Basal cell adenoma and basal cell adenocarcinoma of the parotid gland: clinical findings and surgical outcomes in a single-institution study.

BACKGROUND: Basal cell adenoma (BCA) is a rare benign tumor within the salivary glands. Basal cell adenocarcinoma (BCAC), the malignant counterpart of BCA, is also an exceedingly rare tumor with very limited clinical studies conducted. This study aims to investigate the clinical characteristics, demographics, and surgical outcomes of patients diagnosed with BCA and BCAC within the parotid gland. METHODS: A retrospective analysis from May 2003 to August 2023 was performed for all patients undergoing parotidectomy for masses. Retrospective data on gender, age, tumor characteristics, and outcomes were collected. Surgical approaches, including negative margin attainment, capsule removal, and histological diagnosis, were also detailed. RESULTS: The study included 1268 patients who underwent parotidectomy, resulting in 81 cases of BCA and 7 cases of BCAC. BCA patients, with a mean age of 55.1 years, showed diverse age distribution and predominantly presented in the 50s. In BCAC cases, seven female patients exhibited a predominant location in the deep lobes. FNA revealed BCAC in three out of seven cases, and subsequent parotidectomy was performed, resulting in no observed recurrences or metastases. CONCLUSION: This study reports the largest number of BCA cases from a single institution and provides comprehensive insights into the demographics, tumor characteristics, and clinical outcomes of both BCA and BCAC. Although further research should be conducted, based on clinical follow-up results, appropriately including the capsule in the tumor excision indicates favorable outcomes, especially when the tumor size is not large.

Molecular Aspects of Mucoepidermoid Carcinoma and Adenoid Cystic Carcinoma of the Salivary Gland.

BACKGROUND: Salivary gland tumors (SGTs) are rare and highly heterogeneous lesions, making diagnosis a challenging activity. In addition, the small number of studies and samples evaluated difficults the determination of prognosis and diagnosis. Despite the solid advances achieved by research, there is still an intense need to investigate biomarkers for diagnosis, prognosis and that explain the evolution and progression of SGTs. METHODS: We performed a comprehensive literature review of the molecular alterations focusing on the most frequent malignant SGTs: mucoepidermoid carcinoma and adenoid cystic carcinoma. RESULTS: Due to the importance of biomarkers in the tumorigenenic process, this review aimed to address the mechanisms involved and to describe molecular and biomarker pathways to better understand some aspects of the pathophysiology of salivary gland tumorigenesis. CONCLUSIONS: Molecular analysis is essential not only to improve the diagnosis and prognosis of the tumors but also to identify novel driver pathways in the precision medicine scenario.

Proceedings of the 2024 North American Society of Head and Neck Pathology Companion Meeting, Baltimore, MD, March 24, 2024: Navigating Ancillary Studies in Basaloid/Blue Salivary Tumors.

BACKGROUND: Basaloid salivary tumors can demonstrate significant morphologic overlap and be challenging to diagnose. METHODS: A review of select ancillary studies in basaloid salivary tumors was performed. RESULTS: A number of immunohistochemical stains, including PLAG1, HMGA2, ß-catenin, MYB, and RAS Q61R, have been more recently incorporated into the diagnostic workup of basaloid salivary tumors. CONCLUSIONS: Although reported variability in their performance has perhaps limited their widespread adoption, these immunohistochemical studies can nevertheless be useful in supporting pathologic diagnoses, particularly when considered in more specific differentials or when used as a panel with other markers.

Adenoid Cystic Carcinoma, Clinical Presentation, Current Treatment and Approaches Towards Novel Therapies.

Adenoid cystic carcinoma (AdCC) is a rare cancer originating from secretory glands with unknown aetiology. It is one of the most dominant malignant salivary tumours (MST). However, it can arise in other areas of the head and neck region and in secretory glands outside this area. It occurs at all ages, but is more frequent between 50-70 years of age and more common in females than in males. The symptoms of AdCC are generally unspecific and the clinical diagnosis of AdCC maybe challenging, partially due to its heterogenous histopathology and indolent growth. Moreover, there is a lack of good prognostic markers, and due to its rarity, it is difficult to predict which therapeutic methods are the most optimal for each patient, especially since very late recurrences occur. This review presents some major characteristics of AdCC and some current treatments for this disease.

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.

Case of clear-cell oncocytoma of parotid gland and literature review.

Oncocytoma is a benign tumor of the salivary gland. Its incidence is very low and very seldom documen-ted in literature. Clear-cell dominant oncocytoma is even less common. The tumor's clinical symptoms and imaging results are nonspecific, so distinguishing other salivary gland tumors (such as oncocytic carcinoma) from clear-cell renal carcinoma is difficult, possibly leading to misdiagnosis and maltreatment. Here, a case of clear-cell dominant oncocytoma was presented, and the relevant literature was evaluated to investigate the diagnosis and management of clear-cell dominant oncocytoma.

Molecular testing in salivary gland cytopathology: A practical overview in conjunction with the Milan system.

Recently, significant advances in the molecular characterization of salivary gland neoplasms have facilitated the classification and diagnosis of specific diagnostic entities. In the highly challenging diagnostic scenario of salivary malignancies, molecular testing is increasingly being adopted in routine practice to refine the cytological diagnosis of salivary lesions. Here, we reviewed the most recent evidence in the field of salivary glands molecular cytopathology.

Pulmonary salivary gland tumor-hyalinizing clear cell carcinoma: a literature review.

Primary pulmonary hyalinizing clear cell carcinoma (HCCC) is a very rare lung tumor that accounts for less than 0.09% of all primary lung tumors and has no specific epidemiology. The correct diagnosis requires imaging, laboratory, pathological, immunohistochemical, and molecular examination. The most typical feature of pulmonary HCCC is the clear cell component with clear stroma. In addition, the fusion gene EWSR1::ATF1 due to t(12;22)(q13;q12) is essential for the pathological diagnosis of pulmonary HCCC. The main treatment for pulmonary HCCC is surgery. This review focus on the pathological features, immunohistochemical examination, mutation analysis and treatment of pulmonary HCCC.

Rapid on-site evaluation improves diagnostic performance of fine-needle aspiration cytology for salivary lesions: Comparison of data from two cancer centers in southern China.

OBJECTIVES: To evaluate the diagnostic performance of Milan system for reporting salivary gland cytopathology (MSRSGC) in two southern China tertiary cancer centers and investigate the impact of rapid on-site evaluation (ROSE) on FNAC performance. MATERIALS AND METHODS: Five hundred and forty-nine patients who underwent FNAC for salivary lesions with surgical follow-up from two centers were enrolled in this retrospective cohort study. All slides were recategorized using MSRSGC after consensus on diagnostic criteria for each category. The diagnostic performance of FNAC for salivary lesions was evaluated and compared and the impact of ROSE on FNAC performance was analyzed. RESULTS: The distribution of cases per category based on the MSRSGC criteria in the whole series was as followed: ND 49 (8.9%), NN 76 (14.4%), BN 262 (47.7%), AUS 20 (3.6%), SUMP 43 (7.8%), SM 21 (3.8%), M 78 (14.2%). The SUMC series had significantly more ND distributions than JXCH did (16.2% vs. 0, p = .000). Risk of malignancy for each category in the total series was as followed: 42.9% for ND, 9.2% for NN, 3.8% for BN, 30.0% for AUS, 23.3% for SUMP, 81.0% for SM, and 94.9% for M. When ND and AUS/SUMP were excluded, the sensitivity, specificity, PPV, NPV, and accuracy were 84.0%, 97.1%, 89.9%, 95.1%, and 94.0%, respectively; sensitivity, specificity, PPV, NPV, and accuracy were comparable between the two centers. CONCLUSIONS: FNAC using MSRSGC provides a good tool in preoperative evaluation for salivary lesions in southern China. ROSE improves its diagnostic performance by reducing the ratio of the ND category.

[Correlation of MYB/NFIB gene fusion with the grade and prognosis of head and neck adenoid cystic carcinoma and the concordance of two detection methods].

Objective: To explore the correlation between MYB/NFIB gene fusion and clinicopathological features such as tumor grade and prognosis of head and neck adenoid cystic carcinoma (ACC), and to assess the concordant rate of fluorescent in situ hybridization (FISH) with MYB and NFIB immunohistochemistry. Methods: FISH detection of MYB/NFIB gene fusion was performed on 48 head and neck ACC cases and 15 non-ACC salivary gland tumors at National Cancer Center/National Clinical Research Center for Cancer/Cancer Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing, China during April 2014 and January 2020. ACC cases were divided into grade Ⅰ-Ⅱ, grade Ⅲ and high-grade transformation, according to pathological grading criteria. Prognosis, FISH results and other clinicopathological characteristics were analyzed. MYB and NFIB immunohistochemistry was performed on the 48 ACC and 15 non-ACC cases. The diagnostic accuracy of FISH and immunohistochemistry was compared. Results: FISH detected MYB/NFIB gene fusion in 41.7% (20/48) of the ACC. Its positive rate was inversely correlated with higher pathological grades (P=0.036). The higher histological grade was linked to worse progression-free survival (P=0.024), whereas there was no correlation between the status of gene fusion detected by FISH and progression-free survival (P=0.536). FISH didnot detect MYB/NFIB gene fusion in 15 non-ACC salivary gland tumors The specificity of diagnosing ACC is 100% for both FISH detection of gene fusion and immunohistochemical detection of MYB expression. However, the sensitivity for both methods was only about 41.7%, respectively. By combining FISH and MYB immunohistochemistry, the sensitivity for diagnosing ACC was increased to 66.7%. Conclusions: MYB/NFIB gene fusion has a lower detection rate in grade Ⅲ ACC and high-grade transformation ACC. Meanwhile gene fusion status is not correlated with prognosis. The sensitivity for diagnosing ACC can be improved by combining FISH and MYB immunohistochemistry.

Gastric Metastasis from Salivary Duct Carcinoma Mimicking Scirrhous Gastric Cancer.

A 59-year-old man underwent submandibular gland excision for salivary duct carcinoma (SDC). One year later, esophagogastroduodenoscopy indicated gastric diffuse mucosal thickening with luminal contraction, mimicking scirrhous gastric carcinoma. Biopsy specimens showed dense proliferation of neoplastic cells expressing androgen receptor and human epidermal growth factor 2, indicating SDC. Gastric diffuse infiltrative metastasis is generally characteristic of gastric metastasis from invasive ductal carcinoma, which shows histologic features similar to SDC. This is the first known report of gastric diffusely infiltrating metastasis in an SDC patient. Rapidly progressing, diffuse gastric wall thickening should also be considered indicative of salivary tumor-associated gastric metastasis.

The Milan System for Reporting Salivary Gland Cytopathology: the experience of a tertiary cancer center with emphasis on non-mucinous cysts and improving diagnostic results.

INTRODUCTION: The Milan System for Reporting Salivary Gland Cytopathology (MSRSGC) was introduced in 2018 to standardize cytology reporting and guide patient treatment. We aimed to evaluate the utility of this system applied to patients at our cancer center. MATERIALS AND METHODS: We retrospectively reviewed cases of salivary gland fine-needle aspirations (FNAs) performed in our institution (2019-2022). All were performed by radiologists and immediately assessed for specimen adequacy. The cytologic findings were classified into the MSRSGC except for non-mucinous cystic contents (NMCC) where the lesion was radiologically consistent with a cyst and totally collapsed after FNA. Such lesions were categorized as non-neoplastic (NN) instead of non-diagnostic (ND). The cytologic findings were compared to corresponding histologic findings (212 available cases), and the risk of malignancy was calculated. RESULTS: Five hundred five FNAs were categorized as: 25 (4.95%) ND; 86 (17.03%) NN, of which 39 were NMCC; 9 (1.78%) atypia of undetermined significance; 138 (27.33%) benign neoplasms; 57 (11.29%) salivary gland neoplasm of undetermined malignant potential; 16 (3.17%) suspicious for malignancy; and 174 (34.46%) malignant. The risk of malignancy rates for the following categories were: ND, 40%; NN, 25%; atypia of undetermined significance, 0%; benign neoplasms, 1%; salivary gland neoplasm of undetermined malignant potential, 54.54%; suspicious for malignancy, 90.9%; and malignant, 100%. Thirty-one NMCC with available follow-up resolved/remained stable. CONCLUSIONS: Our results validate the reproducibility of the MSRSGC applied in our cancer center. Based on the benign course of cysts with NMCC, we propose that such cases be categorized as NN, provided the cyst is totally resolved after FNA.

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.

Concordance of fine needle aspiration cytology and final histology of salivary gland tumours.

BACKGROUND: Fine needle aspiration cytology (FNAC) is a widely used diagnostic tool to evaluate salivary gland tumours. The Milan system for reporting salivary gland cytopathology allows for standardisation and facilitates cytologicalhistological correlation. However, FNAC findings can still pose a diagnostic challenge. The accuracy of FNAC should be assessed at each centre. The aim of this study was to assess the concordance of FNAC and final histology of salivary gland tumours in three academic hospitals affiliated with the University of Pretoria, South Africa. METHODS: The study was a cross-sectional retrospective analytical study of 214 patients who underwent an operation for salivary gland tumours. All patients with FNAC and histology results between 2007-2017 were included. Patients were recruited from three University of Pretoria, South Africa, affiliated hospitals: Steve Biko Academic, Kalafong Provincial Tertiary and Tembisa Provincial Tertiary Hospital. RESULTS: Of the 214 patients with salivary gland tumours, the majority were located in the parotid gland (56.1%). Pleomorphic adenoma was the most common tumour (62.6%). The FNAC sensitivity, specificity and diagnostic accuracy (receiver operating characteristic) were 92.7%, 98.1% and 0.95 respectively. The concordance between salivary gland tumour FNAC and final histology was 96.95% with a Cohen's kappa coefficient of 0.91 (p= 0.0001). CONCLUSION: There is strong concordance between FNAC and histology of salivary gland tumours. FNAC is an accurate, minimally invasive diagnostic tool with high sensitivity and specificity. It provides the clinician with a reliable preoperative diagnosis determining whether the salivary gland tumour is benign or malignant.

[Salivary gland neoplasms]. / Tumeurs des glandes salivaires.

SALIVARY GLAND NEOPLASMS. Salivary gland tumors represent a heterogeneous group of lesions, with various anatomical locations. The most frequent site of involvement is the parotid, and the most frequent histology is pleomorphic adenoma, a benign tumor with the potential for recurrence and malignant transformation. Approximately one third of tumors are malignant, and the prognosis depends on the tumor histology and histoprognostic criteria. The diagnostic strategy focuses on determining the benignity or malignancy of these lesions to choose appropriate treatment. It is based on multiparametric magnetic resonance imaging (MRI) [associating morphological images with diffusion and perfusion sequences], and on fine needle aspiration. Surgery is almost always indicated to allow histological confirmation and treatment of the disease. The extent of resection, the need for elective neck dissection, and the indication of adjuvant therapy (postoperative radiotherapy) depend on the tumor histology.

TUMEURS DES GLANDES SALIVAIRES. Les tumeurs des glandes salivaires représentent un groupe hétérogène de lésions, de localisations anatomiques variées. L'atteinte la plus fréquente est parotidienne, et l'histologie est le plus souvent l'adénome pléomorphe, tumeur bénigne avec un potentiel de récidive et de transformation maligne. Environ un tiers des tumeurs sont malignes ; le pronostic dépend alors de l'histologie tumorale et de critères histopronostiques. La démarche diagnostique s'attache à déterminer la bénignité ou la malignité de ces lésions pour un choix thérapeutique adapté. Elle se fonde sur l'imagerie par résonance magnétique (IRM) multiparamétrique (associant des images morphologiques à des séquences de diffusion et de perfusion) et sur la cytoponction à l'aiguille fine. La chirurgie est presque toujours indiquée pour permettre la confirmation histologique et traiter la maladie. L'étendue de la résection, la réalisation d'un curage cervical et d'un traitement adjuvant (radiothérapie postopératoire) dépendent de l'histologie tumorale.

Secretory carcinoma of salivary glands with NTRK3 break-apart molecular rearrangement: Potential misdiagnosis with mucoepidermoid carcinoma.

A woman presented a right submandibular gland lesion with cytologic diagnosis of mucoepidermoid carcinoma. Patient underwent sialoadenectomy en bloc with supraomohyoid neck dissection. Positivity for ETV6-NTRK3 genes fusion on surgical sample led to final diagnosis of secretory carcinoma (SC). Secretory carcinoma has been renamed by WHO in 2017 from mammary-analogue-secretory carcinoma (MASC). Only 649 have been reported until 2019. While cytologic alteration are shared with other neoplasms as the acinic cell and mucoepidermoid carcinomas, ETV6-NTRK3 rearrangement is pathognomonic of SC. Although usually indolent and with low-stage presentation, SC has higher rate of local recurrences and nodal involvement than ACC. Surgical treatment represent the gold standard. Real prevalence of SC is probably underestimated due to the recent WHO 2017 reclassification. While cytologic analysis does not allow to discriminate SC from other malignancies, chromosomal examination is recommended. When low-grade SC is diagnosed, complete surgical resection assures good prognosis.

Basal cell adenoma and pleomorphic adenoma of the parotid gland: a single center experience.

BACKGROUND: Basal cell adenoma (BCA) and pleomorphic adenoma (PA) are among the most common benign neoplasms of the salivary glands. The aim of this study was to analyze and compare the diagnosis, treatment, and recurrence rate of these two different types of parotid benign tumors. METHODS: A retrospective analysis of all cases of parotid gland BCA and PA surgically treated between January 1, 1990, and December 31, 2019, was performed at our university. RESULTS: A total of 349 patients were enrolled in the present study, 311 of which (89.1%) were affected by PA, and 38 patients (10.9%) by BCA. The most frequently performed surgery was partial parotidectomy for both groups (85.9% in PA and 65.8% in BCA). Perioperative complications - often transient and of short duration - occurred within 48 hours of surgery and were observed in 30.6% of PA patients and in 18.4% of BCA patients; furthermore, recurrences were noticed in 19 PA patients (6.2%) and in 3 BCA patients (7.9%) (rates in range with the available literature data). CONCLUSIONS: To the best of our knowledge, this study is one of the largest single-center series in the literature comparing diagnosis, treatment, recurrence rate and clinical-pathological features of two different types of benign parotid gland tumors, BCA, and PA.

A systematic review on the role of biomarkers in liquid biopsies and saliva samples in the monitoring of salivary gland cancer.

BACKGROUND: Salivary gland cancer is a rare disease, and approximately 20% of tumors in the salivary glands are malignant. Reliable biomarkers may have a role in monitoring salivary gland cancer. AIM: To review the current literature on the role of biomarkers in liquid biopsies and saliva samples in the monitoring of salivary gland cancer. MATERIALS AND METHOD: This study systematically reviewed the literature on studies detecting salivary gland cancer by biomarkers in liquid biopsies and saliva samples by systematically searching PubMed and Embase between 1 January 2013 and 7 March 2023. RESULTS: Five studies covering 64 malignant cases of salivary gland cancer were included, which considered inflammatory biomarkers or markers of genetic material in either blood or saliva. In saliva, there were demonstrated elevations of CA-19-9 in malignant cases, and elevations of miRNA in malignant and benign cases. In blood, there were demonstrated elevations of IL-33 in malignant and benign cases, elevations of ctDNA in malignant cases, and elevations of CTC in malignant cases. CONCLUSION AND SIGNIFICANCE: The studies indicate that there is potential in the detection method. The studies detecting genetic material by liquid biopsies showed the most promising results. At present, there is still progression to be made before the method can be implemented for diagnostic use.