Tumores de parótida en población pediátrica: 10 años de experiencia / PAROTID TUMORS IN PEDIATRIC POPULATION: 10 YEARS OF EXPERIENCE

Introducción: Los tumores de las glándulas salivales son infrecuentes en la población en general y comprenden menos del 3% de los tumores de cabeza y cuello1 . En pediatría el 90% corresponden a tumores de la glándula parótida,2 siendo en su mayoría tumores benignos. Objetivo: Describir la incidencia de tumores parotídeos en un centro de referencia, haciendo hincapié en la variabilidad etiológica y la presentación por grupos etarios. Materiales y métodos: Se trata de un estudio de cohorte retrospectivo de pacientes menores de 18 años con tumores de parótida en un periodo de 10 años: de 2011 a 2021, seguidos en el Servicio de Cirugía General Pediátrica del Hospital Italiano de Buenos Aires. Se incluyeron todos los pacientes pediátricos que presentaron tumor en la región parotídea seguidos o tratados en este centro, se excluyeron aquellos que no se pudieron recabar los datos de forma completa. Las variables analizadas fueron edad, sexo, forma de presentación, estudios complementarios, tipo de tratamiento, histología, complicaciones. Resultados: Se analizaron un total de 33 pacientes, de estos un 64% (N = 21) fueron mujeres, con una relación mujer/hombre de 1,7. La media de edad al diagnóstico fue de 8.5 años, siendo 2 de diagnóstico prenatal y hasta los 18 años. La localización fue en un 57% (N = 19) izquierda, el 43% restante derecha. La forma de presentación más frecuente fue la masa palpable en región parotídea 75% (N=25), en dos pacientes fue un hallazgo en estudios por imágenes: resonancia prenatal y otro por resonancia de cerebro. Conclusiones: en pediatría, a diferencia de la población adulta, se presentan una gran variedad de diagnósticos, que incluye desde lesiones vasculares hasta tumores malignos. Debido a que las lesiones parotídeas malignas son clínicamente indistinguibles de las benignas, es importante establecer un diagnóstico preciso. Esta serie representa esta diversidad etiológica en pediatría, así como la distribución etaria comparada con la descrita por la literatura

Introduction: Salivary gland tumors are rare in the general population and comprise less than 3% of head and neck tumors1. In pediatrics, 90% correspond to tumors of the parotid gland,2 being mostly benign tumors. Objective: To describe the incidence of parotid tumors in a reference center, emphasizing the etiological variability and the presentation by age groups. Materials and methods: Tis is a retrospective cohort study of patients under 18 years of age with parotid tumors over a period of 10 years: from 2011 to 2021, followed up at the Pediatric General Surgery Service of the Italian Hospital of Buenos Aires. All pediatric patients who presented tumor in the parotid region followed up or treated in this center were included, those who could not collect the data completely were excluded. Te variables analyzed were age, sex, form of presentation, complementary studies, type of treatment, histology, and complications. Results: A total of 33 patients were analyzed, of which 64% (N = 21) were women, with a female/male ratio of 1.7. Te mean age at diagnosis was 8.5 years, with 2 prenatal diagnoses and up to 18 years. Te location was 57% (N = 19) left, the remaining 43% right. Te most frequent form of presentation was a palpable mass in the parotid region 75% (N=25), in two patients it was a finding in imaging studies: prenatal MRI and another by brain MRI. Conclusions: in pediatrics, unlike the adult population, a wide variety of diagnoses are presented, ranging from vascular lesions to malignant tumors. Because malignant parotid lesions are clinically indistinguishable from benign ones, it is important to establish an accurate diagnosis. Tis series represents this etiological diversity in pediatrics, as well as the age distribution compared to that described in the literature

Parotid incidentalomas: a systematic review.

OBJECTIVE: Parotid incidentalomas on fluorodeoxyglucose positron emission tomography/computed tomography imaging are rare. A systematic review was performed to assess their aetiology and association with scanning indication, and to develop an evidence-based algorithm for their management. METHODS: A literature search was performed on 25 August 2020 using the keywords 'incidentaloma', 'incidental finding', 'parotid', 'parotid gland', 'salivary gland' and 'head and neck'. Articles were reviewed by two authors before their inclusion. RESULTS: Forty articles were included, totalling 558 incidentalomas. The mean incidence was 0.74 per cent. Lung cancer was the most common imaging indication. The most common aetiologies were cystadenolymphoma, pleomorphic salivary adenoma and metastases. Only cystadenolymphoma (p = 0.015) and pleomorphic salivary adenoma (p = 0.011) were significantly associated with a primary malignancy (lung). The most common further investigations were aspiration cytology, ultrasound imaging and core biopsy, usually prior to parotidectomy. CONCLUSION: If appropriate, parotid incidentalomas should be followed up with flexible endoscopy, skin examinations, and head and neck examinations. Ultrasound-guided core biopsy provides higher accuracy, avoiding repeat sampling and excision biopsy.

Shear wave elastography as a new method to identify parotid lymphoma in primary Sjögren Syndrome patients: an observational study.

Parotid non-Hodgkin lymphoma (NHL) in primary Sjögren syndrome (pSS) has no specific biomarker for diagnosis. Salivary glands ultrasound (US) is largely used, but its contribution in detecting parotid NHL has not been established. The aim of our study was to determine the added value of bidimensional shear wave elastography (2D-SWE) in pSS diagnosis and to determine its accuracy in identifying parotid NHL. Grey-scale US (GSUS) and 2D-SWE of salivary glands were performed in 35 patients with pSS and 35 healthy controls. The GSUS scores were calculated and the mean of three SWE consecutive measurements was used to appreciate the gland stiffness. SWE increase the diagnostic rate at a cut-off of 6.45 kPa (from 88.6 to 94.2%, p < 0.001) only if applied in patients with insufficient GSUS criteria for pSS diagnosis. The parotid glands with NHL (8 patients, all mucosa-associated lymphoid tissue type) had hyperechoic bands in more than half of the glandular parenchyma (in 68.75% of the glands), large hypoechoic area > 20 mm (all glands), traced gland area over 5 cm2 (all glands), parotid US score greater than 13 (in 68.75% of the glands), and high stiffness (elasticity modulus 13.9 ± 4.08 vs 6.32 ± 2.24) (all p < 0.001). These findings give high sensitivity (92.3%), specificity (100%), and positive (100%) and negative predictive values (98.3%) for NHL identification. The rest of GSUS findings did not correlate with the classic risk factors for lymphoma development (all p > 0.05). 2D-SWE had added value for pSS diagnosis in cases where GSUS aspect is normal or nonspecific. The higher stiffness of parotid NHL can be used for early diagnosis, biopsy guidance, and, possible, for treatment monitoring.

Warthin's tumor with necrotizing tuberculous granulomatous inflammation causing severe facial nerve adhesion in parotid gland: A case report and literature review.

RATIONALE: Warthin's tumor is the second most common tumor arising from the parotid gland, but it rarely occurs concomitantly with tuberculous granulomatous inflammation with only 13 documented case reports in the English literature. PATIENT CONCERNS: An 82-year-old woman had a left infraauricular mass for approximately 3 years that had significantly increased in size over the previous 1 month. DIAGNOSES: A diagnosis of Warthin's tumor was made by ultrasonography (US)-guided core needle biopsy. Pathological examinations of the specimen obtained by total extirpation confirmed that the tumor was superimposed with tuberculous granuloma. INTERVENTIONS: The core biopsy wound did not heal and there was formation of a skin fistula tract with persistent discharge. During the operation with en bloc resection of the necrotic parotid tumor, adhesion between the branches of the facial nerve was too tight to allow preservation. OUTCOMES: A diagnosis of necrotic Warthin's tumor superimposed with tuberculous granuloma was made. Due to the high-clinical suspicion of tuberculosis (TB) due to Mycobacterium tuberculosis infection, anti-TB chemotherapy was given. LESSONS: Poor wound healing from a core biopsy and formation of a skin fistulous tract with persistent discharge should raise concern regarding potential extrapulmonary tuberculous infection. Although very rare, tuberculous granuloma concomitant with Warthin's tumor should be considered in the differential diagnosis of a parotid mass lesion. Adhesion of branches of the facial nerve should be expected, and sacrifice of the nerve may be planned. This consideration can be explained to the patient in preoperative counseling and planning. Anti-TB chemotherapy should be given in cases with a definite pathological report associated with speculative clinical presentation.

Surgical treatment strategy in Warthin tumor of the parotid gland / Estratégia de tratamento cirúrgico no tumor de Warthin de glândula parótida

Abstract Introduction: Warthin tumors are the second most common benign tumors of the parotid gland. We examined the clinical features of Warthin tumors in our hospital, and analyzed the consistency within the literatures. Objective: The aim of this study is to analyze the clinical features of Warthin tumors in our 10-year experience of 118 Warthin tumors undergoing surgery at a single institute. Methods: From December 2006 to December 2016, 110 patients who underwent surgical treatment for Warthin tumors were identified based on their medical records. Results: A total of 118 parotid gland operations were performed in 110 patients. Almost 90% of Warthin tumors were found in males, and average patient age was 66.1 ± 6.1 years. The prevalence of smoking history was 89.1% (98/110). Eight patients (7.3%) had bilateral Warthin tumors. Seventy-seven lesions (65.3%) were located in the parotid tail portion, followed by 34 lesions in the superficial lobe (28.8%) and 7 lesions in the deep lobe (5.9%). Conclusion: We determined the appropriate extent of surgery depending on the fine needle aspiration cytology and tumor location by computed tomography scans. Partial facial dysfunction after the operation was detected in 12 cases, and facial nerve function recovered within 3 months. Only one patient experienced a recurrence, and was disease free after the re-operation. We suggest that our treatment algorithm, depending on the location of tumors and the result of fine needle aspiration cytology, can be useful to determine the appropriate extent of surgery for Warthin tumors.

Resumo Introdução: Os tumores de Warthin são os segundos tumores benignos mais comuns da glândula parótida. Avaliamos as características clínicas dos tumores de Warthin em nosso hospital e analisamos a consistência com a literatura. Objetivo: Analisar as características clínicas dos tumores de Warthin em nossa experiência de 10 anos de 118 tumores de Warthin submetidos a tratamento cirúrgico em um único instituto. Método: De dezembro de 2006 a dezembro de 2016, 110 pacientes que receberam tratamento cirúrgico para tumores de Warthin foram identificados com base em seus prontuários médicos. Resultados: Foram feitas 118 cirurgias na glândula parótida em 110 pacientes. Quase 90% dos tumores de Warthin foram encontrados em homens e a média da idade dos pacientes foi de 66,1 ± 6,1 anos. A prevalência de tabagismo foi de 89,1% (98/110). Oito pacientes (7,3%) tinham tumores de Warthin bilaterais na glândula parótida. Das lesões, 77 (65,3%) localizavam-se na porção da cauda da parótida, seguidas por 34 no lobo superficial (28,8%) e 7 no lobo profundo (5,9%). Conclusão: Determinamos a extensão apropriada da cirurgia de acordo com a punção aspirativa com agulha fina e localização do tumor por tomografia computadorizada. Disfunção facial parcial após a cirurgia foi detectada em 12 casos e a função do nervo facial foi recuperada em 3 meses. Apenas um paciente apresentou recidiva e ficou livre da doença após reoperação. Sugerimos que nosso algoritmo de tratamento, a depender da localização dos tumores e do resultado da PAAF, pode ser útil para determinar a extensão apropriada da cirurgia para os tumores de Warthin.

Radiation-induced angiosarcoma of the parotid gland after postoperative radiotherapy for hypopharyngeal carcinoma.

Secondary carcinogenesis within the irradiation range is one of the most severe problems in cancer survivors. A 60-year-old woman developed hypopharyngeal carcinoma, and she received radical surgery and postoperative radiotherapy. Eight years later, brown pigmentation and induration were observed in the left subaural region. Fine-needle aspiration biopsy revealed malignancy and the parotid tumor was diagnosed as recurrence of hypopharyngeal carcinoma. Neoadjuvant chemotherapy followed by radical parotidectomy was performed. The pathological diagnosis was angiosarcoma, which was most likely induced by past irradiation. About two months after surgery, lung metastases were detected. Docetaxel did not affect to lung metastases, but paclitaxel therapy was partially effective. The lung tumors increased in size, and brain metastases developed, resulting in death. Both neoadjuvant chemotherapy and radical surgery played important roles in the local disease control. Administration of newer agents as adjuvant chemotherapeutic agent should also be considered for improving the prognosis.

Surgical treatment strategy in Warthin tumor of the parotid gland.

INTRODUCTION: Warthin tumors are the second most common benign tumors of the parotid gland. We examined the clinical features of Warthin tumors in our hospital, and analyzed the consistency within the literatures. OBJECTIVE: The aim of this study is to analyze the clinical features of Warthin tumors in our 10-year experience of 118 Warthin tumors undergoing surgery at a single institute. METHODS: From December 2006 to December 2016, 110 patients who underwent surgical treatment for Warthin tumors were identified based on their medical records. RESULTS: A total of 118 parotid gland operations were performed in 110 patients. Almost 90% of Warthin tumors were found in males, and average patient age was 66.1±6.1 years. The prevalence of smoking history was 89.1% (98/110). Eight patients (7.3%) had bilateral Warthin tumors. Seventy-seven lesions (65.3%) were located in the parotid tail portion, followed by 34 lesions in the superficial lobe (28.8%) and 7 lesions in the deep lobe (5.9%). CONCLUSION: We determined the appropriate extent of surgery depending on the fine needle aspiration cytology and tumor location by computed tomography scans. Partial facial dysfunction after the operation was detected in 12 cases, and facial nerve function recovered within 3 months. Only one patient experienced a recurrence, and was disease free after the re-operation. We suggest that our treatment algorithm, depending on the location of tumors and the result of fine needle aspiration cytology, can be useful to determine the appropriate extent of surgery for Warthin tumors.

Follicular dendritic cell sarcoma presenting as a painless lump in the parotid.

Follicular dendritic cell sarcoma (FDCS) is a rare neoplasm of the antigen presenting cells of the immune system. The majority occur in lymph nodes but around 30% can occur extranodally including in the spleen, lungs, head and neck and liver. We present an unusual case of an FDCS of the parotid gland in a 51-year-old woman with a history of Hodgkin's lymphoma treated with combination chemotherapy and modified mantle radiotherapy. Only four cases of an intraparotid FDCS have been previously reported. The patient underwent a superficial parotidectomy and level 2/3 neck dissection. A diagnosis of an intraparotid FDCS (25 mm) with no nodal disease was made. Given this patient's history of radiotherapy 20 years previously, we speculate the possibility of postradiation sarcoma.

Mucoepidermoid carcinoma post-radioactive iodine treatment of papillary thyroid carcinoma: unique presentation and putative etiologic association.

We report the development of mucoepidermoid carcinomas of the parotid gland in 2 adult patients after a relatively short duration of radioactive iodine (RAI) treatment of papillary thyroid carcinoma. Both instances, together with those previously reported, underscore the selective nature of the mucoepidermoid carcinoma phenotype development in patients with papillary thyroid carcinoma as a consequence of RAI treatment. Efforts to alleviate salivary pathophysiologic damage by RAI in these patients are warranted.

Probabilistic Multiple-Bias Modeling Applied to the Canadian Data From the Interphone Study of Mobile Phone Use and Risk of Glioma, Meningioma, Acoustic Neuroma, and Parotid Gland Tumors.

We undertook a re-analysis of the Canadian data from the 13-country case-control Interphone Study (2001-2004), in which researchers evaluated the associations of mobile phone use with the risks of brain, acoustic neuroma, and parotid gland tumors. In the main publication of the multinational Interphone Study, investigators concluded that biases and errors prevented a causal interpretation. We applied a probabilistic multiple-bias model to address possible biases simultaneously, using validation data from billing records and nonparticipant questionnaires as information on recall error and selective participation. In our modeling, we sought to adjust for these sources of uncertainty and to facilitate interpretation. For glioma, when comparing those in the highest quartile of use (>558 lifetime hours) to those who were not regular users, the odds ratio was 2.0 (95% confidence interval: 1.2, 3.4). After adjustment for selection and recall biases, the odds ratio was 2.2 (95% limits: 1.3, 4.1). There was little evidence of an increase in the risk of meningioma, acoustic neuroma, or parotid gland tumors in relation to mobile phone use. Adjustments for selection and recall biases did not materially affect interpretation in our results from Canadian data.

Multiple clear cell acanthomas and a sebaceous lymphadenoma presenting in a patient with Cowden syndrome - a case report.

Cowden syndrome (CS) is an uncommon autosomal dominant multiorgan/system genodermatosis. It is characterized by the development of multiple hamartomas of endodermal, mesodermal and ectodermal origin, an increased lifetime risk of breast, thyroid, endometrial and other cancers and an identifiable germline mutation. Mucocutaneous hamartomas are the most common lesions seen and mainly include facial trichilemmomas, oral mucosal papillomas and benign acral keratoses. Herein, we report a case of a 63-year-old Caucasian male with a long-established diagnosis of CS and history of thyroid cancer, colonic polyps, and innumerable trichilemmomas, seborrheic keratoses, squamous papillomas and non-melanoma skin cancers excised in the past. He presented in four separate occasions with small skin-colored papulonodular lesions that upon excision revealed to be clear cell acanthomas. He also developed a tumor in the preauricular area that was completely resected and was found to be a sebaceous lymphadenoma (SLA) of the parotid gland. This is to our knowledge, the second report of clear cell acanthoma and also the second reported case of SLA in a patient with CS.

Mobile Phone Use and the Risk of Parotid Gland Tumors: A Retrospective Case-Control Study.

BACKGROUND: Mobile phones are integral part of the modern lifestyle. As they emit radio frequency electromagnetic field, their role in carcinogenesis needs to be ascertained. The goal of this study was to investigate the association between the use of cellular phones and the risk for parotid gland tumors. MATERIALS AND METHODS: A total of 26 patients diagnosed with parotid gland tumors and 61 healthy controls were enrolled through a hospital-based retrospective case-control study. The patients were referred and admitted to a tertiary hospital from January 1996 to March 2013. RESULTS: The Odds of exposure were 3.47 times higher among patients compared to their controls. 95% CI suggested that the true Odds Ratio (OR) at the population level could be somewhere between 1.3 and 9.23 and so the observed OR was statistically significant at 5% level of significance. CONCLUSIONS: Overall, an association between the exposure of cellular phone use for more than 1 hour daily and parotid tumor was observed. This association should be interpreted with caution because of the relatively small sample size.

Metastatic pleomorphic adenoma to the supraspinatus muscle: a case report and review of a rare aggressive clinical entity.

We report a case of a 65-year-old female with a recurrent right parotid pleomorphic adenoma (PA) 24 years after initial surgical excision. Positron-emission tomography (PET) and computed tomography (CT) demonstrated an unusual suspicious FDG-avid erosive rim enhancing mass centered in the right supraspinatus muscle. Cytology from CT-guided aspiration of the mass was consistent with a histologically benign PA, and the patient was diagnosed with metastatic pleomorphic adenoma (MPA). The patient later developed diffuse pulmonary metastases and died within 3 months. MPA, although rare, is recognised as a potentially lethal malignant complication of recurrent or longstanding benign PA. As no biochemical or genetic parameters are predictive of malignant change, patients presenting with recurrent PA should be considered for screening for metastatic disease.

Idiopathic pseudoaneurysm: a rare cause of parotid mass.

A 77-year-old woman presented with a pulsatile blue-tinged mass over the left parotid, present for 5 years, and slowly enlarging. The size of the mass fluctuated during this period and there was no associated pain. The patient reported no history of surgery or trauma. Vascular lesions are rarely encountered within parotid glands in adults. MRI with contrast demonstrated a soft tissue lesion; despite the presence of feeding vessels it was thought unlikely to be an arterial venous malformation or aneurysmal change. The appearance was thought to be in keeping with an enlarged lymph node or sebaceous cyst. Fine-needle aspiration was obtained using ultrasound guidance and yielded bloody fluid. The lesion was closely related to the superficial temporal artery and a diagnosis of a thrombosed pseudoaneurysm was made. The vessel had thrombosed and the decision was made to enact a watchful waiting policy. The patient will be monitored in the outpatient setting.

HER2 and EGFR gene copy number alterations are predominant in high-grade salivary mucoepidermoid carcinoma irrespective of MAML2 fusion status.

AIMS: In this study, we aimed to investigate the molecular mechanisms underlying the development of mucoepidermoid carcinoma (MEC). METHODS AND RESULTS: In 31 cases, we examined the MAML2 fusion status using reverse transcriptase-polymerase chain reaction, and HER2 and EGFR status using immunohistochemistry and chromogenic in-situ hybridization. MAML2 fusions were detected in 15 (57.7%) of 26 MECs analysed, including 11 of 16 (68.8%) low-grade, two of four (50%) intermediate-grade and two of six (33.3%) high-grade MECs. HER2 gene amplification and an increased EGFR gene copy number (with balanced chromosome 7 high-polysomy) were each detected in four of 28 (14.3%) MECs analysed. Irrespective of MAML2 fusion status, all seven high-grade MECs had an increased gene copy number of either HER2 or EGFR, in a mutually exclusive manner, whereas such abnormalities were extremely rare in low- and intermediate-grade MEC. CONCLUSIONS: These results suggest that HER2 or EGFR gene abnormality could play an important role in the development of high-grade MEC, and also in the progression from MAML2 fusion-positive low-/intermediate-grade to high-grade in a subset of MEC. Furthermore, we suggest that high-grade MEC comprises a heterogeneous group of tumours in terms of molecular pathogenesis, in particular MAML2 fusion status.

[MALT-type parotid lymphoma--a case report and the review of the literature]. / Chloniak typu MALT slinianki przyusznej--opis przypadku i przeglad pismiennictwa.

Primary lymphomas of the salivary glands are rare. It is estimated that they constitute no more than 5% of all lymphomas in different locations. The most common subtype developing in parotid glands is marginal zone B-cell mucosa associated lymphoid tissue type lymphoma (MALT) that belongs to a group of low-grade tumours. There are many factors associated with the incidence of that proliferative process: environmental and infectious agents as well as immune deficiency states. We describe a case of primary non-Hodgkin's lymphoma of the parotid gland arising in the background of previously undiagnosed and untreated Sjögren's syndrome in a 52-year-old woman. The article concerns a short review of the literature regarding etiology, symptoms, treatment and survival prognosis in that rare disease as well. MALT lymphomas should always be considered in the differential diagnosis of the tumors and swelling of the parotid gland area. A special, regular monitoring should include all patients with Sjögren's syndrome as those with the proven greater risk of developing that proliferative disease. The role of the laryngologist in the case of MALT-type lymphoma of the parotid gland should focus on a diagnosis and possible tumor cytoreduction with maximal saving of the facial nerve. The essential treatment of this pathology is one of the oncologists and haematologists.

Back to the roots of Warthin's tumor of the parotid gland.

The exact etiopathogenesis of the Warthin's tumor of the parotid gland is still unclear. Since the time of the first histological description of this lesion by Hildebrand and later Albrecht and Arzt many different partly confusing hypothesis about pathogenesis of Warthin's tumor were introduced. However, the review of published data about the pathogenesis of this lesion revealed that recent literature is in conformity with the supposed hypothesis about pathogenesis of Warthin's tumor of those authors about the origin of this lesion from lymph nodes almost a century ago.

Atypical presentation of mucoepidermoid carcinoma after radiation therapy for the treatment of keloids.

Radiation therapy to the head and neck for the treatment of benign diseases carries the potential for the late development of carcinoma. Low-dose radiation has been used as an adjunctive treatment for recurrent keloids, especially massive keloids, but the carcinogenic potential of ionizing radiation in this setting remains controversial. We report the case of a 37-year-old black woman with a history of severe earlobe keloids who had been first treated with resection and postoperative radiation at the age of 9 years. When she had reached the age of 36 years, she required reoperation for massive keloid scarring, after which she underwent a second course of postoperative radiation to the right side of her face and neck. Some 20 months after the second administration of radiation therapy, she developed a mucoepidermoid carcinoma in the right parotid gland. The tumor was successfully treated with surgery.