Oncocytic papillary cystadenoma of the larynx: a case report.

BACKGROUND: Cystadenoma of the salivary glands is a rare benign clinical condition affecting both major and minor salivary glands equally. It constitutes approximately 2% of total neoplasms and 4.2-4.7% of benign formations in minor salivary glands. Typically presenting as a slow-growing, painless neoplasm, it can be distinguished from Cystadenolymphoma (Whartin's Tumor) by the absence of lymphoid elements in histological examination. While mostly located in the oral cavity and oropharynx, it can also be found in sinonasal mucosa, and rare cases have been identified in the larynx. CASE PRESENTATION: A 75-year-old Caucasian woman presented to the ear, nose, and throat department with complaints of dysphonia and headaches persisting for several months. Dysphonia had developed months after an unspecified vocal cord surgery elsewhere. Flexible laryngoscopy identified a left-sided cystic swelling affecting the supraglottic space, leading to respiratory obstruction and dysphonia. Head and neck computed tomography confirmed a 1.9 × 1.7 cm bilobed cystic mass originating from the left Morgagni ventricle. Microlaryngoscopy with CO2 laser excision and biopsy revealed a histopathological diagnosis of oncocytic papillary cystadenoma. Post-surgery, the patient fully recovered from dysphonia, with no significant complications noted. Long-term clinical surveillance was advised to detect potential recurrences promptly. CONCLUSION: Ectopic minor salivary gland tumors, both benign and malignant, should be taken into consideration as potential differential diagnosis for any swelling arising within the upper digestive tract mucosa. Ears, nose, and throat clinical examination completed by videolaryngoscopy can easily point out the location of the mass. Imaging is mandatory for differential diagnosis and for surgical planning. Surgical excision can provide both diagnosis and definitive cure.

Buccal gland within the bucinator muscle.

There are major and minor salivary glands that aid in the digestive process. Major glands are discrete and exist in predictable locations; minor salivary glands are more widespread and usually found dispersed in the mucosa of the mouth. Glands have their own contractile abilities, which allow them to secrete products without the assistance of vasculature or skeletal, or smooth muscle. This study will describe a cadaveric histological specimen in which an ectopic buccal gland was embedded within bucinator muscle fibers. Potential causes and explanations for this finding will be discussed, as well.

Adenoid Cystic Carcinoma of the Minor Salivary Glands: A Systematic Review and Meta-Analysis of Clinical Characteristics and Management Strategies.

BACKGROUND: Intraoral adenoid cystic carcinoma (ACC) arising from minor salivary glands (MSG) is a rare malignancy associated with delayed diagnosis and unfavorable outcomes. This study aimed to comprehensively review ACC of MSGs, focusing on clinical characteristics, imaging modalities, treatment approaches, and long-term outcomes. METHODS: A systematic search was conducted in PubMed, Web of Science, and MEDLINE databases to identify relevant articles reporting cases of ACC of MSGs between January 1997 and March 2023. The study was registered in PROSPERO (ID: CRD42023449478). A total of 10 studies that met the inclusion criteria were selected for critical review. In total, 902 patients were diagnosed with ACC of MSGs with an age range of 44.3 to 63 years, and an average age of 56.6 years. The female to male ratio ranges from 1:1 to 2.4:1. Regarding the primary site of ACC, the palate was the most common location, accounting for 30.5% to 83.3%, followed by the buccal mucosa, floor of the mouth, and lip and the retromolar area. For histology, the solid mass pattern was the most prevalent, seen in 95.2% of patients, followed by the cribriform pattern. Regarding treatment modalities, surgery was the most common approach, applied in 76.3% of cases, with a combination of surgery and radiotherapy used in 29.0% of cases. A smaller fraction, 3.2%, received a combination of surgery, chemotherapy, and radiotherapy, and 8.3% underwent radiotherapy alone. Local recurrence rates varied between 1% and 28.5%, and distant metastasis occurred in 18.2% to 33.3% of cases, predominantly to lymph nodes (14.5%). An analysis of overall survival across various stages and patient numbers indicated a 5-year survival rate of 68.0%. The findings of this study provide valuable insights for physicians in making treatment decisions and emphasize the need for ongoing research and collaborative clinical efforts to improve the management and outcomes of this challenging disease. CONCLUSION: ACC of MSGs is a multifaceted condition typically manifesting as asymptomatic enlargement and ulceration. This disease is marked by distinct histopathological patterns and perineural invasion (PNI). Recognizing these distinctive aspects is key in shaping the treatment plan, which can range from surgical procedures to radiation therapy, chemotherapy, and evolving targeted treatments. Continuous research and collaborative clinical efforts remain critical for ongoing progress in the treatment and management of this challenging condition.

Polymorphous Adenocarcinoma: A Systematic Review of the Literature and Presentation of Two Cases in a Less-Considered Anatomical Site.

BACKGROUND: Polymorphous adenocarcinoma (PAC) is the second-most common malignant tumour of the minor salivary glands. Although PAC predominantly affects the palate, it can also involve the buccal mucosa. This systematic review aims to investigate the literature data about PAC. Furthermore, we report two cases of patients affected by PAC in an infrequently considered anatomical site. METHODS: According to PRISMA guidelines, a systematic review search was conducted on PubMed, Scopus, and Web of Science. Observational studies conducted on patients with a histological diagnosis of PAC were selected and analysed. Furthermore, two cases of patients with PAC affecting the buccal mucosa were reported. RESULTS: Twenty-nine studies were included, and 143 patients affected by PAC were analysed (62 males, 75 females, and 6 undefined, with a mean age of 57.4 ± 14.5 years). The palate was the most affected site (99/143, 69.2%), followed by the buccal mucosa (12/143, 8.4%). Moreover, we report two cases of patients with PAC affecting the buccal mucosa (one male and one female, with a mean age of 70.5 ± 2.5 years). CONCLUSIONS: The present study underscores the importance of considering the buccal mucosa as a possible location of minor salivary gland tumours; although it is a less-considered affliction, it is not uncommon.

Human Minor Salivary Glands: A Readily Available Source of Salivary Stem/Progenitor Cells for Regenerative Applications.

Resident stem/progenitor cells within the secretory salivary glands offer a potential therapeutic resource for use in the regeneration of salivary glands needed to restore saliva production in patients with chronic xerostomia, or dry mouth. Methods were developed previously to isolate human stem/progenitor cells (hS/PCs) from major salivary glands (parotid/submandibular). Abundant minor salivary glands located in readily accessible locations in the oral cavity and lip could provide an additional valuable therapeutic resource. An advantage of this cell resource is that these minor glands about the size of grape seeds can be harvested from healthy donors using minimally invasive surgical procedures. The disadvantage of using minor glands is that they contain many fewer cells than do major glands, and thus harvested cells need to be expanded in the lab to create a therapeutic resource. While earlier work has described isolation of proliferative cell populations from minor salivary glands that could be used in regenerative medicine, most of these expanded cells possess properties of mesenchymal cells rather than the epithelial population that secretes salivary products.Here, we describe in detail our recently established methods to isolate and expand hS/PCs isolated from human labial minor salivary glands. Expanded hS/PC populations are epithelial assessed by their expression of epithelial progenitor markers K5 and K14. Like expandable cell populations previously isolated from the major salivary glands, these cells also express nuclear p63, consistent with their ability to be expanded after explant culture. When hS/PCs with these properties are encapsulated into a customized 3D biomimetic hyaluronic acid-based hydrogel, they will assemble into microstructures that retain some progenitor markers while also beginning to differentiate. The increased expression of secreted mucin MUC-7 was used to demonstrate differentiation and secretory potential in assembled hS/PC microstructures. Compared to hS/PCs from major glands, those from minor salivary glands tend to be more heterogeneous in early passage; thus, use of K5/K14/p63 as an early quality assessment tool is highly recommended. Additionally, hS/PCs from minor glands are sensitive to stress and if mishandled will demonstrate a stress response that leads to their transitioning to a flat, squamous cell-like appearance that is of limited utility in regenerative medicine applications. We conclude that properly handled hS/PCs from minor salivary glands represent a powerful new source of therapeutic cells for applications including treating patients with chronic xerostomia.

Molecular B-cell clonality assay in minor salivary glands as a useful tool for the lymphoma risk assessment in Sjögren's syndrome.

OBJECTIVES: Non-Hodgkin's lymphoma (NHL) risk assessment is crucial in Sjögren's syndrome (SS). We studied the prevalence of clonal immunoglobulin gene rearrangements in minor salivary glands (MSG) and their correlations with lymphoma occurrence and with previously established NHL predictors. METHODS: Molecular B-cell expansion was studied in fresh-frozen MSG of 207 patients with either suspected SS or with suspected lymphoma during SS, using a standardised multiplex PCR assay combined with heteroduplex analysis by microcapillary electrophoresis. The assignation of clonal cases was based on EuroClonality consortium guidelines. RESULTS: Among 207 studied patients, 31 (15%) had MSG monoclonal B-cell infiltration. Monoclonality was significantly more frequent in patients with SS (28/123, 22.8%) compared with patients without SS (3/84, 3.6%, P<0.001). Monoclonal B-cell infiltration in MSG of SS patients correlated significantly with ongoing salivary gland NHL, salivary gland swelling, CD4+ T-cell lymphopenia, rheumatoid factor (RF) activity, low complement levels and type 2 mixed cryoglobulinemia. The accumulation of biological risk factors was associated with a higher rate of MSG B-cell monoclonality given that patients with only positive RF had no probability of MSG B-cell monoclonality, RF-positive patients with 1 or 2 other risk factors had a 25.0% and 85.7% probability of MSG B-cell monoclonality, respectively. CONCLUSION: The detection of MSG monoclonal B-cell expansion by this easy-to-perform molecular assay is useful, both at the time of diagnosis and during the course of SS. Monoclonal B-cell expansion is associated with a subset of SS patients presenting either ongoing lymphoma or other established lymphoma predictive factors.

Lymph node surgery for salivary gland cancer: REFCOR recommendations by the formal consensus method.

OBJECTIVE: To determine the indications for neck dissection in the management of parotid, submandibular or minor salivary gland cancers depending on the clinical situation: i.e., clinical lymph node involvement (cN+) or not (cN0); low or high risk of occult nodal metastasis; diagnosis of malignancy before, during or after surgery. MATERIAL AND METHODS: The French Network of Rare Head and Neck Tumors (REFCOR) formed a steering group which drafted a narrative review of the literature published on Medline and proposed recommendations. The level of adherence to the recommendations was then assessed by a rating group according to the formal consensus method. RESULTS: In cN+ salivary gland cancer, ipsilateral neck dissection is recommended. In cN0 salivary gland cancer, ipsilateral neck dissection is recommended, except for tumors at low risk of occult nodal metastasis. If definitive pathology reveals a high risk of occult nodal involvement, additional neck treatment is recommended: ipsilateral neck dissection or elective nodal irradiation. CONCLUSION: The rate of occult lymph node involvement, and therefore the indication for elective neck dissection, depends primarily on the pathologic grade of the salivary gland cancer.

Basal cell adenocarcinoma of the soft palate: A closer look for an exceedingly rare salivary gland tumor.

Basal cell adenocarcinoma (BCAd) is an extremely rare primary biphasic carcinoma of the salivary glands with few well-documented cases reported in the literature. Herein, we report a rare case of a 44-year-old male patient who presented an oral medicine service with an erythematous nodular lesion on the soft palate, measuring 1.5 cm in its largest diameter, with a 5-year duration. The clinical diagnosis was pleomorphic adenoma, and an excisional biopsy was performed. Histopathological analysis revealed a biphasic infiltrative tumor composed of a mixture of central ductal cells and abluminal basal cells with slight atypia arranged in solid, trabecular, tubular and cribriform growth patterns in a loose stroma. The peripheral cells show a palisading arrangement with round hyperchromatic nuclei and scanty cytoplasm. Occasional mitotic figures were seen. Few spindle-shaped cells suggestive of myoepithelial cells were present in the stroma surrounding the basaloid tumor nests. The diagnosis was BCAd. The patient was referred to a head and neck service and has been followed up for 8 months with no signs of recurrence. In conclusion, although the diagnosis of BCAd can be challenging due to its rarity and morphological overlap with other salivary gland lesions, a meticulous morphological assessment is key for accurate diagnosis, especially in cases originating from minor salivary glands. Surgical excision with a wide safety margin is the treatment of choice and long-term follow-up is recommended to monitor possible recurrences.

Pleomorphic adenomia of lower lip: A case report.

Pleomorphic adenoma is a benign tumor of the salivary glands. It commonly occurs in the parotid gland, palate, upper lip and cheek. The authors present a rare case of a pleomorphic adenoma of the lower lip in a 30 years old female admitted on 20th of July, 2022 at Akbar Niazi Teaching Hospital, Islamabad with a complaint of painless, slightly itchy swelling on the lower lip for the last four months. Careful history and examination revealed a swelling of the lower lip which had gradually increased in size but was static for the last three months. As the patient complained of cosmetic and social inconvenience, it was surgically managed. Any post-operative complications were ruled out and the patient was sent home in a good condition. Much research is warranted to know the exact etiopathogenesis and appropriate management of pleomorphic adenoma of the lower lip.

UHFUS: A Valuable Tool in Evaluating Exocrine Gland Abnormalities in Sjögren's Disease.

Sjögren's Disease (SjD) is a chronic autoimmune disorder that affects the salivary and lacrimal glands, leading to xerostomia and xerophthalmia. Ultrasonography of Major Salivary Glands (SGUS) is a well-established tool for the identification of the salivary glands' abnormalities in SjD. Recently, a growing interest has arisen in the assessment of the other exocrine glands with ultrasonography: lacrimal glands (LGUS) and labial salivary glands (LSGUS). The objective of this study is to explore the practical applications of ultra-high frequency ultrasound (UHFUS) in the assessment of lacrimal glands and labial salivary glands. Indeed, UHFUS, with its improved spatial resolution compared to conventional ultrasonography, allows for the evaluation of microscopic structures and has been successfully applied in various medical fields. In lacrimal glands, conventional high-frequency ultrasound (HFUS) can detect characteristic inflammatory changes, atrophic alterations, blood flow patterns, and neoplastic lesions associated with SjD. However, sometimes it is challenging to identify lacrimal glands characteristics, thus making UHFUS a promising tool. Regarding labial salivary glands, limited research is available with conventional HFUS, but UHFUS proves to be a good tool to evaluate glandular inhomogeneity and to guide labial salivary glands biopsy. The comprehensive understanding of organ involvement facilitated by UHFUS may significantly improve the management of SjD patients.

Histopathological evaluation of minor salivary gland aberrations in oral squamous cell carcinoma.

Background: Oral squamous cell carcinoma (OSCC) is one of the most established oral cancers in India, with high morbidity and mortality. The most common etiological agent associated with it is tobacco (in any form), which releases chemical carcinogens that affect not only the oral epithelial lining but also deep stromal structures such as minor salivary glands. They may cause changes in ductal or acinar part of gland depending on tumor grade, thus providing a fertile soil for tumor growth and recurrence. Aims and Objective: To observe the frequency of minor salivary gland changes associated with tobacco as well as to measure the length and depth of ductal involvement in routine tissue sections of OED and OSCC. Materials and Methods: A total of 94 hematoxylin and eosinstained archival slides which included cases of well, moderate, and poorly differentiated OSCC and oral epithelial dysplasia were histopathologically evaluated to observe changes in different components of minor salivary gland. Ductal hyperplasia, ductal metaplasia, mucous pooling within duct, acinar degeneration, pattern of malignant cell invasion (single/clusters), inflammatory infiltrate, eosinophilic cuffing around the gland, and glandular/vascular involvement were evaluated in each slide and correlated with different grades of OSCC. Results: Ductal hyperplasia, inflammatory cell infiltrate, mucous pooling, and pattern of malignant cell infiltration came out to be statistically significant with the highest percentage of changes being observed in poorly differentiated squamous cell carcinoma > moderately differentiated squamous cell carcinoma> well differentiated squamous cell carcinoma>oral epithelial dysplasia. Further, the results of this study suggest that extension of dysplasia or squamous cell carcinoma from overlying oral epithelium along salivary gland ducts is an uncommon finding. Conclusion: Hence, histopathological interpretation for OED and OSCC should also include changes related to associated minor salivary gland tissue as detection and eradication of the putative precursors are the best way of decreasing the overall morbidity caused by tumors.

A juvenile pleomorphic adenoma of the palate.

Salivary gland tumors in children are rare; involvement of accessory salivary glands is exceptional. We report a case of pleomorphic adenoma of the palate in a child (an 8-year-old girl) addressed by her dentist for discovering a swelling in the palate. Clinical examination revealed a firm, non-tender, nodular swelling in the left hard palate, measuring 1.5 cm × 1.5 cm, situated adjacent to the upper left second molar. Physical examination did not show signs of inflammation or surface ulceration. Oral cavity computed tomography scan did not show bone lysis. The tumor was removed with negative margins. No recurrence was noted. We aim to describe the clinical, radiological features, as well as the management of this rare localization of pleomorphic adenoma.

Diagnostic role of minor salivary glands biopsy in Sjögren's syndrome: correlations between histology and autoimmunity in a large, monocentric cohort.

Introduction: Based on ACR/EULAR classification criteria, minor salivary glands biopsy (MSGB) is a useful diagnostic tool for the diagnosis of primary Sjögren's syndrome (SS). The main objective of our study was to evaluate the diagnostic role of MSGB, as well as to highlight correlations between histological findings and autoimmune profiles. Material and methods: We retrospectively evaluated histological and autoimmunity data from patients who underwent MSGB in our department in cases of suspected SS, from March 2011 to December 2018. Salivary gland samples were evaluated using Chisholm and Mason (CM) grading and the focus score (FS). Results: A total of 1,264 patients (108 males, 1,156 females) were included. The median age was 55.22 ±13.51 years (range: 15-87). In univariate binary logistic regression, CM ≥ 3 and FS ≥ 1 were significantly predicted by antinuclear antibodies (ANA), anti-extractable nuclear antigens (ENA) and anti-Ro/SSA titer as well as anti-La/SSB, anti-Ro/SSA, rheumatoid factor (RF) and anti-citrullinated protein antibodies (ACPA) positivity. In multivariate analysis, CM ≥ 3 and MSGB positivity were significantly associated with ANA titer; FS ≥ 1 was not associated with laboratory findings. A positive biopsy was associated with laboratory findings, as ANA and ENA titers, anti-Ro/SSA, anti-La/SSB, RF and ACPA positivity may discriminate patients with SS-related histological findings. Conclusions: Minor salivary glands biopsy is a useful tool to diagnose SS in cases of highly suggestive clinical symptoms but in the absence of a specific autoimmunity.

The Role of the Akt Signaling Pathway in Sjögren's Syndrome.

Primary Sjögren's syndrome (pSS) is a chronic autoimmune disorder with diverse clinical picture and high prevalence of B-cell non-Hodgkin lymphoma (NHL), that possibly raises from the chronic activation of B-cells. The mechanisms underlying the development of neoplasia in pSS remain elusive. Activated Akt/mTOR pathway is a uniform finding in cancer, whereas its significance in haematologic malignancies is highlighted by the plethora of inhibitors with promising therapeutic efficacy. PI3K-Akt activation has been involved in the TLR3-induced apoptosis of cultured salivary gland epithelial cells (SGECs), whereas upregulated expression of the phosphorylated ribosomal S6 protein (pS6), an end-result of PI3K signalling, has been detected in the infiltrating T and B lymphocytes at the MSG lesions of pSS patients; nevertheless, without specifying if this was mediated by the Akt/mTOR or Ras/ERK pathways. To this end, the role of Akt/mTOR pathway in pSS and associated lymphomagenesis, will be investigated by the immunohistochemical detection of the entire and phosphorylated protein forms of Akt kinase and two of its substrates, namely the FoxO1 transcription factor and the proline-rich Akt substrate of 40-kDa (PRAS40) in MSGs of pSS patients with variable histological and clinical phenotype, as well as sicca-complaining controls. Subsequently, the role of this pathway will be evaluated in in-vitro inhibition experiments, studying the effect of specific inhibitors in the phenotype, function, and interaction of SGECs and B cells. The current proposal is expected to promote the understanding of pSS pathogenesis, enlighten the mechanisms underlying related lymphomagenesis and possible therapeutic targets.

Predictive factors for survival and treatment outcomes of patients with minor salivary gland malignancies: a retrospective study.

INTRODUCTION: Aim of this study was to explore the incidence, pathology, clinical behaviour and evaluate factors predictive on survival and treatment outcomes in a cohort of patients with minor salivary gland (MiSG) malignancies treated at a single center over a period of 25 years. MATERIALS AND METHODS: Patients who had received primary treatment for MiSG malignancy during 25 years observation period were identified. Outcomes that were evaluated were overall survival (OS), disease specific survival (DSS), recurrence free survival (RFS), locoregional recurrence free survival (LRFS) and distant metastasis free survival (DFS). RESULTS: A total of 88 patients with MSG malignancies were included in the study. The most common location for MiSG malignancies was the oral cavity (65 tumors; 77%). Cumulative OS for 5 and 10 year follow up period was 82% and 62% respectively. Cumulative DSS for 5 and 10 year follow up period was 85% and 73% respectively. Twenty one (23%) patients developed distant metastases during follow-up. High-grade pathology and tumor stage were significant variables on multivariate analysis for all survival and treatment outcomes. CONCLUSIONS: Minor salivary gland malignancies are minor only by name. Tumor histological grade, AJCC tumor stage and pT stage were the strongest predictive factors for survival and treatment outcomes. The elective neck dissection could be considered therapeutic approach for selected cases of high grade MiSG malignancies. Distant metastases were the main cause of death and treatment failure.

[Evaluation of morphological activity of primary Sjogren's syndrome on bioptates of minor salivary glands]. / Otsenka morfologicheskoi aktivnosti bolezni Shegrena po bioptatam malykh slyunnykh zhelez.

BACKGROUND: The results of the morphological study of the minor salivary glands can be used to assess the activity of the primary Sjogren's syndrome and to decide on adequate therapy.The existing protocol of The Sjögren's International Clinical Collaborative Alliance (SICCA) prescribes the methodology for examining biopsy specimens for suspected Sjögren's disease, however, experts interpret data from the analysis of histological preparations differently. OBJECTIVE: To identify morphological forms of sialadenitis, as well as to determine the focus score in Russian patients based on the retrospective analysis of minor salivary glands biopsies of patients with primary Sjogren's syndrome. MATERIAL AND METHODS: Biopsies of minor salivary glands were studied in 92 patients with primary Sjogren's syndrome and 42 patients without rheumatic disease. RESULTS: Focal lymphocytic sialadenitis was detected in 69 patients with primary Sjogren's syndrome. The focus score in patients with primary Sjogren's syndrome was 7.32 (2.8-14.17). In patients without rheumatic diseases, this index was 0.48 (p<0.05). Patients with confluent lymphocytic foci need immunohistochemical examination and dynamic monitoring to exclude lymphoproliferative diseases. CONCLUSION: The index of morphological activity of sialadenitis in primary Sjogren's syndrome ranges from 2.8 to 14.17 and reflects the activity of the underlying disease.It should be taken into account in the diagnosis and prescription of adequate therapy. Further study of the correlations of morphological and clinical and laboratory parameters will lead to clarification of the criterion signs of the disease.

Locational and Clinical Varieties of Warthin Tumor: Two Rare Case Presentations.

Warthin tumor (WT) is the second most common benign tumor of the parotid gland with male predominance and is seen in the elderly population in the left mandibular and cervical regions. Extraparotid WT rarely presents as a mass in the right cervical region in middle-aged adults. Here, we present two extraordinary cases of WT seen in middle age, both in the right cervical region. The first is a 50-year-old man with an asymptomatic neck mass, and the second is a 60-year-old woman with a fast-growing mass with enhancement in positron emission tomography-computed tomography (PET-CT) with suspicions of malignancy.

Cheilitis Glandularis: A clinicopathologic study with emphasis on etiopathogenesis.

OBJECTIVES: Cheilitis Glandularis (CG) is an uncommon entity of obscure etiology. A cases series is presented with emphasis on etiopathogenesis. MATERIALS AND METHODS: Fourteen CG cases were analyzed according to their demographic and clinicopathologic characteristics. RESULTS: The mean age of the patients with CG was 68.1 years, while a male-to-female ratio of 1.8:1 was observed. One or more potential causative factors were identified for each patient, including long-term smoking (9 cases), xerostomia (4 cases), cosmetic filler injections (2 cases), and actinic cheilitis (1 case). The lesions were located on the lips, buccal mucosa, or both in 7, 2, and 5 cases, respectively. Multiple submucosal nodules with dilated ductal orifices and mucous or purulent discharge were observed in all cases. Histopathologically, ductal ectasia with metaplasia, intraductal mucin, and chronic or mixed inflammation were noted, as well as pools of hyaluronic acid in 2 cases with a history of cosmetic filler injections. CONCLUSIONS: CG etiopathogenesis is probably multifactorial. Reduced salivary flow rate and increased viscosity of saliva, potentially caused by long-term smoking, diabetes mellitus, and drug-induced xerostomia, may participate in the initial pathogenesis, while local irritants, for example, poor oral hygiene and local trauma, may further contribute to the development and aggravation of the condition.

Lymphoepithelial Subtype of Oral Squamous Cell Carcinoma: Report of an EBV-Negative Case and Literature Review.

Lymphoepithelial carcinoma (LEC) of the oral mucosa is a rare histopathologic subtype of squamous cell carcinoma (SCC), which shares morphologic similarities with nasopharyngeal carcinoma (NPC), non-keratinizing undifferentiated subtype. The admixture of neoplastic epithelial tumor cells and a dense lymphoplasmacytic infiltrate makes microscopic diagnosis challenging. LEC etiopathogenesis has been variably associated with Epstein-Barr virus (EBV) infection, depending on the specific anatomic location and racial predilection, with a higher incidence in endemic populations. Although described in several subsites of the head and neck region, including the major salivary glands, the oral mucosa is considered an infrequent location for LEC development, deriving either from minor salivary glands (MSGs) or the surface epithelium. Herein, we report a rare case of an EBV-negative LEC arising from the oral surface epithelium, presenting as gingival swelling, and review the pertinent English-language literature, which revealed only 26 previously reported oral LECs. Our case is only the fourth oral LEC originating from the surface epithelium and the first one to affect the gingiva.