Mucoepidermoid Research Articles

Cervical Lymph Node Invasion in Pediatric Salivary Gland Malignancies: Clinical Overview and Therapeutic Implications.

Pediatric salivary gland malignancies (SGM) present challenges in managing cervical nodes. We aimed to characterize lymph node invasion to inform decisions regarding the need of systematic wide lymph node dissection (WLND). International retrospective study, conducted across seven large French and American pediatric centers, including pediatric patients (0-18 years) diagnosed with SGM from 2000 to 2020. Among the 82 patients (median age 13 years), the parotid gland was frequently affected (60 cases). Histotypes comprised mucoepidermoid (mucoepidermoid carcinoma [MEC], 43 cases), acinic cells (acinic cells carcinoma [AcCC], 22 cases), adenoid cystic (adenoid cystic carcinoma [AdCC], 8 cases), (MASC, 6 cases), and adenocarcinoma (3 cases). Primary treatments were surgery (82 cases) and radiotherapy (20 cases; median dosage 64 gray). Cervical nodes therapy included WLND (≥2 levels, 29 cases), limited nodes resection (LNR; one level, 13 cases), and/or irradiation (4 cases; median 54 gray; range 52.0-63.0). At diagnosis, six patients had cervical node invasion (CNI) managed with LNR (four cases), WLND (two cases), and radiotherapy (three cases). After a median follow-up of 6 years (range 1-22), nine patients had tumor event: local (four cases), cervical relapse/progression (three cases) or combined (two cases). Of the nine with CNI, at diagnosis or relapse, four had MASC. Five-year event-free and overall survival (OS) rates were, respectively, 90.1% and 98.8%. CNI is rare in pediatric SGM but noted in 11% of cases, with higher incidence in MASC. Overall, outcome in SGM is good with a tailored locoregional multidisciplinary approach. Systematic lymph node dissection should be reconsidered. This international multi-institutional study analyzed the clinical presentation and the cervical pattern of relapse of 82 pediatric patients with newly diagnosed salivary gland malignancies. Overall, nodal invasion was rare at diagnosis and only noted in 7%. In addition, 6% developed nodal relapse during follow-up. Incidence of nodal spread was frequent in mammary analogue secretory carcinoma (MASC). The overall outcome was promising with a tailored locoregional multidisciplinary approach. Systematic lymph node dissection should be reconsidered in pediatric salivary gland tumors.

Postradiotherapy Response Assessment Using 18 F-FDG PET/CT in Salivary Gland Carcinoma-A Multicenter Study.

This multicenter study investigates the efficacy of 18 F-FDG PET/CT in postradiotherapy (post-RT) response evaluation in salivary gland carcinoma (SGC). We retrospectively reviewed 115 SGC patients who underwent definitive or adjuvant RT followed by 18 F-FDG PET/CT between 2004 and 2021. Most tumors were parotid gland malignancies (50%). The most common histological subtypes were adenoid cystic (29%) and mucoepidermoid carcinomas (18%). The median follow-up was 65 months. Post-RT anatomic images (CT/MRI) revealed complete response (CR) in 51 patients (44%). Among 53 patients with partial response or stable disease, only 17 (32%) patients experienced locoregional recurrence, with a 5-year locoregional control rate of 69%. Post-RT 18 F-FDG PET/CT documented metabolic CR in 81 patients (70%). Metabolic complete responders had significantly higher 5-year locoregional control (90% vs 43%), distant metastasis-free survival (80% vs 48%), progression-free survival (76% vs 24%), and overall survival rates (89% vs 42%) compared with non-complete responders (all P < 0.001), as confirmed in both univariate and multivariate analyses. It identified additional viable tumors in 18 cases (16%) and facilitated salvage local therapies in 7 patients (6%). The sensitivity, specificity, positive predictive value, negative predictive value, and accuracy of post-RT 18 F-FDG PET/CT were 63%, 91%, 70%, 88%, and 84%, respectively, in predicting locoregional recurrence. 18 F-FDG PET/CT showed significantly higher sensitivity (88% vs 36%, P = 0.011) in tumors with pre-RT SUV max ≥7.39 compared with those with SUV max <7.39. Post-RT 18 F-FDG PET/CT demonstrates high negative predictive value and specificity, with metabolic CR predicting excellent outcomes. Additionally, it exhibits higher sensitivity for high-SUV max SGC, facilitating early detection of viable tumors.

Multiple brain metastases from mucoepidermoid carcinoma of the parotid gland: case report and review of the literature

Multiple brain metastases from mucoepidermoid carcinoma of the parotid gland: case report and review of the literature

Primary Cutaneous Mucoepidermoid Carcinoma of Parotideomasseteric Region: A Rare Case Report

Mucoepidermoid carcinoma is a distinctive salivary gland malignancy and its presentation as a primary cutaneous carcinoma is a very rare occurrence. It presents grossly as an ill-defined mass with firm to hard consistency and can have cystic areas filled with mucin. Microscopically, they are characteristically composed of mucinous, intermediate and squamoid tumour cells forming cystic and solid patterns. Here we present a case of a 79-year-old female who presented with an ulcerative nodular exophytic growth over right parotideomasseteric region. CECT neck revealed it to be a heterogeneously enhancing lesion arising from the skin with the underlying subcutaneous fat, soft tissues and parotid gland to be normal. On histological and immunohistochemical examination, it was diagnosed as primary cutaneous intermediate grade mucoepidermoid carcinoma. Keywords: Mucoepidermoid carcinoma, cutaneous, rare

Aurora kinase A expression in pleomorphic adenoma, adenoid cystic carcinoma, and mucoepidermoid carcinoma of salivary glands: an immunohistochemical study

BackgroundAurora kinase A (AurkA) plays a vital role in mitosis and is therefore critical in tumors development and progression. There are a few studies on AurkA expression in salivary gland tumors. The aim of the present study was to evaluate the expression pattern of AurkA in the most common benign and malignant salivary gland tumors by immunohistochemistry.MethodsIn this retrospective cross-sectional study, 68 cases including 25 pleomorphic adenomas (PAs), 21 adenoid cystic carcinomas (ADCa), 15 mucoepidermoid carcinomas (MEC), and 7 normal salivary glands (NSG) were enrolled from the archive of the Department of Pathology of Shiraz School of Dentistry, Iran. The expression of AurkA in the tissue samples was assessed by immunohistochemical method and was analyzed using statistical analysis (p < 0.05).ResultsOf total cases analyzed, the majority of benign and malignant tumors were found to involve minor salivary glands compared to major salivary glands (p < 0.001). In addition, all lesions studied expressed AurkA. More than half of the tumor tissues showed AurkA staining percentages between 26 and 50% and 76–100% compared to NSG (p = 0.08). In 44.1% of cases, cells had a weak staining score, 27.9% a moderate score and the rest (27.9%) a strong score (p = 0.64).ConclusionAlthough AurkA was observed to be expressed in all tumor tissues, further studies are needed to clearly understand the role of AurkA and the possibility of using it as a diagnostic, prognostic and therapeutic factor.

Magnetic Three-dimensional Bioprinting Protocol for Salivary Gland Mucoepidermoid Carcinoma Tumoroid Culture.

Magnetic Three-dimensional Bioprinting Protocol for Salivary Gland Mucoepidermoid Carcinoma Tumoroid Culture.

Molecular profile of salivary gland tumors: discovering new targets for therapy

Introduction. Due to variety of morphological subtypes and poor clinical response, comprehensive molecular profiling for salivary gland cancers become a part of treatment strategy for chemotherapy-resistant cases. Tumors of the salivary glands include more than 20 histological types distinguished by their biological characteristics, response to therapy, and prognosis. The search for specific therapeutically significant markers and mutations for various histological types of salivary gland tumors may allow the development of a diagnostic algorithm and improve patient treatment results.Aim. To search for therapeutically significant molecular and genetic targets and immunohistochemical markers in various histological types of malignant salivary gland tumors.Materials and methods. We analyzed data on 280 patients with unresectable recurrent and metastatic salivary gland cancer to identify the main characteristic therapeutically significant immunohistochemical (androgen receptor (AR), epidermal growth factor receptor type 2 (HER2neu), epidermal growth factor receptor (EgfR), programmed cell death receptor 1 (pD-L1) (CpS), сD117, estrogen receptor (ER), progesterone receptor (pR), pan-TRk) and molecular genetic targets (ALK, BRAF, ERBB2, KIT, MET, RET, ROS1, SMO, AR, HRAS, PIK3CA, PDGFRA, NTRK1-3) depending on the histological subtype of the tumor.Results. Based on the results obtained, in comparison with previous studies, a greater diversity of therapeutically significant mutations, fusions and immunohistochemical markers was revealed for various histological types of tumors. Interestingly, increased expression of AR was detected not only in salivary duct carcinoma, but also in carcinoma ex pleomorphic adenoma, adenocarcinoma not otherwise specified, myoepithelial, mucoepidermoid, adenoid cystic, acinic cell, polymorphous adenocarcinomas. ETV6-NTRK3 gene fusions in addition to secretory carcinoma were detected in mucoepidermoid and salivary duct carcinomas. mutations and fusions in the RET gene have been identified in ductal and mucoepidermoid carcinomas, as well as adenocarcinoma not otherwise specified.Conclusion. The results of the study allow to expand the group of patients for molecular genetic and IHC testing in order to individualize therapy.

Mucoepidermoid Carcinoma of the Tracheobronchial Tree: A Rare and Challenging Case in Interventional Pulmonology

Mucoepidermoid Carcinoma of the Tracheobronchial Tree: A Rare and Challenging Case in Interventional Pulmonology

Systematic identification of pathological mechanisms, prognostic biomarkers and therapeutic targets by integrating lncRNA expression variation in salivary gland mucoepidermoid carcinoma

Biologicalprocesses intricately intertwine with tumorigenesis, significantly influencing treatment outcomes and prognosis. However, the mechanisms fostering mucoepidermoid carcinoma (MEC) remain inadequately elucidated. This research utilizes expression profiles of lncRNAs from clinical MEC tissues and matched normal glandular tissues, integrating public data to explore the biological mechanisms and immune microenvironment characteristics of tumorigenesis. Gene set enrichment analysis identified key pathways, and a customized epithelial-mesenchymal transition (EMT) score elucidated the relationship between pathological processes and prognosis, while an immune signature revealed tumor microenvironment characteristics. MECs exhibited significant enrichment in EMT pathway, with key genes such as Secretogranin II, tissue factor pathway inhibitor 2, and periostin identified as contributors to the EMT process. High EMT scores correlated with upregulated EMT and immune response activity, indicating poor prognosis. Single-sample gene set enrichment analysis unveiled the tumors’ immune infiltration signature, suggesting active antigen presentation and a positive immune response for immunotherapy. Additionally, SLC2A1-AS1 and CERS6-AS1 were identified as potential mediators of EMT and the immune environment. This study provides insights into the biological processes of MEC tumorigenesis and identifies potential therapeutic targets for future research.

Expression rate of LAT1 in high-grade parotid gland carcinoma and potential of BNCT as a treatment option for recurrent parotid gland carcinoma.

Boron neutron capture therapy (BNCT) perform as a treatment option for locally advanced or recurrent unresectable head and neck cancers since June 2020 in Japan. The effect of BNCT on parotid carcinoma, which presents a variety of histologic types, remains unclear. The object of this study was to investigate the antitumor efficacy of BNCT against parotid gland carcinoma by focusing on LAT1, which is involved in the uptake of L-BPA, the boron compound used in BNCT. We performed a retrospective review of 42 high-grade parotid gland carcinoma cases. We immunostained specimens using an anti-LAT1 monoclonal antibody to assess the LAT1 expression rate. We divided the patients into two groups: LAT1 expression rate below 10% (negative group) and 10% or higher (positive group), and performed a survival rate analysis. The study included 42 patients categorized by histological three type into mucoepidermoid carcinoma (MEC), salivary duct carcinoma (SDC), and adenoid cystic carcinoma (AdCC). The LAT1 expression rate in the 42 cases was 37.5%, and the rates for each histological type were: AdCC 15.0%; MEC 17.5%; and SDC 60.0%. The 5-year disease-free survival (DFS) rates were 46.8% in the positive group and 81.8% in the negative group (p=0.17). The LAT1 expression rate was 37.5% in cases of high-grade parotid gland carcinoma, and SDC exhibited particularly high expression. From the perspective of LAT1 expression, our results indicated that L-BPA-based BNCT is likely to achieve therapeutic efficacy against parotid gland carcinoma.

Reappraisal of Oncocytic Adenocarcinoma: Unveiling Its Connection to Oncocytic Variants of Salivary Duct Carcinoma and Mucoepidermoid Carcinoma Through ImmunoHisto-Molecular Perspectives.

Oncocytic adenocarcinoma (OC) of the salivary glands is a rare and controversial entity. It was recently reclassified as "salivary carcinoma NOS and emerging entities" in the 2022 WHO classification of head and neck tumors. The lack of specific molecular alterations and its potential affiliation with other salivary gland carcinomas, such as the oncocytic mucoepidermoid carcinomas (OMEC) or the oncocytic subtype of salivary duct carcinomas (OSDC) justified this reclassification. It is becoming essential to clarify the complex spectrum of potential diagnoses surrounding oncocytic tumors. The objective of this study was to explore the histologic features, as well as the immunohistochemical and molecular profiles, of cases previously diagnosed as OC or OMEC of the salivary glands. This study involved 28 cases of carcinomas with a predominantly oncocytic component. The sex distribution was equal. The median age was 59 years (range 10 to 89). Most of these cases originated from the parotid gland (25/28). The mean tumor size was 2.4cm (range 0.5 to 6.5). Primary immuno-morphological and mutation/gene fusion profiles reclassified mainly (64.3%, 18/28). Most of them were reclassified in descending order as OSDC (8/18), OMEC (5/18), and OC (2/18). But 3 cases remained unclassified (3/18). The transcriptomic analysis found a proximity of their transcriptomic profile with the OMEC group and a distance from the OSDCs. These findings imply that OC is not distinct but represents oncocytic variants of other salivary carcinomas. It underscores the importance of thorough morphologic, immunohistochemical, and molecular examinations to accurately diagnose carcinomas with predominant oncocytic components in the salivary glands.

Mucoepidermoid carcinoma in sublingual area: Case report

Mucoepidermoid carcinoma in sublingual area: Case report

Malignant Salivary Gland Tumors in a Tertiary University Hospital in Northern Spain

(1) Background: Salivary gland tumors (SGTs) are a rare and diverse group of neoplasms arising in the parotid, submandibular, sublingual, and minor salivary glands distributed throughout the upper aerodigestive tract. Given the rarity and complexity of MSGTs, understanding their epidemiology across diverse populations is crucial for improving diagnostic and therapeutic strategies. (2) Methods: A retrospective analysis involving 45 patients diagnosed with malignant salivary gland tumors and treated with curative intention between 1 July, 2016 and 1 July, 2021 in a tertiary academic hospital was performed. (3) Results: Histologically, the most common subtype was adenoid cystic carcinoma in 12 (26.7%) cases, followed by carcinoma ex-pleomorphic adenoma in 7 (15.6%) cases, mucoepidermoid carcinoma in 6 (13.3%) cases, and adenocarcinoma in 6 (13.3%) cases. The majority of tumors were located in the parotid gland in 28 cases (62.2%). The three-year overall survival rate was 80% and the three-year specific survival rate was 86.7%. Tumor grade was significantly associated with local recurrence and the appearance of distant metastasis during the follow-up period (p = 0.04). We also evidenced a worse survival in patients with advanced T-Stage (p = 0.038) and positive nodes in the neck (p = 0.015). (4) Conclusions: Overall, our study reinforces the prognostic significance of tumor grade, T-Stage and N-Stage while challenging the conventional understanding of factors such as PNI, LNM, and margin status.

Glandular odontogenic cyst: Case report and literature review

Glandular Odontogenic Cyst (GOC) is a rare and aggressive developmental odontogenic cyst, presenting significant diagnostic and therapeutic challenges. Since its initial characterization, GOC has remained a diagnostic dilemma due to its histopathological similarities with other odontogenic cysts and tumors, such as dentigerous cysts, lateral periodontal cysts, and central mucoepidermoid carcinoma. Accurate diagnosis often requires a meticulous histopathological examination, as well as consideration of clinical and radiographic features to differentiate GOC from other similar lesions.This case report aims to present a rare occurrence of Glandular Odontogenic Cyst in the anterior maxilla, underscoring the importance of comprehensive diagnostic evaluation, histopathological examination and effective management strategies.

Pulmonary mucoepidermoid carcinoma with endobronchial involvement: A case report

Pulmonary mucoepidermoid carcinoma with endobronchial involvement: A case report

Mucoepidermoid carcinoma of unknown primary in the head and neck: A case report and review of the literature.

Mucoepidermoid carcinoma of unknown primary in the head and neck: A case report and review of the literature.

Immunoarchitectural Pattern and Its Potential Prognostic Value in Mucoepidermoid Carcinoma.

To define tumor immunoarchitectural patterns (IPs) and characterize the immune profile in salivary gland mucoepidermoid carcinoma (MEC) toward assessing MEC prognostic significance and implications for immunotherapy. This study analyzed 41 MEC cases, evaluating the tumor IPs and tumor-infiltrating lymphocyte (TIL) parameters by using whole-slide imaging and AI-assisted assessment. Immunohistochemistry of CD3 and CD8 markers was performed to assess key lymphocyte subpopulations. Immune-rich (I-rich) tumors were characterized by high TIL density and were associated with aggressive tumor behavior, particularly in histological high-grade MEC, with a significant increase in TIL density observed in the inner invasive margin compartment (p < 0.05). I-rich tumor cases were linked to poorer disease-free survival (DFS) (Breslow = 4.686, p = 0.03). Aggressive MEC cases displayed a highly heterogeneous TIL profile, however, no TIL patterns showed a significant impact on DFS (p > 0.05). Despite the high immunogenicity suggested by the abundance of T lymphocytes, the protective role of CD8+ T lymphocytes was not prominent in MEC. I-rich tumors appeared more aggressive and unfavorable MEC cases exhibited high heterogeneity in their TIL profiles. Interrogating the role of TILs in the inner invasive tumor margin may offer a new mechanistic understanding for future immunotherapy discovery.

Relative Frequency of Primary Salivary Gland Tumors: Multicenter Study of 796 Cases from Riyadh, Saudi Arabia.

Background and Objectives: Salivary gland tumors (SGTs) are diverse lesions with varying morphological and clinical characteristics. Limited data exist on the distribution of SGTs in Saudi Arabia. We aimed to fill this gap by examining the distribution of SGTs across four tertiary hospitals in Riyadh. Materials and Methods: A retrospective analysis was conducted on SGT cases diagnosed from January 2010 to December 2022 to investigate the clinicopathological features (tumor location, patient sex, and age). Histological slides were evaluated by two independent certified oral pathologists and classified based on the latest 2022 World Health Organization classification (WHO). Results: There were 796 SGTs. Most cases occur in individuals in their 4th to 5th decades of life. The parotid gland was the predominant site (79.4%), followed by the submandibular gland (9.3%). More than half of the tumors (527, 66.2%) were benign, involving major and minor salivary glands. Pleomorphic adenoma was the most common benign tumor, accounting for 354 cases (44.5%), followed by Warthin tumor with 117 cases (14.7%). Mucoepidermoid carcinoma was the most prevalent malignant tumor, identified in 98 patients (12.3%). Additionally, 36 (4.5%) mesenchymal and 30 (3.8%) hematolymphoid non-epithelial SGTs were reported. Conclusions: This multicenter study is the largest of its kind in Saudi Arabia, identifying pleomorphic adenoma and mucoepidermoid carcinoma as the most commonly reported benign and malignant tumors, respectively. These findings offer valuable insights into the understanding of salivary gland tumors globally.

TRPS1 Immunohistochemistry in Salivary Gland Neoplasms: Analysis on Cytology Cell Blocks and Surgical Follow-Up Correlation.

In this study we aim to analyze the TRPS1 immunostaining of salivary gland tumors (SGT) on cytology cell blocks and compare the staining pattern on subsequent surgical resections. Malignant SGTs, oncocytomas and basal cell adenomas diagnosed on fine needle aspiration were retrieved from 2019 to 2021 database. Cases with surgical follow-up were selected. TRPS1 staining was performed on cytology cell blocks and surgical specimens. Scoring was interpreted by two pathologists independently. Our cohort comprised of 58 cases: 45 malignant and 13 benign. TRPS1 scoring was interpreted for 44 cytology and 51 surgical cases. 14 cytology cases lacked tumor cells on deeper levels. For 7 cases, surgical blocks were not retrievable. TRPS1 positivity for cytology and surgical cases were 52% and 47% respectively. In the malignant cohort, TRPS1 was positive in 21/32 (66%) cytology cases and 21/43 (56%) surgical cases. All cases of basal cell adenocarcinoma, carcinoma ex pleomorphic adenoma and salivary duct carcinoma were TRPS1 positive on cytology. All cases of adenoid cystic carcinoma and acinic cell carcinoma were TRPS1 positive on surgical resections. In the benign cohort, TRPS1 was positive in 2/12 (17%) cytology cases; however, none of the surgically resected benign cases showed reactivity (0/8). Cytology-surgical correlation of TRPS1 staining was done in 37 cases. We found 21 concordant and 16 discordant cases. Discordance was highest in mucoepidermoid carcinoma. TRPS1 is not an entirely specific marker for breast carcinoma. TRPS1 positivity was noted in a substantial number of salivary gland malignant neoplasms. Cases demonstrating discordance in TRPS1 staining pattern on cytology-surgical correlation warrant further exploration.

Mucoepidermoid carcinoma: Enhancing diagnostic accuracy and treatment strategy through machine learning models and web-based prognostic tool.

Oral cancer, particularly mucoepidermoid carcinoma (MEC), presents diagnostic challenges due to its histological diversity and rarity. This study aimed to develop machine learning (ML) models to predict survival outcomes for MEC patients and pioneer a clinically accessible prognostic tool. Using the SEER database (2000-2020), we constructed predictive models with five ML algorithms: Random Forest Classifier (RFC), Gradient Boosting Classifier (GBC), Logistic Regression (LR), K-Nearest Neighbors (KNN), and Multilayer Perceptron (MLP). Predictive variables were identified via Cox regression, and Kaplan-Meier analysis assessed survival trends. Model performance was validated through the area under the curve (AUC) of receiver operating characteristic (ROC) curves. This study included 1314 patients diagnosed with MEC of the oral cavity. The RFC demonstrated the highest predictive accuracy (AUC = 0.55), followed by the GBC and RFC (AUC = 0.53). The most affected primary site was the hard palate, followed by the retromolar and cheek mucosa. Survival rates varied with the treatment modality, with the highest rates observed in patients undergoing surgery alone. ML models have identified age, sex, and metastasis as significant prognostic factors influencing survival outcomes, underscoring the complexity and heterogeneity of MEC. This study highlights ML's potential to enhance survival predictions and personalize treatment for MEC patients. We developed the first web-based prognostic tool, providing a novel, accessible solution for improving clinical decision-making in MEC.