Sublingual gland neoplasms: clinicopathological study of 8 cases.

BACKGROUND: Sublingual gland neoplasms are very rare and the majority of them are malignant. The aim of this study was to evaluate the clinical course, treatment, and outcomes of these uncommon neoplasms based on the authors' experience and the recent literature. MATERIAL AND METHODS: The medical charts of 8 patients with primary epithelial sublingual gland tumors treated between 1994 and 2020 were reviewed. RESULTS: Malignant tumors comprised 75% (6/8) of cases. Adenoid cystic carcinoma was the most common (50%, 3/6) and characterized by high risk of local recurrence and lung metastasis. Pleomorphic adenoma was the only representative of benign tumors with no evidence of local recurrence in follow up. CONCLUSIONS: Treatment of choice of sublingual gland tumors is surgery. However, due to the fact that adenoid cystic carcinoma is the most common malignancy with poor prognosis, surgical treatment should be combined with postoperative radiotherapy. Benign sublingual tumors are less common and treatment of choice in these cases is tumor resection together with sublingual gland.

Carcinoma Showing Thymus-Like Differentiation (CASTLE) Arising in the Sublingual Gland.

Carcinoma showing thymic-like differentiation (CASTLE) is a rare tumor most commonly occurring in the thyroid and soft tissues of the neck. We report the first case of CASTLE occurring in the sublingual gland. The patient, a 35-year-old healthy man, presented with a submucosal lesion located in the anterior right floor of the oral cavity and an ipsilateral neck mass. The lesion had been previously investigated by neck computed tomography and ultrasound-guided fine needle aspiration cytology and diagnosed as metastatic squamous cell carcinoma. After oral cavity magnetic resonance imaging, positron emission tomography, and a non-diriment, fine needle aspiration cytology of the sublingual mass, the patient was treated as affected by a sublingual gland malignancy with removal of primary tumor and neck dissection. Morphological and immunohistochemical findings were diagnostic for primary sublingual gland CASTLE. The patient received adjuvant radiotherapy and is free of disease 2 years after treatment. We describe the pathological features of the lesion and discuss the possible differential diagnoses.

A retrospective multicenter study of sublingual gland carcinoma in Japan.

OBJECTIVE: Salivary gland carcinoma is rare among head and neck cancers. Sublingual gland carcinoma, a type of salivary gland carcinoma, is even rarer; therefore, the number of cases at a single institute is too small for sufficient evaluation of tumor characteristics. We conducted a multicenter, retrospective analysis of sublingual gland carcinomas in patients who visited 12 institutions associated with the Kyoto Hospital and Affiliated Facilities Head and Neck Clinical Oncology Group. METHODS: Thirteen previously untreated patients who visited the institutions between 2006 and 2015 were enrolled. The overall survival (OS) and disease-free survival (DFS) rates for all patients and by disease stage were analyzed. Statistical analyses were performed for all patients with respect to disease stage. RESULTS: Eight of thirteen patients were diagnosed with adenoid cystic carcinoma on pathological study. A significant difference in OS rate was observed between patients with Stage I-III and Stage IV disease; however, the difference in DFS rate by disease stage was not significant. CONCLUSION: Stage IV disease was identified as a poor prognostic factor in patients with sublingual gland carcinoma. However, even patients with Stage I-III disease experienced relatively short DFS. Distant metastasis is a serious problem among patients with sublingual gland carcinoma.

The number and ratio of positive lymph nodes are independent prognostic factors for patients with major salivary gland cancer: Results from the surveillance, epidemiology, and End Results dataset.

INTRODUCTION: To investigate whether the positive lymph node number (PLNN) and positive lymph node ratio (PLNR) could predict the prognosis of patients with major salivary gland cancer (MSGC) and to identify the optimal cutoff points for these variables that stratify patients according to their risk of survival. METHODS: We used the Surveillance, Epidemiology, and End Results (SEER) database to identify all patients with MSGC between 1988 and 2014. A logistic regression analysis was carried out to evaluate the risk factors for lymph node metastasis (LNM) in MSGC. The X-tile program was used to identify the cutoff values for the PLNN and PLNR in MSGC patients with LNM. Cox proportional hazards regression models were performed to identify the predictors of cancer-specific survival (CSS). RESULTS: In the SEER database, 8668 eligible patients were identified and 3046 of them had LNM. The logistic regression analysis indicated that older age, male sex, larger tumor size, higher grade, tumor extension and high-risk pathology were associated with LNM. The X-tile program showed that a PLNN>4 and a PLNR>0.15 were prognostic indicators of CSS. A multivariable analysis indicated that, after the factors that might potentially affect the prognosis were adjusted for, the PLNN and PLNR were still associated with CSS. CONCLUSIONS: Our Results demonstrated that the PLNN and PLNR were independent prognostic indicators for MSGC patients with lymph node metastasis.

Adenoid cystic carcinoma of the head and neck: a retrospective multicenter study.

BACKGROUND: Adenoid cystic carcinoma of the head and neck (ACCHN) is rare and difficult to study effective treatment at one institute. Our aim is to identify prognostic factors for this disease by conducting a multicenter study at 11 institutions in Japan. METHODS: A retrospective multicenter study of ACCHN was performed. One hundred and three patients were identified between 2006 and 2015. The overall survival (OS) rate for all patients was calculated, and OS, locoregional control (LRC) rate, or no distant metastasis (NDM) rate was calculated for patients in that the surgery was performed without distant metastasis (DM). Statistical analyses were performed. RESULTS: A significant difference with multivariate analysis was observed in patients in sublingual glands, stage IV and the use of radiation therapy ≥60Gy (sufficient RT) in OS for all patients. A significant difference was observed in the use of sufficient postoperative RT in the OS and the LRC rate, and in pathological surgical margins in the NDM rate. CONCLUSION: Sublingual glands or stage IV was a poorer, and sufficient RT was a better prognostic factor for ACCHN. Sufficient RT was effective to prevent local recurrence after surgical resection. Positive surgical margins caused an increase in DM.

The clinical features of epithelioid hemangioendothelioma in a Han Chinese population: A retrospective analysis.

Epithelioid hemangioendothelioma (EHE) is a rare indolent vascular tumor which occurs at liver, lung, bone, and so on. However, the etiology of EHE is evasive.These patients were enrolled at the First Affiliated Hospital of Xi'an Jiaotong University from January 2011 to December 2015. Retrospective analysis is done by demographic data of clinical manifestations, laboratory parameters, CT imaging, histological, and immunohistochemical features in 9 cases in Shaanxi.Of the patients, 8 were females (88.9%) and 1 were males (11.1%). The age ranged from 34 to 71 years (mean 49 years; median 49 years). Anatomical sites of primary lesions were as follows: liver (n = 6, 66.7%), upper extremities (n = 1, 11.1%), sublingual gland (n = 1, 11.1%), and spine (n = 1, 11.1%). Metastatic disease was diagnosed in 5 cases (55.6%) with occurrence in lung (n = 4, 44.4%), bone (n = 2, 22.2%), upper extremities (n = 1, 11.1%), pleura (n = 1, 11.1%), and spleen (n = 1, 11.1%). Tumor size ranged from 0.5 to 6.8 cm (mean 3 cm). The most tumors were composed of highly cellular areas with small and prominent nucleoli in vesicular nuclei, and ERG (100%) was the most frequently positive in these cases, followed by CD31 (88.9%) and CD34 (77.8%) via histology and immunohistochemistry techniques.EHE is a very rare in Shaanxi. It is significant to find its clinical, radiological, and pathological characters, helping for EHE early diagnosis and treatment, reducing misdiagnosis and improving life quality.

Predictors of outcomes in large cell undifferentiated carcinoma of the major salivary glands.

OBJECTIVE: Major salivary gland large-cell undifferentiated carcinoma (LCUC) is rare and has a poor prognosis. Characterization of patient demographics, tumor characteristics, and predictors of outcome have been limited by low case numbers, as well as grouped analysis with other salivary malignancies. The objective of this study was to address these issues using large-scale national data. STUDY DESIGN: Retrospective case series. METHODS: Data from the National Cancer Database, including cases diagnosed from 1998 to 2012, was analyzed, identifying 247 records of LCUC. Tumor, demographic, and survival information was extracted and analyzed retrospectively. RESULTS: Large-cell undifferentiated carcinoma comprised < 1% of all major salivary gland cancers. Seventy percent of patients presented with advanced-stage disease. The incidence of occult nodal disease was 39%. Surgery followed by radiation was the most common treatment. Five-year overall survival was 36%. Comorbidity, distant metastasis, and positive surgical margins were found to be predictors of overall survival. CONCLUSION: To our knowledge, this represents the largest reported case series of LCUC. The survival analysis demonstrates poorer survival in patients with positive surgical margins; therefore, efforts to complete resection are reasonable. Reported high rates of occult nodal disease also strongly support elective treatment of the neck. LEVEL OF EVIDENCE: 4. Laryngoscope, 2016 127:372-376, 2017.

Risk Factors and Survival Associated with Distant Metastasis in Patients with Carcinoma of the Salivary Gland.

BACKGROUND: Salivary gland cancer (SGC) is rare and has various pathologies and metastatic potentials. Because distant metastasis can be observed after treatment, as well as at initial presentation, this study aimed to investigate the rates, risk factors, and survivals associated with distant metastasis in patients with SGC. METHODS: This study involved 454 consecutive patients with previously untreated SGC who were treated at our tertiary referral center. Clinical factors, operative and pathologic findings, and treatment outcomes were carefully reviewed. Univariate and multivariate analyses were performed to identify factors associated with distant metastasis and their associations with distant metastasis-free survival (DMFS), cancer-specific survival (CSS), and overall survival (OS). RESULTS: Of 454 patients, 95 (20.9 %) presented with distant metastases; of these, 7 (7.4 %) were at the initial stage, while 88 (92.6 %) were detected during a median follow-up of 100 months (range 24-282). Distant metastases to single and multiple organs were found in 64 (67.4 %) and 31 (32.6 %) patients, respectively, with the most common site being the lung (77.9 %). In multivariate analysis, a non-parotid tumor site, high histological grade, perineural invasion, and T3-4 and N2-3 classifications were independent variables of DMFS, while distant metastasis was an independent variable of CSS and OS (p < 0.005 each). The median survival duration after distant metastasis development was 15 months (range 2-103). CONCLUSIONS: Distant metastasis frequently develops after treatment for SGC and is associated with poor survival outcomes; thus, close surveillance may be required for patients with SGC and risk factors.

Salivary gland malignancies - an update on current management for oral healthcare practitioners.

Salivary gland tumours represent a diverse range of tumours with many histological subtypes which occur in major and minor salivary glands. The management of these tumours is complex owing to their heterogeneity. Surgery together with radiotherapy and/or chemotherapy remains the treatment strategy for these tumours. The aim of this review is to examine the current management of these tumours.

Lymphoepithelial Carcinoma of the Sublingual Gland: Case Report and Review of the Literature.

Lymphoepithelial carcinoma represents only 0.4% of salivary gland neoplasms. Generally affecting the parotid gland, it has been reported only twice in the sublingual gland. Controversies concerning the treatment of lymphoepithelial carcinoma exist. Although the literature generally agrees that primary surgery with adjuvant radiotherapy is part of the treatment, the benefit of adjuvant chemotherapy is not well described. This report describes the case of a 55-year-old man diagnosed with lymphoepithelial carcinoma of the sublingual gland. The patient was admitted for progressive pain in the floor of the mouth associated with trismus. Biopsy examination confirmed the diagnosis of lymphoepithelial carcinoma of the sublingual gland and magnetic resonance imaging showed multiple left lymphadenopathies. Surgery consisted of a radical neck dissection type III, surgical resection of the floor of the mouth, and reconstruction with a left facial artery musculomucosal flap. The patient received adjuvant radiotherapy (60 Gy) and adjuvant chemotherapy (3 cycles of cisplatinum 100 mg/m(2)). The patient was disease free at 36 months of follow-up. The evidence base for administering adjuvant chemotherapy in this situation is discussed.

First description of a hybrid tumor of the sublingual gland.

BACKGROUND: Hybrid tumours of the salivary glands are rare neoplasms. They are composed of at least two different tumour entities located in the same topographic area and account for only 0.1% of all salivary gland tumours. The most common component is an adenoid cystic carcinoma. There are several possible forms of hybrid tumours, which are most commonly located in the parotid gland. CASE REPORT: We report on a 59-year-old female, who presented with a lesion of the caruncula of the left sublingual gland. The biopsy showed an adenoid cystic carcinoma in combination with a salivary duct carcinoma. Treatment consisted of tumour resection, bilateral selective neck dissection and adjuvant radiotherapy. Histopathologically, at least 30% of the tumour mass was composed of a salivary duct carcinoma and 70% of an adenoid cystic carcinoma. At 58 months after treatment, the patient is alive without evidence of recurrent disease. CONCLUSION: To our knowledge, the presented case is the first description of a hybrid tumour of the sublingual gland. Furthermore, the post-therapeutic course is encouraging, as hybrid tumours of the salivary glands usually have a poor prognosis.

Primary intestinal-like adenocarcinoma of major salivary glands: 2 instances of previously undocumented phenotype.

BACKGROUND: Primary intestinal-like adenocarcinoma of the major salivary glands has not previously been reported. METHODS: The clinical and radiological findings of 2 patients with primary submandibular and sublingual tumors are presented. Histopathologic and immunohistochemical examinations of tumor sections were performed. Metastatic workup for distant occult primary was carried out. RESULTS: The light-optic and the immunomarkers revealed intestinal-like adenocarcinoma consistent with upper respiratory tract origin. Metastatic workup of both patients was negative for primary gastro-intestinal primary. CONCLUSION: Primary intestinal-like adenocarcinoma can develop in major salivary glands and should be considered in the differential diagnosis.

[Clinical analysis of malignant submandibular tumor].

The 14 cases of malignant submandibular tumor whose treatment outcome we analyzed between 1989 and 2008 included 5 of adenoid cystic carcinoma, 3 of squamous cell carcinoma, 2 each of mucoepidermoid carcinoma, and carcinoma ex pleomorphic adenoma, and 1 each of carcinosarcoma and large-cell carcinoma. One subject was diagnosed with T1, 7 with T2, 4 with T3, and 2 with T4. Lymph node involvement occurred in 5, -1 with N1 and 4 with N2. None had distant metastasis on the first visit. Seven were treated by surgery alone, 3 by surgery followed by radiotherapy, 2 by surgery followed by radio-and chemotherapy, and 1 by optimized supportive care. The surgical resection area was decided by tumor extension. Neck dissection was done in 9. Overall 5-year survival for all cases based on the Kaplan-Meier method was 57%. All with carcinoma ex pleomorphic adenoma, carcinosarcoma, or large-cell carcinoma remain alive. For those with adenoid cystic carcinoma 5-year survival is 80%, with mucoepidermoid carcinoma 50%, with squamous cell carcinoma 0%, and with carcinosarcoma 0%, respectively. The 5-year survival for stage I subjects was 100%, for stage II 83%, for stage III 50%, and for stage IV 0%. Surgical resection and postoperative radiotherapy were done in cases of minimal extraglandular extension or microscopically positive margins, with satisfactory results. Treatment efficacy for high-grade and advanced stage, however, requires more improvement.

Sublingual gland tumors: clinical, pathologic, and therapeutic analysis of 13 patients treated in a single institution.

BACKGROUND: Sublingual gland tumors are rare, although frequently malignant. This study describes the clinicopathologic features and treatment results and reviews the literature. METHODS: Thirteen cases treated between 1996 and 2007 were reviewed with interest on clinical, pathologic, and therapeutic information. Survival data were calculated by the Kaplan-Meier method. RESULTS: Malignancies represented 92.3% of cases. Adenoid cystic carcinoma was the most common malignant type (66.7%). Most patients (83.3%) presented in advanced pathologic TNM stages (III or IV). All cases underwent surgical treatment. Neck dissection was performed in 69.2% with no metastases detected. Ten patients (83.3%) had adjuvant radiotherapy. Distant metastases occurred in 3 patients (25%). The 5-year overall and disease-free survival rates were 78.7% and 87.5%, respectively. CONCLUSIONS: Tumors of the sublingual gland are rare and are usually malignant. Radical surgery and adjuvant radiotherapy seems to offer adequate local and regional control. Unlike distant failure, local recurrence and regional metastases are not common.

The treatment of sublingual gland tumours.

This study assessed the clinical and histological features and therapeutic efficacy of 25 cases of sublingual gland tumours from 1998 to 2008. There were 17 female patients and 8 male, the ratio of females to males was 2.1:1. The mean age was 48.6 years. 4 cases were benign tumours (16%). 21 cases were malignant sublingual gland tumours (84%) and of these, 18 were adenoid cystic carcinoma (86%). Adenoid cystic carcinoma was mainly of the histological type, and the other histological classifications included mucoepidermoid carcinoma, pleomorphic adenoma, myoepithelioma, oncocytoma and polymorphous low-grade adenocarcinoma. Sublingual gland tumours are rare and most are malignant. For malignant sublingual gland tumours, early diagnosis and aggressive surgical treatment, especially for tumours with nerve involvement, is the key to improving prognosis. Free radial forearm flap or pectoralis major myocutaneous flap are appropriate methods for mouth floor reconstruction. For benign sublingual gland tumours, the resection of tumour and sublingual gland is the preferred treatment.

[Basal cell adenocarcinoma of the sublingual gland]. / Basalcelleadenokarcinom i glandula sublingualis.

The cause of a swelling in the floor of the mouth will in most cases be benign. For example, a ranula, sialolithiasis and/or infection. Tumors of the sublingual gland are very rare, but 90% are malignant and therefore malignancy should always be excluded in case of an asymptomatic swelling covered by intact mucosa.

Management of malignant sublingual salivary gland tumors.

The majority of tumors of the sublingual gland are malignant, with adenoid cystic carcinoma and mucoepidermoid carcinoma being the most frequent. Many other malignant tumor types have also been reported. The sublingual gland anatomically is not a unit organ and while it is described anatomically as being confined to the anterior floor of the mouth, salivary tissue may be located laterally along the submandibular duct and posterior floor of the mouth. Diagnosis should be suspected when any thickening or raised lesion presents in this area and a biopsy performed to confirm malignancy before planning further treatment. Surgery is the treatment of choice, and should include an en-block resection of the anterior floor of mouth as a minimum, and may include a portion of mandible, as well as a supraomohyoid neck dissection. Adjuvant radiotherapy should be considered in most of the patients after surgical excision.

[Adenoid cystic carcinoma. Presentation of three clinical cases]. / Carcinoma adenoide quístico. Presentación de tres casos clínicos.

Adenoid cystic carcinomas are malignant epithelial tumors that originate in body regions containing exocrine and eccrine glands. Their most common location is the salivary glands. These tumors have specific, highly individualized histopathological and clinical features. Three cases of adenoid cystic carcinoma in different sites, palate, sublingual gland, and nasal cavity, are reported. The literature is reviewed for information about the fundamental clinical, diagnostic, and therapeutic aspects of these tumors.