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.

Imaging of salivary gland cancers derived from a sublingual gland herniated into the submandibular space: a report of three cases.

Sublingual gland herniation into the submandibular space through a mylohyoid muscle defect is a common anatomical variation; however, salivary gland cancers that arise from a herniated sublingual gland have not been described yet. Here, we report three patients with salivary gland cancers originating from a herniated sublingual gland. All tumors were detected as palpable submandibular masses, located anterior to the submandibular gland, medial to the mandible, and lateral to the mylohyoid muscle, with contact with the sublingual gland through a mylohyoid muscle defect. Intraoperative findings confirmed that the masses were derived from herniated sublingual glands. Pathological examination showed one case of mucoepidermoid carcinoma and two cases of adenoid cystic carcinoma. Imaging findings of the tumor location, in addition to the continuity with the sublingual gland through the mylohyoid muscle defect, are crucial for accurately diagnosing the tumor origin, which is essential for determining the appropriate clinical management.

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.

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.

Sublingual Gland Tumors Worldwide: A Descriptive Retrospective Study of 839 Cases.

PURPOSE: Sublingual gland tumors are rare. We sought to define the general features of sublingual gland tumors for clinical reference. In addition, we evaluated whether it would be safe to speculate that ∼90% sublingual gland tumors will be malignant and that ∼90% of those malignant tumors will be adenoid cystic carcinoma. MATERIALS AND METHODS: In the present study, we have reported data from a pleomorphic adenoma case of the sublingual gland and a case series of sublingual gland tumors. Global data of sublingual gland tumors were retrieved. The cases pathologically identified as either benign or malignant tumors of the sublingual gland were included. The demographic, pathologic, and treatment features were analyzed. RESULTS: Data from 1 recent case of pleomorphic adenoma of the sublingual gland and a 21-case series of sublingual gland tumors were retrieved. A total of 839 cases of sublingual gland tumors were analyzed in the present study. The most commonly encountered age group was 40 to 59 years (47.6%). Of the 367 patients with gender specified, 178 were men (48.5%) and 189 were women (51.5%). Malignant tumors predominated (n = 722 cases; 86.1% of 839). Most malignant tumors were adenoid cystic carcinoma (n = 376), just greater than one half (52.1%) of all malignant tumors. Surgery was the only reported treatment method for the benign tumors. The most common treatment methods for the 164 explicit malignant tumors were surgery plus radiotherapy for 82 patients (50%), followed by surgery alone for 70 patients (42.7%). CONCLUSIONS: To date and to the best of our knowledge, the present study is the most comprehensive study on the demographic, pathologic, and treatment features of global sublingual gland tumors. These findings have shown that ∼90% of sublingual gland tumors will be malignant. However, the assumption that ∼90% malignant sublingual gland tumors will be adenoid cystic carcinoma is incorrect, which could be a new critical clinical reference.

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.

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.

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.

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.

Recurrent sublingual ranula or saliva leakage from the submandibular gland? Anatomical consideration of the ductal system of the sublingual gland.

PURPOSE: Some believe that the recurrence of sublingual ranula results from incomplete removal of the sublingual gland (SLG), but recurrence remains in some patients who undergo repeated excision of the remnant SLG, and the final solution to the recurrence is to remove the ipsilateral submandibular gland (SMG). In the authors' experience, preoperative aspirate from a sublingual ranula was a thick mucus-like fluid resembling egg white, whereas that from recurrent cyst after removal of the SLG was thin serous fluid. Based on the difference of the aspirated fluids, the authors speculated that the recurrent cystic mass might not be a ranula, but rather iatrogenic saliva leakage from the SMG through the previous surgically damaged excretory duct of the SLG (Bartholin duct) that opens into the SMG duct (Wharton duct). MATERIALS AND METHODS: A gross anatomic study was performed of the ductal system of the SLG and the anatomic communication between the Bartholin duct and Wharton duct. RESULTS: Four anatomic SLG duct variants were found. 1) The SLG has 1 Bartholin duct that seems to fuse with the Wharton duct but does not join the Wharton duct, running parallel to the Wharton duct and opening at its own orifice adjacent to and a short distance from the orifice of the Wharton duct (35.8%). 2) The SLG has 1 Bartholin duct that empties into the middle section of the Wharton duct (32.1%). 3) The SLG has 2 Bartholin ducts, one of which joins the Wharton duct and the other opens at its own orifice near that of the Wharton duct on the floor of the mouth (7.1%). 4) The SLG has many fine ducts (Rivinus ducts) that open at the floor of the mouth with no relation to the Wharton duct (25.0%). Of the 4 anatomic SLG duct variations, types 2 and 3 form immediate anatomic communication between the Bartholin duct and Wharton duct (39.2%). CONCLUSIONS: Several conclusions can be made from the present anatomic findings. 1) A certain proportion of Bartholin ducts open into the Wharton duct, and "recurrent ranula" after removal of the SLG can result from iatrogenic saliva leakage from the SMG through the surgically severed Bartholin duct if the aspirated fluid from the "recurrent cyst" is a thin seriflux. 2) The Bartholin duct emptying into the Wharton duct should be ligated during removal of the SLG to prevent local saliva accumulation from the SMG or even caudal "recurrence" as plunging ranula. 3) The surgical incision for SLG removal should be sutured loosely so that the surgically severed Bartholin duct with communication to the Wharton duct can reopen at the floor of mouth as an outlet for possible saliva leakage from the SMG and saliva accumulation can be avoided.

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.

Lymphoepithelial carcinoma--review of the treatment modalities and report of a rare case in the sublingual gland.

Controversies exist in the current literature regarding the treatment of lymphoepithelial carcinoma (LEC). The presented study summaries the literature controversies in the treatment of LEC and present a case of a rare LEC diagnosed in the sublingual gland of an otherwise healthy 21-year-old Arab woman. The patient was admitted for swelling in the sublingual area. A biopsy indicated a diagnosis of LEC of the sublingual gland. Treatment included a wide resection with safe margins, marginal mandibulectomy, functional neck dissection, and a radial forearm free flap for reconstruction. The patient received concomitant radiotherapy and has remained disease free since her last follow-up. The recommended primary treatment, based on the literature review, is primary surgical excision with neck dissection and optional adjuvant radiotherapy to the gland area. Chemotherapy was not found to be beneficial according to the literature.

Tuberculous osteomyelitis of the mandible with diffuse swelling of the floor of the mouth: a case report.

Primary orofacial tuberculosis (TB) is uncommon, especially with regard to the jaw. We report an unusual case for which the final diagnosis was tuberculous osteomyelitis of the mandible with cervical tuberculous lymphadenitis. The follow-up examinations for our patient showed complete regression of the swelling and healing of the mandibular lesion after 4 months of TB antibiotic therapy. The purpose of the present study was to alert clinicians to our findings and encourage them to consider oral TB in the differential diagnosis for jaw lesions with multiple enlarged cervical lymph nodes.

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.

Computer guided surgery for implant placement and dental rehabilitation in a patient undergoing sub-total mandibulectomy and microvascular free flap reconstruction.

A 58-year-old patient presented with an extensive, destructive, recurrent pleomorphic adenoma occupying the mandibular body and the soft tissues of the mouth and neck. Resection of the mandible from right ramus to left condylar process, and implant rehabilitation in both jaws with fixed bridgework was planned. Comprehensive presurgical prosthetic work up was carried out to record the existing dental relationship and guide all stages of the reconstruction. The jaw was first grafted with a segmented, fibular microvascular free-flap, which was fixed in place with a fixation plate prebent on a Rapid Prototype Anatomical Model of the jaw. Reconstruction with implant supported fixed partial dentures took place to the dental scheme planned preresection, using a computer guided approach to implant placement in the complex and unfamiliar anatomy of the extensively grafted mandible. This approach facilitated and expedited implant surgery such that treatment could take place using a minimally invasive approach relatively soon after surgery, prior to commencement of radiotherapy, and highlights the importance of a multidisciplinary approach to treatment for patients having extensive surgery to the jaws. The patient's personal assessment 2 years post surgery was recorded using 1999 University of Washington Quality of Life Questionnaire.

Schwannoma derived from lingual nerve occurring in floor of mouth.

A schwannoma is a benign tumor composed of schwann cells which forms on the periphery of nerves. We report a case of a schwannoma derived from a lingual nerve occurring in the floor of the mouth. The patient was a 27-year-old woman who presented with the complaint of a swelling in the floor of the mouth. It is difficult to distinguish a swelling from a sublingual gland tumor, cyst, or malignant tumor by MRI alone. Therefore, a biopsy and cytological examination were performed one week prior to surgery to determine whether the growth represented a malignancy. The results revealed a class II growth which was suspected to be a schwannoma. Intraoperatively, it became clear that the tumor and lingual nerve were inseparable, making excision of the nerve unavoidable. On the other hand, there was a clear border between the tumor and the sublingual gland, so it was possible to preserve the sublingual gland. In the postoperative pathological diagnosis, a definitive diagnosis was difficult based solely on H-E staining. Therefore, immunohistochemical staining was performed, resulting in a diagnosis of schwannoma. Currently, the patient is still being followed up. The results of this case indicate that preoperative aspiration biopsy cytology is useful in deciding the operative method to be employed.

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.