comum em adultos jovens entre 30 e 50 anos. A diversidade do padrão morfológico histopatológico é um dos seus aspectos mais característicos. Este artigo relata um caso de adenoma pleomórfico de palato mole, enfatizando aspectos diagnósticos e terapêuticos.

Pleomorphic adenoma (PA) is the most common salivary gland neoplasm. The lesion present as firm mass with slow rate of growth and asymptomatic. It can occur at any age, but young adults between 30 and 50 years are common. The diversity morphologic pattern is one of the most characteristic aspects of this lesion. This article report a case of pleomorphic adenoma of the soft palate emphasizing diagnostic aspects and therapeutics.

Clinical significance of regional excision of parotid pleomorphic adenoma / 中国耳鼻咽喉头颈外科

OBJECTIVE To investigate the feas-ibility of preserving parotid duct in regional excision of pleomorphic adenoma. METHODS 32 patients with parotid pleomorphic adenoma were treated by regional excision with the preservation of the parotid duct. RESULTS No relapse occurred during 3-6-year follow-up period. Transient facial nerve palsy occurred in only 3.1%(1/32) of patients and the drainage of the parotid duct on the surgical side was preserved. CONCLUSION Remaining function of the parotid gland function was preserved and the facial nerve was well-protected in the regional excision of pleomorphic adenomas, especially for patients with small pleomorphic adenomas.

Imaging analysis of pleomorphic adenomas of the minor salivary glands of the palate / 中华放射学杂志

Objective To study the imaging features of the pleomorphic adenomas of the minor salivary glands in the palate and to improve its diagnostic validity. Methods CT and MRI manifestations of 6 patients with palatal pleomorphic adenomas confirmed pathologically were analyzed retrospectively. There were 2 males and 4 females, and their age ranged from 19 to 54 years, with the mean age of 36.3 years. Results Among 6 cases, 5 tumors located in hard palate, and 1 in soft palate. 4 lesions were round like, and 2 were lobulated. Most tumors showed soft tissue density on CT, slightly long T 1 and slightly long T 2 signal intensity on MRI. Cystic degeneration could occur in the central part in some lesions, which appeared as much lower density or obviously long T 1 and long T 2 signal intensity. On post contrast MR scans, the lesions usually showed slightly homogeneous enhancement. Conclusions Pleomorphic adenomas of the minor salivary glands were the most common benign tumors in the palate. Combined with the clinical history, typical CT and MRI appearances, a definitive diagnosis can be made.