Managing malignant sublingual gland tumors: a single institution experience with 23 patients.

BACKGROUND: Due to the relative rarity of malignant sublingual gland tumors, diagnosing and treating them clinically pose challenges. Hence, there's a need to explore the pathological types, characteristics, treatment methods, and prognosis of primary malignant tumors of the sublingual gland to improve our understanding and management of these rare yet highly malignant conditions. METHODS: This study reviewed cases of primary malignant sublingual gland tumors, analyzing their characteristics. The treatment methods included surgical excision, with additional radiotherapy, or brachytherapy for advanced stages or positive surgical margins. The study also summarized different treatment approaches, including lymph node dissection and soft tissue reconstruction using free flaps such as the anterolateral thigh flap and forearm flap. RESULTS: We have gathered 23 cases of sublingual gland malignancies treated at the Department of Oral and Maxillofacial Surgery, School and Hospital of Stomatology, Wuhan University, from January 2013 to May 2024. The most common pathological types were adenoid cystic carcinoma and mucoepidermoid carcinoma, with rare cases of mucosa-associated lymphoid tissue (MALT) lymphoma and nonspecific salivary gland clear cell carcinoma. Early diagnosis and surgical intervention were crucial for a favorable prognosis. Marginal mandibulectomy was necessary for cases involving the mandible. Patients with positive preoperative lymph node detection required cervical lymph node dissection. Extensive tissue defects in the floor of the mouth were effectively reconstructed with free flaps to prevent oral-mandibular fistula. CONCLUSION: Surgical excision remains the preferred treatment for malignant sublingual gland tumors. Early diagnosis and comprehensive surgical management are essential for improving prognosis. The study's limitations include a small sample size and short follow-up duration, necessitating further research with larger clinical samples to confirm these findings.

Sublingual Gland Carcinoma Revealed by Choroidal Metastasis.

A 65-year-old man presented with a 1-week history of left eye distortion. An elevated choroidal lesion covering 6 disc diameters was found in the posterior retina of the left eye. Systemic examination revealed sublingual gland carcinoma and multiple lung metastases, and the diagnosis was choroidal metastasis from sublingual gland carcinoma. Following chemotherapy and radiation therapy, the choroidal lesion shrunk and the patient's visual acuity improved. The patient died 23 months after his first visit. To the best of our knowledge, this is the first reported case of choroidal metastasis from sublingual gland carcinoma.

Heterogeneous impact of smoking on major salivary gland cancer according to histopathological subtype: A case-control study.

BACKGROUND: Major salivary gland cancers (M-SGCs) are rare, and have distinct heterogeneous histopathological subtypes. To the authors' knowledge, no consistent evidence of an association between cigarette smoking and the risk of M-SGCs has appeared to date. Furthermore, evidence of potential heterogeneity in the impact of smoking on histopathological subtypes is scarce, despite the fact that the histopathological subtypes of M-SGC exhibit different genetic features. METHODS: The authors conducted a case-control study to investigate the association between smoking and M-SGC by histopathological subtype. Cases were 81 patients with M-SGCs and the controls were 810 age-matched and sex-matched first-visit outpatients without cancer treated at Aichi Cancer Center Hospital from 1988 to 2005. Odds ratios (OR) and 95% confidence intervals (95% CI) were assessed by conditional logistic regression analysis with adjustment for potential confounders. RESULTS: Smoking was found to be associated with a significantly increased risk of M-SGC overall, with an OR of 3.45 (95% CI, 1.58-7.51; P =.001) for heavy smokers compared with never-smokers. A significant dose-response relationship was observed (P for trend, .001). When stratified by histological subtype, no obvious impact of smoking was observed among patients with mucoepidermoid carcinoma (MEC). In contrast, smoking demonstrated a significantly increased risk of M-SGCs other than MEC, with an OR of 5.15 (95% CI, 2.06-12.87; P<.001) for heavy smokers compared with never-smokers. The authors observed possible heterogeneity with regard to the impact of smoking on risk between MEC and M-SGCs other than MEC (P for heterogeneity, .052). CONCLUSIONS: The results of the current study demonstrate a significant positive association between cigarette smoking and the risk of M-SGC overall. However, the impact of smoking appeared to be limited to M-SGCs other than MEC. Cancer 2018;124:118-24. © 2017 American Cancer Society.

Prognostic Factors in Malignant Sublingual Salivary Gland Tumors.

PURPOSE: The present study identified the prognostic factors and outcomes for malignant sublingual salivary gland tumors, which are rare. MATERIALS AND METHODS: A retrospective cohort study of patients treated for malignant sublingual salivary gland tumors from 1997 to 2011 was performed. The predictor variables, including age, gender, tumor stage, nodal stage, perineural invasion, margin status, and lymphovascular invasion, were analyzed. The Cox regression model was used to determine the prognostic factors for locoregional recurrence, distant metastasis, and survival. RESULTS: Of the 38 patients, 16 (42.1%) were men. Their mean age was 53 years (range 36 to 75). A total of 11 patients (28.9%) had T3-T4 tumors, and 6 (15.8%) had positive neck lymph nodes. The recurrence rate at 5 years was 18.4%. The distant metastasis rate at 5 years was 23.7%. Multivariable analysis confirmed the independent prognostic importance of patient age, N stage, and limited tongue mobility in locoregional recurrence and mortality at 5 years. CONCLUSIONS: Our results suggest that patient age, N stage, and limited tongue mobility are useful as independent predictors of locoregional recurrence and mortality in patients with malignant sublingual salivary gland tumors.

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.

Distant Metastases in Patients with Carcinoma of the Major Salivary Glands.

BACKGROUND: This study aimed to show the incidence of distant metastases (DM) in salivary gland cancer as well as the types of histology most commonly associated with it and to identify factors predictive of DM. METHODS: The study identified 301 patients who underwent surgery for cancer of the major salivary glands at Memorial Sloan-Kettering Cancer center between 1985 and 2009. Clinical, tumor, and treatment characteristics were recorded. Tumors were categorized as low-, intermediate-, and high-risk pathology based on histologic subtype and grade. Factors predictive of distant recurrence-free probability (DRFP) were determined by uni- and multivariable analyses. RESULTS: The primary tumor was parotid in 266 patients (88 %), and 96 tumors (32 %) were clinical T3/T4. For 57 patients (18.9 %), DM developed with a 5-year DRFP of 72.7 %. The most common site of metastasis was the lung (50 %). The clinical predictors were male gender, cT4 stage, cN+ stage, and clinical overall stage. The multivariable analysis of clinical variables showed male gender (p = 0.018), cT4 stage (p < 0.001), and cN+ stage (p = 0.004) to be significant. The pathologic predictors were high-risk and high-grade pathology, vascular invasion, perineural invasion, positive margins, pT4 stage, pN+ stage, and overall stage. The multivariable analysis of pathologic variables showed high-grade pathology (p < 0.001), perineural invasion (p = 0.005), and pN+ stage (p = 0.002) to be significant. CONCLUSIONS: Distant metastases developed in approximately 20 % of the patients with salivary gland cancer. The most common site of metastases was the lung. The significant predictors of DM were cT4, cN+, male gender, high-grade pathology, perineural invasion, and positive nodal disease.

Extracardiac rhabdomyomas--presentation of two cases with analysis of AKT/mTOR and ERK1/2 signaling.

Extracardiac rhabdomyomas (RM) are very rare benign tumors with a poorly understood pathogenesis. In this report we describe two RM cases--a sublingual adult type tumor and a genital type tumor involving the uterine cervix. The patho-clinical characteristics, as well as the pioneer immunohistochemical analysis of ERK1/2 and AKT/mTOR pathway status is included. The expression of key proteins involved in above signaling gives new insight into the biology of extracardiac RM.

Hyalinizing clear cell carcinoma of the sublingual gland: A case report and literature review.

RATIONALE: Hyalinizing clear cell carcinoma (HCCC) of the salivary glands is a rare low-grade malignant tumor. This type of tumor is particularly uncommon in the sublingual glands. PATIENT CONCERNS: A 57-year-old female with a mass on the left side of the floor of the mouth that had been present for 2 months. The computed tomography scan of the neck revealed a nodular abnormal density shadow in the left sublingual area, measuring approximately 2.6 cm × 1.9 cm. DIAGNOSES: Primary HCCC of the sublingual gland. INTERVENTIONS: The patient underwent surgical treatment and reconstruction using a left anterolateral femoral free flap, which showed immunohistochemical positivity for CK 5/6, CK 7, CK (AE1/AE3), and Ki-67 (<5%), but negative for SMA and S-100. OUTCOMES: No recurrence was observed during the 12-month postoperative follow-up period. LESSONS: The absence of characteristic clinical manifestations makes HCCC highly susceptible to misdiagnoses. This case presents a rare instance of HCCC in the sublingual gland, providing a reference for the clinical diagnosis and treatment of the disease.

Reconstructive strategies following surgical resection of malignant sublingual gland tumors: A single institution experience.

OBJECTIVE: To investigate the characteristics and treatment modalities of malignant tumors originating from the sublingual gland, as well as evaluate the therapeutic outcomes following free flap reconstruction. METHODS: A retrospective statistical analysis was conducted on the clinical data of nine patients diagnosed with malignant neoplasms tumor of the sublingual gland. RESULTS: Nine case of malignant tumors originated from the sublingual glandular tissue, encompassing eight adenoid cystic carcinoma (ACC) and a single case of bipartite differentiated carcinoma-a hybrid of epithelial-myoepithelial carcinoma and adenoid cystic carcinoma. Among the nine patients, four anterolateral thigh flaps were used (three of which were thin flaps), and five forearm flaps were also empoyed. The size of flaps varied, with the lengths ranging from 4 cm to 9 cm, and the widths ranging from 2.5 cm to 6 cm. The vessels chosen for anastomosis were the superior thyroid artery in seven cases, the facial artery in one case, and the lingual artery in one case. Among the eight patients who underwent dissection of cervical lymph nodes, metastasis were found in one case. Two patients underwent adjuvant radiotherapy. Upon postoperative follow-up, there was no recurrence in any of the nine patients . CONCLUSION: The anterolateral thigh perforator flap thinning technique can be employed for postoperative reconstruction of malignant sublingual gland tumors.

Schwannoma arising from the sublingual gland.

Sublingual gland tumors, especially mesenchymal tumors, are extremely rare. We describe the first reported case of schwannoma arising from the sublingual gland with details of the histopathologic and immunohistochemical features. A 70-year-old woman developed a painless swelling on the floor of the mouth. The excised material was sublingual gland tissue with an ovoid, grayish-yellow solid tumorous mass at the cut surface. The tumor was composed of proliferated spindle-shaped tumor cells exhibiting palisading patterns. In the center of the tumor, a small salivary gland component was recognized. Immunohistochemically, the tumor cells were strongly positive for S-100 protein but negative for neurofilament protein. The Ki-67 labeling index was 4.58. The clear presence of a remnant sublingual gland lobule in the present tumor provided convincing evidence that it was a schwannoma arising from the sublingual gland and thus the first of its type to be reported.

Pleomorphic adenoma of the sublingual salivary gland: an unusual diagnostic challenge.

OBJECTIVES: To describe a rare case of pleomorphic adenoma of the sublingual gland. CLINICAL PRESENTATION AND INTERVENTION: An 80-year-old patient presented with a painless solid submucosal mass of 4 months' duration in the anterior floor of the mouth, appearing as a distinct homogeneous, radiolucent lesion in CT imaging. Histologic findings after complete removal were suggestive of a pleomorphic adenoma almost completely surrounded by a fibrous capsule. No recurrence was observed after a 12-month follow-up. CONCLUSION: This case showed the possibility of sublingual salivary gland tumors and highlighted the need to consider them when making a differential diagnosis of masses located in the floor of the mouth.

A comprehensive analysis of parotid and salivary gland cancer: worse outcomes for male gender.

BACKGROUND: To determine the effects of patient demographics, socioeconomic status (SES) and clinical variables on outcomes for patients with salivary and parotid gland tumors. METHODS: Florida cancer registry and inpatient hospital data were queried for cancer of the salivary glands diagnosed between 1998-2002. RESULTS: A total of 1573 patients were identified. Women were diagnosed at a younger age (median age (years): women 60.8 versus men 64.3, P=0.003). Men were more often diagnosed with high grade tumors (65.1% versus 41.9% for women, P<0.001) and advanced disease stage (>stage III: 60.2 versus 49.4%, P<0.001), but underwent surgical extirpation and received radiation at equal rates compared with women. Overall 5-year survival rates was superior in women (67.4% versus 55.6%, P=0.001). By multivariate analysis, adjusted for patient comorbidities, age over 65 (HR 3.43 P=0.008), advanced disease stage (HR 8.05 P<0.001), and high tumor grade (HR 2.33, P<0.001) were independent predictors of worse prognosis. Improved outcomes were observed for female gender (HR 0.68, P=0.011). Tumors located in the parotid gland (HR 0.631 P=0.003) and receiving both surgical extirpation and radiation were predictors of improved survival. CONCLUSION: Salivary gland tumors carry a worse prognosis than tumors of the parotid. Male patients have worse outcomes.

Adenoid cystic carcinoma of the sublingual gland developing lung metastasis 20 years after primary treatment: A case report and literature review.

RATIONALE: Adenoid cystic carcinoma (ACC) is a rare malignant tumor that primarily occurs in the salivary glands. Distant metastases can develop despite favorable local control. Moreover, distant metastasis of ACC can occur after a long time interval without local recurrence. We report the first case of ACC of the sublingual gland that developed lung metastasis 20 years after primary treatment. PATIENT CONCERNS: A 52-year-old man was referred to our department with a 1-year history of painful swelling on the right oral floor. DIAGNOSIS: An incisional biopsy was performed, and histopathological examination revealed malignancy. INTERVENTIONS: Surgical excision of the right oral floor and right supra-omohyoid neck dissection with postoperative chemoradiation therapy were performed, and ACC of the sublingual gland was diagnosed. Left pulmonary metastasis was detected 20 years after the primary treatment. Metastasectomy was performed; however, subsequently, skin and bone metastases developed. OUTCOMES: After receiving palliative care, the patient died of multiple organ failure. LESSONS: As late distant metastasis of salivary ACC can develop, patients who undergo primary treatment need a long-term, strict follow-up plan even if locoregional control is favorable.

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.

Rhabdomyomas of the submandibular and sublingual glands.

Rhabdomyomas are rare benign mesenchymal tumors distinguished in cardiac and extracardiac forms. Extracardiac rhabdomyomas are classified as adult type, fetal type, and the genital type. Adult extracardiac rhabdomyomas are mostly found in the head and neck and usually present a slow growth. We report a case of a patient with a manifestation in the submandibular and sublingual glands.

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.

Development of sentinel lymph node biopsy technique in patients with salivary gland cancer using the IDEAL framework.

BACKGROUND: Salivary cancer is rare and comprises a variety of histological subtypes and clinical behaviors. There is no agreed method of estimating the risk of occult metastasis or managing the clinically N0 neck.Sentinel node biopsy (SNB) may offer a solution but previous studies have not produced a reliable imaging protocol. This study uses novel technology and trial methodology to develop a reliable SNB technique, with primary aim to identify peri-and intraglandular sentinel nodes. METHODS: IDEAL framework was used to undertake SNB in clinically node negative salivary gland cancer. Patients with cT1-2 N0 salivary cancer were eligible. Lymphoscintigraphy was undertaken using Tc-99 m labelled nanocoll. Injection technique as well as adjunctive use of freehand SPECT (fhSPECT), near-infrared (NIR) fluorescence imaging, and navigation-guided surgery were used and optimisied during the study protocol. RESULTS: 10 patients were recruited. Initial protocol of peritumoural injection of Tc99 m nanocoll showed poor image resolution. Subsequent adjustment to single intratumoural injection allowed identification of intraglandular sentinel nodes. Fh/SPECT and NIR fluorescence imaging found intraglandular lymph nodes otherwise not recognizable to the naked eye. In two cases occult lymph node metastasis were identified. CONCLUSION: This study has shown the IDEAL framework is vital in allowing iterative changes in surgical protocol in the light of experience. This study has produced a reliable method for detection of sentinel nodes, in particular the ability to identify intra- and periglandular nodes with diagnosis of occult metastatic deposits and no false negative results. Our protocol can be readily transferred in to larger scale studies.

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.

Malignant sublingual gland tumors: a retrospective clinicopathologic study of 28 cases.

OBJECTIVES: To evaluate the clinicopathologic features and therapeutic efficacy of malignant sublingual gland tumors. PATIENTS AND METHODS: From 1955 to 2005, the clinicopathologic data of 28 patients treated in our hospital were obtained from their medical records and histopathologic slides. RESULTS: There were 16 males and 12 females, with a mean age of 50.3 years. Eighteen cases (64.3%) were adenoid cystic carcinoma; 16 (57.1%) cases were clinically staged as III-IV. Adenoid cystic carcinoma was mainly of the histologic type, and the other histologic classifications included mucoepidermoid carcinoma, myoepithelial carcinoma, polymorphous low-grade adenocarcinoma, adenocarcinoma and malignant pleomorphic adenoma. Pulmonary metastasis and tumor recurrence were the main death reasons. Eleven patients remain alive and well 34-312 months (median 108) after treatment. CONCLUSIONS: Malignant sublingual gland tumors are extremely rare and most are adenoid cystic carcinoma. Surgery is the main treatment option. For adenoid cystic carcinoma, hematogenous spread is common, and pulmonary metastasis is a common pathway of the distant metastasis. For some patients having lung metastasis, regional control is also important as there are some examples of patients surviving many years with asymptomatic pulmonary metastases. Postoperative radiation therapy may be adjuvant for selected patients with high-stage and high-grade tumors, or when there is concern about the inadequacy of the resection. The effect of chemotherapy remains elusive.

NUT Midline Carcinoma of the Sublingual Gland: Clinical Presentation and Review.

NUT midline carcinoma (NMC) is a rare and aggressive disease encountered in the midline of the head and neck or mediastinum. Due to its sparse incidence and subtle pathologic features, we aim to increase knowledge and awareness for this pathologic entity. We present an exemplary case of a young, healthy male presenting with oral cavity pain and cervical lymphadenopathy. This patient was initially diagnosed with an unspecified, highly aggressive sublingual gland malignancy and underwent locoregional resection with free flap reconstruction however suffered a rapid local recurrence and widely extensive metastasis within just 1 month. After rigorous analysis, final pathologic diagnosis revealed a poorly differentiated carcinoma with evidence of squamous differentiation that eventually, post-mortem tested positive for NMC. Only one prior case of sublingual gland NMC has been previously reported as we discuss the literature regarding all sublingual gland malignancies as well as the pathologic features and treatment options for NMC. We recommend consideration of testing for the NUT proto-oncogene at the time of biopsy in the clinical setting of a poorly differentiated midline carcinoma, especially with squamous differentiation, of the head or neck in order to identify patients for clinical trial enrollment and appropriately counsel on the poor clinical prognosis. Improving clinician awareness is critical to increase diagnostic accuracy and need to study prospective treatment outcomes as the first step toward improving management of this difficult disease.