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.

Extracapsular dissection versus total excision for benign submandibular gland tumors.

INTRODUCTION: Benign submandibular gland tumors pose challenges in balancing treatment effectiveness and preserving gland function. This study aimed to compare gland-preserving surgery, extracapsular dissection (ECD), with total excision in managing these tumors, focusing on function preservation and recurrence rate. METHODS: Fifty consecutive patients with treatment-naïve benign submandibular gland tumors were alternatively allocated to receive ECD (n = 25) or total excision (n = 25) without randomization procedures. Intraoperative findings, postsurgical complications, subjective satisfaction, and gland function were assessed. Follow-up data were collected for a median duration of 55 months (24-80 months) to monitor recurrences. RESULTS: ECD demonstrated significant advantages, including shorter operation time, reduced bleeding, and preservation of the facial artery and vein (P < 0.05). Both groups exhibited acceptable postsurgical pain and taste sensations. Complications were minimal and similar between the two groups. ECD resulted in superior facial contour satisfaction (P = 0.030) and preserved gland function, as evidenced by salivary scintigraphy. No recurrences were observed in either group during the follow-up period. CONCLUSIONS: ECD is a practical approach for benign submandibular gland tumors, offering favorable functional outcomes, reduced surgical morbidity, shorter operation times, and improved cosmetic results.

Pleomorphic Adenoma of the Submandibular Gland in a 10-Year-Old Child: A Case Report.

We report the case of a 10-year-old girl with a painless slowly growing mass that had developed over the course of 2 years in the left submandibular area. Physical examination revealed a firm painless submandibular mass with no other associated signs. Ultrasound graphics were in favor of a sebaceous cyst. Fine-needle aspiration and magnetic resonance images (MRI) concluded to a pleomorphic adenoma of the submandibular gland. The patient underwent left submandibulectomy. Histopathological examination confirmed the diagnosis of pleomorphic adenoma arising from the submandibular gland. The postoperative course was uneventful. Pleomorphic adenoma of the submandibular gland represents a diagnostic and therapeutic dilemma in children. Preoperative evaluation, including MRI and fine-needle aspiration, is recommended. Total submandibulectomy is also recommended to minimize recurrences and to avoid malignant transformation.

Benign submandibular gland tumours: outcomes of gland-preserving excision by endoscopic or conventional approach.

Endoscope-assisted surgery is becoming a preferred technique in salivary gland surgery. However, this technique has not yet been applied in submandibular gland (SMG) preservation surgery. This retrospective study was performed to evaluate the outcomes of endoscope-assisted gland-preserving surgery through a hairline incision in patients with benign SMG tumours. The study included 38 patients with benign SMG tumours who underwent tumour excision with gland preservation: 19 who underwent local excision of the tumour through an endoscope-assisted hairline approach and 19 who received the conventional cervical approach. The feasibility of the surgical procedure, perioperative patient variables, and postoperative appearance and functional outcomes were evaluated. Patients in both groups had their tumours removed successfully with tumour-free margins. The intraoperative blood loss, postoperative amount of drainage, mean length of the incision, and unstimulated saliva flow rate did not differ between the two groups. There was no difference in the stimulated saliva flow rate between the preserved gland and unaffected SMG. The aesthetic result was better in the endoscope-assisted hairline incision group. No tumour recurrence occurred during follow-up (range 12-52 months). Thus, gland-preserving tumour dissection appears to be a safe method for benign SMG tumours, with good functional results. Furthermore, the endoscope-assisted hairline incision is a feasible method with excellent cosmetic results.

The rate of occult nodal metastasis in submandibular gland malignancies: A case series and meta-analysis.

OBJECTIVE: Submandibular gland (SMG) malignancies are exceedingly rare. Lymph node metastasis is one of the most important determinants of outcome in SMG malignancies. The aim of this study was to investigate the overall rate of occult neck nodal metastasis in SMG malignancies. STUDY DESIGN: The study design is a meta-analysis of all studies on patients with a primary SMG malignancy, without evidence of neck nodal metastasis, who underwent an elective neck dissection (END). The search strategy identified 158 papers that appeared in print from January 1980 to July 2020. All eligible patients from the Tel-Aviv Medical Center were analyzed and consolidated into a case series. A total of 12 retrospective studies that included 306 suitable patients met inclusion criteria. RESULTS: The risk for occult metastasis in primary SMG malignancies was 0.0% to 50.0%, with a fixed effect model of 19.52% (95% CI, 14.9%-24.5%). The analyzed studies included a wide range of pathologies. The most common malignancies were adenoid cystic carcinoma and mucoepidermoid carcinoma. CONCLUSIONS: The overall rate of occult neck metastasis in SMG malignancies is relatively high, and an END should be the default intervention in these cases. An END is unwarranted in tumors judged clinically to be low stage and low grade.

Management of malignant submandibular gland tumors: A 30-year experience from a single center.

OBJECTIVE: Owing to histologic heterogeneity, biological behavior, and rarity, recommendations for the treatment of malignant submandibular gland tumors (MSGT) are inconsistent. The aim of this study was to present a single-center experience in the treatment of MSGT with an emphasis on surgical treatment, including indication on elective neck dissection (END). STUDY DESIGN: Twenty-four MSGT were primary surgically treated (gland excision with neck dissection). Their records were retrospectively collected and analyzed. RESULTS: The most frequent histology was adenoid cystic carcinoma (41.6%), followed by mucoepidermoid carcinoma (25%) and carcinoma ex pleomorphic adenoma (16.7%). There were 18 elective and 6 therapeutic neck dissections. Histopathologic examination confirmed 29% (7/24) of positive neck dissection specimens. The Kaplan-Meier analysis presented rates of disease-specific survival, disease-free survival, and overall survival (OS) of 81%, 78%, and 52% at 5 years, respectively. Patients undergoing postoperative radiotherapy had significantly higher OS rates compared with patients treated with surgery alone (P = .0209). CONCLUSION: Results of this study suggest that END has questionable benefit in early stage MSGT. Elective selective neck dissection levels I-III is recommended in high-grade and advanced stage MSGT without evidence of multilevel lymphadenopathy.

Functional gland-preserving surgery for submandibular gland pleomorphic adenoma.

PURPOSE: Conventional surgery for submandibular gland tumour is total excision of the gland affected by the tumour. Gland-preserving surgery is commonly used for benign tumours arising in the parotid gland but not in the submandibular gland. This study evaluated long-term oncological and functional outcomes after gland-preserving surgery in patients with pleomorphic adenoma of the submandibular gland via the submental approach. METHODS: This longitudinal study included 105 consecutive patients with submandibular gland pleomorphic adenoma who underwent the gland-preserving surgery combined with the en-bloc resection of tumours via the submental approach. Salivary scintigraphy was performed 6 months after surgery, and ultrasonography was regularly followed. Intraoperative findings, postoperative complications, cosmetic and salivary functions, and tumour recurrence were assessed in these patients. RESULTS: Median tumour size and submental incision length were 2.0 and 3.3 cm, respectively. Median operation time and amount of blood loss were 25 min and 18.5 mL, respectively. None had marginal or lingual nerve paralysis and most patients were satisfied with incision scar and facial contour. The salivary function of the affected gland was equal to that of the unaffected gland. One patient (0.9%) had single nodular recurrence 54 months after surgery and others had no recurrence for follow-up of median 96 months. CONCLUSIONS: Pleomorphic adenoma of the submandibular gland can be safely removed by the gland-preserving surgery via the submental approach which has operation time, cosmetic and functional benefits with compromising oncological outcomes.

Primary oncocytic carcinoma of ectopic salivary gland: a unique case.

Oncocytic carcinoma of the salivary glands is an extremely rare malignant neoplasm. The parotid is the most frequently involved salivary gland, but tumours in the submandibular gland and minor salivary glands have also been described. It appears that oncocytic carcinoma of an ectopic salivary gland has not been reported so far. The unique case of a 78-year-old male patient diagnosed with oncocytic adenocarcinoma of an ectopic salivary gland is reported here. The patient underwent surgery and adjuvant radiotherapy. The clinical, morphological, and immunohistochemical features of this rare entity are also described.

Epithelial-Myoepithelial Carcinoma of the Submandibular Gland: Case Report.

Epithelial-myoepithelial carcinoma (EMC) is a rare malignant salivary gland tumor that occurs mostly in the parotid gland. We report a case of EMC of the submandibular gland in a young man. The patient was aware of a slow-growing mass in the right submandibular gland for 1 year. Clinical examination and ultrasound confirmed a right submandibular mass, 2.5 × 3 cm2 in size. Ultrasound-guided fine-needle aspiration indicated a diagnosis of pleomorphic adenoma, which was also suggested by magnetic resonance imaging. The submandibular gland tumor was excised. Immunohistochemical analysis showed carcinoma ex pleomorphic adenoma with a major epithelial-myoepithelial component. The patient was not treated with radiotherapy after surgery. No recurrence was observed during 24 months of follow-up. Because the morphology of EMC is similar to that of a benign tumor, it is frequently initially misdiagnosed. Surgery is always the most effective diagnostic and therapeutic measure for salivary gland tumors, especially those that grow slowly. Resection with negative margins is the treatment of choice for EMC; use of adjuvant radiotherapy is controversial.

Unusual presentation of submandibular lingual nerve sheath tumour as sublingual stone.

A 60-year-old woman was referred to the otolaryngologist for 18 months of left-sided tongue pain and taste changes. Surgeon-performed ultrasound of the submandibular region revealed a hyperechoic mass. Wharton's duct was dilated proximally and the submandibular gland demonstrated normal vascularity. While these findings were highly suspicious for submandibular gland sialolith, an in-office attempt at sialolithotomy suggested an alternate process or mass. After imaging failed to further elucidate an aetiology, surgical exploration revealed a well-circumscribed submandibular mass associated with the lingual nerve. The mass was removed en-bloc and pathology revealed a schwannoma of the lingual nerve.

Giant submandibular sialolipoma masquerading as huge goitre: a case report.

BACKGROUND: Sialolipoma is a rare tumour which may arise from both major and minor salivary glands and has recently been described as a variant of salivary gland lipomatous lesions. CASE PRESENTATION: We report a 54-year-old male who presented with a 7-year history of large right anterior neck swelling. He was clinically euthyroid and had no compressive or infiltrative symptoms. He sought medical attention due to the discomfort exerted by the weight of the mass and was keen for excision. The swelling appeared like a goitre but physical examination proved otherwise. Imaging was suggestive of a benign tumour arising from the right parapharyngeal fossa. The mass was surgically excised and was noted to be adherent to part of the submandibular gland. Histopathological examination revealed a new variant of benign adipocytic tumour of salivary gland or sialolipoma arising from the submandibular gland. Besides being the largest sialolipoma to be reported, there are also no reports of giant submandibular sialolipomas masquerading as a huge goitre in appearance. CONCLUSION: Submandibular sialolipomas can present in really large sizes and appear as a giant goitre. It is important to differentiate between benign lipomas from liposarcomas and tailor the management accordingly. Surgical enucleation is the preferred choice of treatment for these benign tumours with low recurrence rates.

Self-removing passive drain to facilitate postoperative care via telehealth during the COVID-19 pandemic.

BACKGROUND: Telehealth postoperative visits are an attractive strategy to minimize exposure, especially during the SARS-CoV-2 (COVID-19) pandemic. The use of a surgical drain often prevents this minimal-exposure approach in that patients return to the outpatient clinic for drain removal. METHODS AND RESULTS: Following unilateral neck dissection, the customary closed-suction drain was replaced with a self-removing, passive drain dressing to facilitate same-day discharge and telehealth postoperative follow-up. The patient removed the dressing and drain at home during a telehealth visit on postoperative day 4 and she healed favorably without any signs of infection or seroma. CONCLUSIONS: When thoughtfully applied in the appropriate clinical context, small practice adaptations like this can facilitate telehealth solutions that diminish unnecessary exposure for patients, their caregivers, and health care staff.

A case of oncocytic lipoadenoma of the submandibular gland and its diagnostic cytology challenges.

Lipomatous tumors of the salivary gland in general are rare and oncocytic lipoadenomas are even much rarer. Most of the reported cases of oncocytic lipoadenoma were located in the parotid gland and to the best of our knowledge, only four cases reported to arise from the submandibular gland. The preoperative interpretation of the aspirates taken from these lesions can be difficult and challenging. We report a case of a 41 year old gentleman who presented with submandibular gland swelling. CT scan of the neck revealed an oval-shape enhancing mass in the inferior aspect of the right submandibular gland measuring 19 × 12 mm. Fine needle aspiration biopsy (FNAB) showed hypocellular smears with few clusters of epithelial cells having oncocytic appearance. The cytologic findings were interpreted as "oncocytic lesion." Submandibular gland excision was done. Histologically, a final diagnosis of oncocytic lipoadenoma was rendered. We present this rare entity to add to the few cases reported to date and to upsurge awareness during cytologic examination of oncocytic salivary gland lesions.

Conservative and functional surgery in the treatment of salivary gland tumours.

The principle of modern oncological surgery is to conserve the functional organs or tissues as much as possible based on eradication of the tumour. For salivary gland tumours, conservative and functional salivary surgery, including partial sialoadenectomy as well as anatomical and functional preservation of the facial nerve, great auricular nerve, superficial musculo-aponeurotic system (SMAS), and Stensen's duct, has become increasingly popular. In the present review, we discuss the following aspects of conservative and functional surgery in the treatment of salivary gland tumours: (i) partial superficial parotidectomy (PP) to treat benign parotid gland tumours, (ii) modification of surgical incisions to improve cosmetic results, (iii) modification of the surgical approach to decrease complications, (iv) extracardial dissection to treat benign superficial parotid tumours, (v) partial sialoadenectomy to treat benign submandibular gland tumours, and (vi) 125I brachytherapy to preserve facial nerves. The majority of the operated parotid or submandibular glands are preserved, and surgical complications are also decreased. Conservative and functional surgery plays a significant role in maintaining normal salivary gland function and in improving patients' quality of life during the treatment of salivary gland tumours and thus should be further promoted.

Positive Surgical Margins in Submandibular Malignancies: Facility and Practice Variation.

OBJECTIVES: Identify positive margin rate in a national cohort of patients with submandibular carcinoma, identify predictors of positive margins, and associate margins with overall survival. STUDY DESIGN: Retrospective cohort. SETTING: Commission on Cancer-accredited hospitals. SUBJECTS AND METHODS: We included patients in the National Cancer Database from 2004 to 2014 who were diagnosed with submandibular carcinoma and underwent primary surgical resection. We determined the rate of positive surgical margins and associated patient, tumor, and treatment factors with positive margins via univariable and multivariable logistic regression analysis. We associated margin status with overall survival by Kaplan-Meier curve and Cox proportional hazards regression. RESULTS: We identified 1150 patients with submandibular malignancy undergoing surgical resection. Positive margin rate was 41.0%. Increased odds of positive margins were seen in patients with advanced T stage (vs T1, T3: odds ratio [OR] = 3.04, P < .001; T4a: OR = 2.89, P < .001), adenoid cystic carcinoma histology (OR = 1.54, P = .020), and those treated at nonacademic facilities (OR = 1.41, P = .008). Patients who underwent a preoperative diagnostic biopsy had decreased odds of positive margins (OR = 0.72, P = .014). Positive margins were associated with reduced overall survival (58% vs 69% 5-year overall survival, P < .001; hazard ratio = 1.49, P = .001) when controlling for patient, tumor, and management factors. CONCLUSIONS: The national positive margin rate of submandibular carcinoma is 41.0%. Preoperative biopsy and treatment at academic institutions independently decreased the risk of positive margins, and positive margins were independently associated with diminished overall survival. Positive margin rate for submandibular carcinoma may be considered a benchmark for quality of care.