Salivary gland secretory carcinoma presenting as a cervical soft tissue mass: a case report.

BACKGROUND: Secretory carcinoma (SC) has been described as a distinct salivary gland tumor in the fourth edition of the World Health Organization (WHO) classification of head and neck tumors. SC is generally considered as a slow-growing low-grade malignant tumor, while several cases have been reported with high-grade features, and even metastases in the literature up until now. In this article, a soft tissue SC case is discussed with high-grade microscopic features and neural invasion. A review of the salivary gland SC cases with aggressive behavior is also debated. CASE PRESENTATION: A 65-year-old Caucasian man presented with a left neck mass for the past six months. The imaging studies demonstrated a very large cystic cervical mass (46 × 23 mm) with papillary projections in the anterolateral aspect of the left neck zone Vb. He underwent left radical neck dissection (level I-V) and was followed up for 12 months with the diagnosis of Secretory carcinoma. CONCLUSION: Although SC generally has a good outcome, multiple recurrences and unusual metastases may occur, which should be considered by either the pathologists or clinicians.

Post-operative radiotherapy in adenoid cystic carcinoma of salivary glands versus surgery alone: what is the evidence about survival and local control? A systematic review and meta-analysis.

BACKGROUND: Adenoid cystic carcinoma of the salivary glands is a relatively rare malignancy characterized by slow growth and a poor prognosis, and effective treatments remain challenging to identify. This systematic review, following the PRISMA guidelines, aimed to analyze the potential benefits of post-operative radiotherapy in terms of local control of recurrences and survival advantages when compared with surgery alone in patients with adenoid cystic carcinoma. METHODS: A comprehensive systematic review was conducted by searching the MEDLINE, Cochrane, EMBASE, and OVID databases from January 1999 to July 2022. The goal was to identify articles comparing surgery alone with surgery plus postoperative radiotherapy for adenoid cystic carcinoma of the salivary glands. Downs and Black Checklist was used to assess the methodological quality and risk of bias of each included study. The data analysis was performed using Review Manager version 5.4.1. RESULTS: This review included 8 studies comprising a total of 3103 patients, divided based on the analyzed outcomes. The pooled odds ratio for overall survival at 5 years was 0.87 (95% confidence interval 0.43-1.76, p = 0.70), and at 10 years was 1.23 (95% confidence interval 0.69-2.16, p = 0.48). In both cases, no statistically significant differences were observed. However, the pooled odds ratio for local control at 5 years was 3.37 (95% confidence interval 1.35-8.42, p = 0.009), providing strong support for the use of post-operative radiation. CONCLUSIONS: The findings from the meta-analysis suggest that post-operative radiotherapy significantly improves local control in patients with adenoid cystic carcinoma. However, there was no statistically significant increase in survival at 5 and 10 years. It is essential to note that the quality of the studies included in this meta-analysis ranged from fair to poor. To better clarify the indications for post-operative radiotherapy, future high-quality research is needed, particularly with improved stratification of patient groups. Additionally, it is important to recognize that achieving local control in adenoid cystic carcinoma is crucial for enhancing the overall quality of life for patients. We acknowledge that this review was not registered in the PROSPERO database, and the data pooling was conducted using a random effects model.

Oncologic Safety of Close Margins in Patients With Low- to Intermediate-Grade Major Salivary Gland Carcinoma.

Importance: Postoperative radiation therapy for close surgical margins in low- to intermediate-grade salivary carcinomas lacks multi-institutional supportive evidence. Objective: To evaluate the oncologic outcomes for low- and intermediate-grade salivary carcinomas with close and positive margins. Design, Setting, and Participants: The American Head and Neck Society Salivary Gland Section conducted a retrospective cohort study from 2010 to 2019 at 41 centers. Margins were classified as R0 (negative), R1 (microscopically positive), or R2 (macroscopically positive). R0 margins were subclassified into clear (>1 mm) or close (≤1 mm). Data analysis was performed from June to October 2023. Main Outcomes and Measures: Main outcomes were risk factors for local recurrence. Results: A total of 865 patients (median [IQR] age at surgery, 56 [43-66] years; 553 female individuals [64%] and 312 male individuals [36%]) were included. Of these, 801 (93%) had parotid carcinoma and 64 (7%) had submandibular gland carcinoma, and 748 (86%) had low-grade tumors and 117 (14%) had intermediate-grade tumors, with the following surgical margins: R0 in 673 (78%), R1 in 168 (19%), and R2 in 24 (3%). Close margins were found in 395 of 499 patients with R0 margins (79%), for whom margin distances were measured. A total of 305 patients (35%) underwent postoperative radiation therapy. Of all 865 patients, 35 (4%) had local recurrence with a median (IQR) follow-up of 35.3 (13.9-59.1) months. In patients with close margins as the sole risk factor for recurrence, the local recurrence rates were similar between those who underwent postoperative radiation therapy (0 of 46) or observation (4 of 165 [2%]). Patients with clear margins (n = 104) had no recurrences. The local recurrence rate in patients with R1 or R2 margins was better in those irradiated (2 of 128 [2%]) compared to observed (13 of 64 [20%]) (hazard ratio [HR], 0.05; 95% CI, 0.01-0.24). Multivariable analysis for local recurrence found the following independent factors: age at diagnosis (HR for a 10-year increase in age, 1.33; 95% CI, 1.06-1.67), R1 vs R0 (HR, 5.21; 95% CI, 2.58-10.54), lymphovascular invasion (HR, 4.47; 95% CI, 1.43-13.99), and postoperative radiation therapy (HR, 0.10; 95% CI, 0.04-0.29). The 3-year local recurrence-free survivals for the study population were 96% vs 97% in the close margin group. Conclusions and Relevance: In this cohort study of patients with low- and intermediate-grade major salivary gland carcinoma, postoperative radiation therapy for positive margins was associated with decreased risk of local recurrence. In isolation from other risk factors for local recurrence, select patients with close surgical margins (≤1 mm) may safely be considered for observation.

The characteristics of Polish patients with salivary gland tumors: a ten-year single-center experience.

OBJECTIVES: The study aims to provide insights into the characteristics of Polish patients with different salivary gland pathologies. MATERIALS AND METHODS: This is a retrospective study conducted at a single center, including patients who underwent surgery for salivary gland pathologies between 2012 and 2022. RESULTS: This study included 239 patients who underwent surgery for salivary gland tumors or inflammatory diseases. Malignant tumors were diagnosed in 9.8% of participants, while 64% had benign tumors and 21% suffered from inflammation. The occurrence of complications after surgery was relatively low, with 9.9% of participants experiencing slight facial weakness or mild dysfunction, and 3% experiencing complete paralysis of the facial nerves. Significant differences were observed between patients with cancers and those with benign tumors and inflammation in terms of age. Cancers were more common in females (67% vs. 33%) and predominantly localized in the parotid glands (95%). CONCLUSION: Benign tumors, such as Warthin's tumors and polymorphous adenoma, were predominantly found in the parotid glands of patients aged 39-72 years. On the other hand, inflammatory diseases were primarily localized within the submandibular glands of males aged 40-68 years. Additionally, the presence of a malignant tumor was associated with longer hospitalization periods related to surgery and a higher risk of severe complications. CLINICAL RELEVANCE: This study on Polish patients with salivary gland tumors provides valuable clinical insights that can aid in diagnosis, treatment planning, patient counseling, and further research in the field of oncology. It contributes to the overall understanding of salivary gland tumors, potentially benefiting both patients and healthcare providers.

Malignant Minor Salivary gland neoplasms ofLarynx: Our Experience.

<br><b>Introduction:</b> Malignant minor salivary gland tumors are rare, accounting for fewer than 1% of all laryngeal cancers.</br> <br><b>Aim:</b> This study aims to share our experiences regarding clinical, radiological, pathological profiles and their management.</br> <br><b>Materials and methods:</b> The current study reviews 11 cases of malignant minor salivary gland tumors of the larynx treated surgically at our Institute between 2005 and 2019.</br> <br><b>Results:</b> The mean age of the patients was 54 years (range 38-75 years) with six females and five males in the series (1.2:1). Subglottis and trachea were the sites of origin in 54% of the cases, and hoarseness with dyspnea were the most common presenting symptoms. There were nine Adenoid cystic and two Mucoepidermoid carcinoma patients. Surgery was the primary mode of treatment.</br> <br><b>Conclusions:</b> Most of the larynx's malignant minor salivary gland tumors are submucosal in origin. The outcome and prognosis vary considerably based on the tumor's histology, grade, and stage.</br>.

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.

Is radical radiotherapy with/without surgery an effective treatment in the lymphoepithelial carcinoma of the salivary gland？.

BACKGROUND: There is limited information of radical radiotherapy (RT) on lymphoepithelial carcinoma of salivary gland (LECSG) regarding to the rarity of the disease. We conducted this retrospective study that evaluated the feasibility and efficacy of radical RT with/without surgery in LECSG. METHODS: We retrospectively reviewed patients that were pathologically diagnosed of LECSG and had definite or suspicious residual disease. The prescribed dose given to P-GTV and/or P-GTV-LN was 66 to 70.4 Gy. The clinical target volume (CTV) involved ipsilateral salivary gland and corresponding lymph node drainage area. RESULTS: A total of 56 patients were included. With a median follow-up of 60 months (range: 8 to 151 months), the 1-, 5-, and 10-year progression-free survival (PFS) rates were 94.6%, 84.7% and 84.7%; locoregional progression-free survival (LRPFS) rates were 98.2%, 87.4% and 87.4%; distance metastasis-free survival (DMFS) rates were 94.6%, 86.7% and 86.7%; and overall survival (OS) rates were 98.2%, 92.4% and 89.0%, respectively. A total of 7 patients without surgery were included. All patients were alive and only one patient experienced failure of distant metastasis four months after RT. The results of univariate analysis showed that compared with N stage, the number of positive lymph nodes (2 positive lymph nodes) was better prognostic predictor especially in PFS. There were no treatment-related deaths and most toxicities of RT were mild. CONCLUSIONS: Radical RT with/without surgery in LECSG for definite or suspicious residual disease is feasibility and efficacy. Most toxicities of RT were mild due to the target volume involved ipsilateral area.

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.

Feline minor salivary gland adenocarcinoma: retrospective case series and literature review.

CASE SERIES SUMMARY: Salivary gland adenocarcinoma, of major or minor salivary gland origin, is an uncommon tumor in cats. This article describes the clinical features, morbidity and survival rates of four cats with salivary gland adenocarcinoma arising from minor salivary gland tissue. Medical records from a private multicenter dentistry and oral surgery practice were reviewed for the period between 2007 and 2021. Four cats were included in this retrospective case series study, with oral masses on either the right or left caudal mandibular labial buccal mucosa. The inclusion criteria included a diagnosis of salivary gland adenocarcinoma in an anatomical location with lack of involvement of a major salivary gland, complete medical history and a follow-up of at least 6 months. The age range of the cats was 9-15 years; three of the cats were castrated males and one was a spayed female. Curative intent surgery was performed in three cats, whereas palliative surgery (debulking) owing to extensive soft tissue invasion was performed in one cat. Survival times were in the range of 210-1730 (mean 787) days. All four cats were euthanized owing to local recurrence and decreased quality of life, regardless of treatment modality. RELEVANCE AND NOVEL INFORMATION: There are limited documented studies reporting the prevalence of salivary gland neoplasia affecting minor disseminated glands in the oral cavity of feline patients. Salivary gland adenocarcinoma should be a differential in cats presenting with caudal labial masses. Surgical resection has been the recommended treatment for salivary gland neoplasia of major salivary gland origin. According to this current case series, we propose that early aggressive surgical treatment with wide surgical margins should be performed for cats with salivary gland adenocarcinoma of minor salivary gland origin. Surgery increased the quality and duration of life; however, each patient was euthanized owing to local recurrence and morbidity.

Milan system for reporting salivary gland cytology in diagnosis and surgery of parotid gland lesions.

INTRODUCTION: A cytology report is always a challenge for both head and neck surgeons and cytopathologists to diagnose and manage parotid gland (PG) diseases, because of the various similar features between the lesions. OBJECTIVES: The present study was conducted to assess our practice using the Milan System for Reporting Salivary Cytopathology (MSRSGC) and to evaluate the risk of malignancy (ROM) in different categories. PATIENTS AND METHODS: The patients with parotid gland lesions were diagnosed by clinical examination, ultrasound, and FNAC under ultrasound guidance at our hospital from 1 May 2019 to 30 April 2021. The FNAC results were divided into six categories according to the Milan system. We calculated the ROM for each category of the Milan system based on histopathological follow-up. RESULTS: This study included 204 patients. There were 115 men (56 %) aged 46-60 years. Pathology results were 33 cases for malignant and 182 cases for benign. The rate of malignancy for each category according to the MSRSGC were 23.1 % (non-neoplastic), 20 % (atypical), 50 % (neoplastic), 1 % (benign), 10.3 % (salivary neoplasm of uncertain neoplastic potential), 84.6 % (suspicious for malignancy), and 100 % (malignant) categories. Sensitivity, specificity, positive predictive value, negative predictive value and accuracy of FNAC with application of Milan system were respectively 90.9 %, 98.2 %, 90.9 %, 98.9 % and 97 %. CONCLUSION: When the Milan system was applied, FNAC had a high efficacy, suggesting that MSRSGC can improve the communication between the cytopathologist and the surgeon. This system can allow the surgeon to decide the extent of the surgery.

The protective role of postoperative radiation therapy in low and intermediate grade major salivary gland malignancies: A study of the Canadian Head and Neck Collaborative Research Initiative.

BACKGROUND: The objective of this study was to examine the utility of postoperative radiation for low and intermediate grade cancers of the parotid and submandibular glands. METHODS: The authors conducted a retrospective, Canadian-led, international, multi-institutional analysis of a patient cohort with low or intermediate grade salivary gland cancer of the parotid or submandibular gland who were treated from 2010 until 2020 with or without postoperative radiation therapy. A multivariable, marginal Cox proportional hazards regression analysis was performed to quantify the association between locoregional recurrence (LRR) and receipt of postoperative radiation therapy while accounting for patient-level factors and the clustering of patients by institution. RESULTS: In total, 621 patients across 14 tertiary care centers were included in the study; of these, 309 patients (49.8%) received postoperative radiation therapy. Tumor histologies included 182 (29.3%) acinic cell carcinomas, 312 (50.2%) mucoepidermoid carcinomas, and 137 (20.5%) other low or intermediate grade primary salivary gland carcinomas. Kaplan-Meier LRR-free survival at 10 years was 89.0% (95% confidence interval [CI], 84.9%-93.3%). In multivariable Cox regression analysis, postoperative radiation therapy was independently associated with a lower hazard of LRR (adjusted hazard ratio, 0.53; 95% CI, 0.29-0.97). The multivariable model estimated that the marginal probability of LRR within 10 years was 15.4% without radiation and 8.8% with radiation. The number needed to treat was 16 patients (95% CI, 14-18 patients). Radiation therapy had no benefit in patients who had early stage, low-grade salivary gland cancer without evidence of nodal disease and negative margins. CONCLUSIONS: Postoperative radiation therapy may reduce LLR in some low and intermediate grade salivary gland cancers with adverse features, but it had no benefit in patients who had early stage, low-grade salivary gland cancer with negative margins.

Salivary Ductal Carcinoma: A Case Report.

ABSTRACT: Salivary ductal carcinoma is an extremely rare tumor located in the parotid gland. This case details a salivary ductal carcinoma within the parotid gland of a 59-year-old man and its management.

Could definitive radiotherapy be a treatment option for lymphoepithelial carcinoma of major salivary gland: Comparison of clinical outcomes of upfront surgery and upfront chemoradiotherapy.

OBJECTIVES: The optimal treatment and associated clinical outcomes for lymphoepithelial carcinoma of the major salivary gland (LECSG) are currently unclear. As such, the purpose of this study was to assess the survival rates of LECSG patients who received either upfront surgery or upfront chemoradiotherapy (CRT). MATERIALS AND METHODS: In this retrospective study, we analyzed cases of LECSG patients treated at our center from January 2010 to April 2021. The cumulative incidences of overall survival rate (OS) and locoregional failure-free survival rate (LRFFS) were evaluated using the Kaplan-Meier method. In order to balance potential risk factors between the treatment groups, we conducted propensity score matching (PSM) at a 1:1 ratio. RESULTS: The study enrolled a total of 107 patients, among whom 24 received surgery alone, 56 underwent surgery combined with postoperative radiotherapy, and 27 underwent definitive radiotherapy. The 5-year LRFFS rate and 5-year OS rate for the entire cohort were 86.6% and 84.4%, respectively. Following PSM, the 5-year LRFFS and OS rates for the upfront CRT cases were comparable to those of upfront surgery, both before and after matching. However, the upfront surgery group showed a tendency toward more de novo facial nerve injury and post-treatment facial nerve injury. CONCLUSION: The results of this study suggest that upfront CRT is as effective as upfront surgery in terms of locoregional control and overall survival for LECSG patients. Therefore, upfront CRT could be considered a viable treatment option, potentially avoiding the risks associated with surgical intervention.

Unusual presentation of cribriform adenocarcinoma of salivary glands: A case report.

According to the last edition of the World Health Organization classification, cribriform adenocarcinoma of salivary glands (CASGs) was considered a variant of polymorphus adenocarcinoma although many authors proposed CASG as a distinct neoplasm. The aim of this study was to report an unusual presentation of CASG in the buccal mucosa of a 63-year-old male patient that showed signs of encapsulation and no evidence of lymph node metastasis. The lesion was composed of lobules of tumoral cells arranged in solid nests, sheets, papillary, and cribriform or glomeruloid patterns. Most of the peripheral cells show palisaded arrangement with peripheral clefting from the adjacent stroma. Surgical resection of the lesion was done and further neck dissection was recommended.

Intraductal Carcinoma: The Carcinoma In Situ of the Salivary Gland.

Intraductal carcinoma of the salivary gland (IDC) is a rare in situ neoplasm of the salivary gland with similar features to the ductal carcinoma in situ of the breast. This report aims to present the clinical presentation and histological features of IDC. The authors present a 90-year-old gentleman with an indurated and painless tumor within the right parotid. Preoperative diagnostics, including fine needle aspiration cytology, ultrasound scan, and magnetic resonance imaging, were suggestive of Warthin tumor. The tumour was excised by extracapsular dissection. The patient is disease free within the follow-up period of 33 months. Intraductal carcinoma is an indolent phenotype with only a few reported cases with nodal metastases, and to the best of our knowledge, no cases have been reported with distant metastases. Complete surgical excision is recommended to prevent a recurrence. The knowledge of this underreported salivary gland malignancy is important to prevent misdiagnosis and insufficient treatment.

Postoperative Radiation Therapy Refusal in Major Salivary Gland Cancers.

OBJECTIVE: Major salivary gland cancers (MSGCs) are often treated with primary surgery followed by adjuvant therapy for high-risk pathology. Patients with these cancers may opt out of recommended postoperative radiation therapy (PORT) for many reasons and consequently may suffer worse outcomes. STUDY DESIGN: Retrospective cohort study. SETTING: National Cancer Database. METHODS: Patients diagnosed with MSGC from 2004 to 2016 were identified, and overall survival and risk factors for refusal of recommended PORT were analyzed based on demographic, socioeconomic, and clinical factors. Multivariable logistic regression and a Cox model were used to conduct the analysis. RESULTS: 211 out of 4704 qualifying patients (4.5%) refused recommended PORT. Multivariable analysis demonstrated increased PORT refusal for age >74 years (odds ratio OR 4.34, confidence interval [CI] [2.43-7.85]), Asian race (OR 2.25, CI [1.10-4.23]), and certain facility types (comprehensive cancer center, OR 2.39, CI [1.08-6.34]; academic research program, OR 3.29, CI [1.49-8.74]; and integrated network cancer program, OR 2.75, CI [1.14-7.7]). N2 stage was associated with decreased PORT refusal (OR 0.67, CI [0.45-0.98]). The 5-year overall survival for patients who received and refused PORT were significantly different at 65.8% and 53.8%, respectively (p < .001). When controlling for several factors, PORT refusal was independently associated with significantly lower overall survival (HR 1.54, CI [1.21-1.98]). CONCLUSION: Patient refusal of recommended PORT in MSGC is rare, associated with various disease and socioeconomic factors, and may decrease overall survival. Our findings can assist clinicians in counseling patients and identifying those who may be more likely to opt out of recommended PORT.

Survival outcomes, prognostic factors, and effect of adjuvant radiotherapy and prophylactic neck dissection in salivary acinic cell carcinoma: A prospective multicenter REFCOR study of 187 patients.

BACKGROUND: Acinic cell carcinomas (AciCCs) are malignant tumours of the salivary glands. The aim of this work was to analyse data from the national REFCOR multicenter cohort (i) to investigate the prognostic factors influencing survival outcomes in AciCC, (ii) to assess the impact on survival of postoperative radiotherapy (RT) in patients treated for AciCC without high-grade transformation and (iii) to explore the prognostic impact of prophylactic neck dissection (ND) in patients treated for AciCC of the major salivary glands. PATIENTS AND METHODS: Data from all the patients treated for salivary AciCC between 2009 and 2020 were extracted from the REFCOR database. Survival outcomes and prognostic factors influencing Disease-Free Survival (DFS) and Overall Survival (OS) were investigated using univariate and multivariate analyses. Propensity score matching was used to assess the impact of postoperative RT and prophylactic ND on DFS. RESULTS: A total of 187 patients were included. After a median follow-up of 53 months, their 5-year OS and DFS rates were 92.8% and 76.2%, respectively. In multivariate analysis, male sex, older age, higher T and N status, and high grade were independently associated with a worse DFS. In the subpopulation analysed after propensity score matching, patients with cN0 AciCC without high-grade transformation who were treated by surgery and RT did not have an improved DFS compared to patients who were treated by surgery alone (hazard ratio (HR) = 0.87, p = 0.8). Factors associated with nodal invasion were T3-T4 status and intermediate/high histological grade. After propensity score matching, prophylactic ND was associated with a trend toward a better DFS (HR = 0.46, p = 0.16). CONCLUSIONS: These results suggest that (i) long-term follow-up (>5 years) should be considered in patients with AciCC, (ii) treatment by surgery alone could be an option in selected cN0 patients with AciCC without high-grade transformation and (iii) prophylactic ND may be considered preferentially in patients with T3-T4 status and/or intermediate/high histological grade.

Metastatic Spinal Tumor from Benign Pleomorphic Adenoma: Case Report and Literature Review.

PURPOSE: Pleomorphic adenomas tend to arise in the salivary glands. They are rare and histologically benign but can result in distant metastasis, and their characteristics need further investigation. We report a case of locally recurring benign primary palatal pleomorphic adenoma that resulted in spinal metastases and review the relevant literature. CASE REPORT: A 58-year-old woman had undergone surgery for a palatal pleomorphic adenoma 22 years earlier and 6 subsequent operations for local recurrences. During follow-up, metastases to multiple organs, including the spine, were diagnosed and 4 CyberKnife treatments were performed. She suffered right flank pain and slight paralysis of the right leg; radiological findings showed a growing metastatic spinal tumor. She underwent removal of a thoracic vertebral tumor and posterolateral fusion. Postoperatively, her symptoms improved. Histopathological analysis indicated a pleomorphic adenoma and no evidence of malignancy. Although there was no local recurrence, 23 months after surgery, a fifth CyberKnife procedure was performed for a growing salivary gland tumor and she is currently being followed up. CONCLUSION: We described a rare case of benign pleomorphic adenoma that metastasized to the spine. Long-term follow-up for recurrence and metastasis is required for patients with benign pleomorphic adenoma.