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.

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.

Ultrasound in the diagnosis and differential diagnosis of enoral and plunging ranula: a detailed and comparative analysis.

PURPOSE: To develop sonographic criteria for ranula that to allow rapid and precise diagnosis, differentiation between enoral (ER) and plunging ranula (PR), and differential diagnosis from other competing pathologies in this region. METHODS: Patients who presented with or were referred with ranula between 2002 and 2022 were assessed in a retrospective study. After clinical investigation, ultrasound examinations were performed in all cases. Several sonographic parameters describing the echotexture, shape and size of ranulas, their relationship to important surrounding anatomical landmarks and the characteristic spreading pattern of ERs and PRs were elaborated and evaluated. RESULTS: 207 ranulas were included (82.12% ERs and 17.87% PRs). The ranulas were all in close anatomical relationship to the sublingual gland (SLG) and mylohyoid muscle (MM). The echo texture was hypoechoic to anechoic in 97.6% of the lesions. In comparison with ERs, PRs were larger and irregular in shape significantly more often (P = 0.0001). There were significant differences between ERs and PRs in their exact location relative to the SLG (superficial, deep, anterior, each P = 0.0001; posterior, P = 0.03) and level of the MM (above, below, above and below, P = 0.0001 each). The exact extent and plunging pattern were depicted in all PRs, but naturally in none of the ERs. CONCLUSIONS: The ultrasound criteria developed in this study, confirming previously published results, indicate that ultrasound is an excellent diagnostic tool for diagnosing ranula and differentiating between ERs and PRs.

Sublingual Gland Observed on Salivary Gland Scan.

ABSTRACT: The major salivary glands, namely, the parotid, submandibular, and sublingual glands, are important in maintaining oral cavity health. A salivary gland scan is used to evaluate the uptake and excretory function of the salivary glands. By intravenously injecting 99m TcO 4- , which is distributed like chloride ions in the body, the glands become visible on the salivary gland scan. The parotid and submandibular glands are typically appreciated on the salivary gland scan, but the sublingual gland is not. We present a rare image of a prominent sublingual gland on a salivary gland scan.

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.

Schwannoma arising from the sublingual glandular branch of the lingual nerve radiologically masquerading as sublingual gland tumor.

We report a rare case of schwannoma arising from the sublingual glandular branch of the lingual nerve radiologically masquerading as sublingual gland tumor. A 42-year-old female was referred to our department with a painless swelling in the left submandibular region. Contrast-enhanced computed tomography showed a well-circumscribed, heterogeneous low-density tumor with cystic change in the left sublingual region. Magnetic resonance imaging showed a well-circumscribed, heterogeneous sublingual tumor with low-signal intensity on T1-weighted image and high-signal intensity in T2-weighted image. The lesion was diagnosed radiologically as benign sublingual gland tumor. The patient underwent resection of sublingual gland tumor under general anesthesia. There was no definitive continuity between the tumor and the sublingual gland, and the tumor originated from sublingual glandular branch of the lingual nerve. Pathological examination of the specimen showed schwannoma with highly cellular areas (Antoni A) and hypocellular areas (Antoni B). The postoperative course was uneventful without lingual nerve palsy, and there was no recurrence 4 years after surgery.

Plunging Ranulas and Prevalence of the "Tail Sign" in 126 Consecutive Cases.

OBJECTIVES: Plunging (or diving) ranulas are extravasation pseudocysts arising from the sublingual gland that present as soft submandibular swelling. The "tail sign" has been widely reported as pathognomonic for their diagnosis. It is described as a smooth tapering comet-shaped unilocular fluid mass with its "tail" in the collapsed sublingual space (SLS) and its "head" in the posterior submandibular space. This sign is based on the premise that extravasated saliva from the sublingual gland in the SLS escapes and plunges, over the posterior edge of the mylohyoid muscle into the submandibular space. Therefore, some fluid must be present in the posterior SLS in almost all patients with plunging ranulas. This study aimed to determine the frequency of fluid seen with ultrasound (US) in the posterior SLS to corroborate the tail sign. METHODS: A total of 126 consecutive cases of surgically proven plunging ranulas were investigated with US over 13 years. The findings were reviewed retrospectively for the prevalence of fluid in the posterior SLS. RESULTS: Thirteen patients (10.3%) showed SLS fluid on US images. Most showed fluid extension through a mylohyoid dehiscence. Only 2 patients (1.6%) showed fluid within the posterior SLS, and 1 patient alone in this entire study showed all of the components of the classically described tail sign. CONCLUSIONS: This largest ever radiologic study showed low prevalence of the tail sign in 2 of 126 patients. A mylohyoid dehiscence was the more common route for extravasation. Absence of the tail sign does not exclude the diagnosis of a plunging ranula.

Differentiation of sublingual thyroglossal duct cyst from midline dermoid cyst with diffusion weighted imaging.

PURPOSE: to differentiate sublingual thyroglossal duct cyst (TGDC) from midline dermoid cyst (DC) with diffusion weighted imaging (DWI). MATERIALS AND METHODS: Retrospective analysis of 22 consecutive patients (11 male and 11 female aged 5-15 years) with midline cystic lesion at floor of mouth. They underwent DWI of floor of mouth. Apparent diffusion coefficient (ADC) of the cystic lesions was calculated and correlated with surgical findings. RESULTS: The mean ADC value of TGDC of 1st observer was (2.20 ±â€¯0.28 × 10-3 mm2/s) and of 2nd observer was (2.28 ±â€¯0.27 × 10-3 mm2/s) was significantly higher than that of DC (P = 0.001) whose ADC of 1st observer was (1.55 ±â€¯0.15 × 10-3 mm2/s) and of 2nd observer was (1.53 ±â€¯0.11 × 10-3 mm2/s). There was excellent inter-observer agreement of both readings (r = 92%, P = 0.001). When ADC of 1.76 and 1.62 × 10-3 mm2/s was used as a threshold value for differentiating TGDC from DC, the best results were obtained with area under the curve of 0.94 and 0.96, accuracy of 90% and 86%, sensitivity of 91% and 91%%, specificity of 90% and 80%, negative predictive value of 90% and 88% and positive predictive value of 92% and 84% of both reviewers respectively. CONCLUSION: DWI is reliable and reproducible imaging modality for differentiation sublingual TGDC from DC.

Anatomic and magnetic resonance imaging features of the salivary glands in the dog.

The aim of the study was to describe the anatomical location and the topographic relationship of the major salivary glands to associated tissues in six dog cadavers by high-field magnetic resonance imaging (MRI) and gross anatomical dissections and sections. Spin-echo T1-weighted and T2-weighted magnetic resonance (MR) images were obtained in transverse, sagittal, and dorsal planes with a 1.5 T magnet. The digastric muscle, the external auditory canal, the eyeball, and the medial pterygoid muscle were the most consistent topographic landmarks for the identification of the mandibular, parotid, and zygomatic salivary glands, respectively. The best differentiation and contrast of the glands was observed in the T2-weighted images. In contrast with the osseous and muscular tissues, the salivary glands appeared hyperintense in greater or lesser measure except for the parotid gland, which displayed a hypointense signal. The rostral part of the monostomatic sublingual gland was precisely located between the tongue and the mylohyoid muscle. The best delineation between the caudal part of the monostomatic sublingual gland and the cranial aspect of the mandibular gland was observed in dorsal MRI at the level of the palate and the branches of the superior dental arcade. Good evaluation of the mandibular, parotid, caudal part of the monostomatic sublingual, zygomatic and buccal ventral glands was possible in sagittal MRI in close relation with the external auditory canal and the temporomandibular joint. The ventral buccal glands were also observed near the buccal vestibule as was the palatine glands within the soft palate.

Sublingual gland excision for the surgical management of plunging ranula.

OBJECTIVE: A plunging ranula is a pseudocystic collection of mucin extravasated from the sublingual gland into the floor of mouth and through the mylohyoid muscle into the neck. While the lining of a ranula is non-secreting and resection of the sublingual gland is adequate for simple sublingual mucoceles, many surgeons attempt to address plunging ranulas with extensive transoral and transcervical dissections. We review our experience managing plunging ranulas with intraoral sublingual gland excision and ranula drainage alone. METHODS: This is a case series of patients with plunging ranulas who underwent transoral sublingual gland excision and ranula drainage in the past 10 years at the Massachusetts Eye and Ear. All ranulas were confirmed by radiographic imaging. Data were gathered from the medical record and telephone surveys. RESULTS: Twenty-one patients with 22 distinct ranulas underwent this surgical approach. Average ranula size on imaging was 4.3 cm (SD = 1.3). Thirteen patients with 14 ranulas were followed up for greater than 6 months while the remaining 7 patients were lost to follow-up. Median follow-up for the 13 patients was 30 months (range 6 to 80). One ranula recurred requiring excision of residual sublingual gland (7%). One patient developed a local infection that was treated with antibiotics (7%). No long term complications were reported. CONCLUSION: Simple transoral excision of the sublingual gland with ranula drainage is sufficient for treatment of plunging ranulas. It is essential to obtain a full resection of the gland to prevent relapse. This limited approach has low rates of complications and ranula recurrence.

Imaging of Submandibular and Sublingual Salivary Glands.

The submandibular and sublingual salivary glands are major salivary glands with a wide spectrum of pathologic conditions. The corresponding spaces along the floor of mouth have complex anatomy, best evaluated with cross-sectional imaging. The spectrum of diseases in these regions varies from simple infection to advanced malignancy, not just from the gland itself but also from the surrounding structures. The most common abnormalities in these spaces are inflammatory and infectious, and computed tomography is currently the most common imaging modality used. The anatomy of these spaces is much better depicted with MR; however, all the modalities have their unique roles.

Monitoring the biochemical alterations in hypertension affected salivary gland tissues using Fourier transform infrared hyperspectral imaging.

Fourier transform infrared spectroscopy (FTIR) imaging has been applied to investigate biochemical differences between salivary glands from control and hypertensive rats. Male Sprague-Dawley rats were divided into two groups including a control group and another hypertension group that were treated orally, with N-nitro-l-arginine methyl ester (l-NAME) via drinking water for 3 weeks to develop hypertension. In the control group, rats were treated with only drinking water for 3 weeks. The formalin-fixed paraffin embedded tissue specimens from submandibular and sublingual glands were analysed with a FTIR focal plane array imaging spectrometer and multi-composite images of all tissue sections were analysed simultaneously using Unsupervised Hierarchical Cluster Analysis (UHCA) and the extracted spectra were further analysed using Partial Least Squares Discriminant Analysis (PLS-DA). In general, hypertension affected salivary gland tissues were characterised by higher concentrations of triglycerides as evidenced by an increase in the 1745 cm-1 band. Higher concentrations of carbohydrates and proteins were also observed in the hypertensive group along with a decrease in bands associated with nucleic acids. PLS-DA scores plots provided good differentiation in sublingual gland tissues between control (n = 3734 spectra) and hypertension (n = 4538) and also in submandibular gland tissues between control (n = 5051) and hypertension (n = 4408). We have shown that FTIR imaging can be used to differentiate the macromolecular information between physiological and pathological conditions in tissue biopsy specimens. In the next phase, we will investigate the infrared predictive markers of hypertension in biofluids including serum and saliva using attenuated total refection spectroscopy.

Diagnostic performance of MR imaging of three major salivary glands for Sjögren's syndrome.

OBJECTIVE: We analyzed the diagnostic performance of the MR imaging findings of the parotid, submandibular, and sublingual glands to discriminate between patients with and without Sjögren's syndrome. METHODS: We retrospectively analyzed the correlation between the MR imaging and histopathological findings obtained from 69 patients with clinically suspected Sjögren's syndrome. We evaluated the heterogeneous signal intensity distribution on T1- and T2-weighted images, the multiple high-signal-intensity spots on MR sialograms, and the volume of the parotid, submandibular, and sublingual salivary glands. RESULTS: The multiple high-signal-intensity spots in the parotid gland showed the highest sensitivity and diagnostic accuracy (82% and 83%, respectively). In addition, the multiple high-signal-intensity spots and the heterogeneous signal intensity distribution in the submandibular gland showed high specificity (100% and 88%, respectively). The volume of the submandibular gland, but not that of the parotid or sublingual gland, was smaller in patients with Sjögren's syndrome. CONCLUSIONS: The presence of multiple high-signal-intensity spots on an MR sialogram in the parotid gland should be considered the best diagnostic indicator for Sjögren's syndrome. The presence of spots, heterogeneity, and the change to smaller volumes in the submandibular gland were also helpful because of their high specificity, particularly in advanced cases.

Plunging Ranulas Revisited: A CT Study with Emphasis on a Defect of the Mylohyoid Muscle as the Primary Route of Lesion Propagation.

OBJECTIVE: The purpose of this study was to clarify the pathogenesis of plunging ranulas in regard of the pathway of lesion propagation using CT scans. MATERIALS AND METHODS: We retrospectively reviewed CT scans of 41 patients with plunging ranula. We divided plunging ranulas into two types: type 1 was defined as those directly passing through a defect of the mylohyoid muscle with the presence (type 1A) or absence (type 1B) of the tail sign and type 2 as those through the traditional posterior route along the free edge of the mylohyoid muscle. Images were also analyzed for the extent of the lesion in respect to the spaces involved. As for type 1 lesions, we recorded the location of the defect of the mylohyoid muscle and the position of the sublingual gland in relation to the defect. RESULTS: CT scans demonstrated type 1 lesion in 36 (88%), including type 1A in 14 and type 1B in 22, and type 2 lesion in 5 (12%). Irrespective of the type, the submandibular space was seen to be involved in all cases either alone or in combination with one or more adjacent spaces. Of the 36 patients with type 1 lesions, the anterior one-third was the most common location of the defect of the mylohyoid muscle, seen in 22 patients. The sublingual gland partially herniated in 30 patients. CONCLUSION: Our results suggest that the majority of plunging ranulas take an anterior shortcut through a defect of the mylohyoid muscle.

Case of mucoepidermoid carcinoma of the sublingual gland accompanied with extensive dystrophic calcification and intratumoral bone formation.

BACKGROUND: Mucoepidermoid carcinoma (MEC) is the most common salivary gland malignancy. Although the histopathological findings are diverse, no case of salivary MEC accompanied with bone formation has been reported. Herein, we present the first case of MEC with intratumoral bone formation. METHODS AND RESULTS: A case of a 75-year-old female patient with an asymptomatic mass of the oral floor is presented. Enhanced CT revealed a tumorous mass with radiopaque materials in the oral floor. A pathological diagnosis of MEC was obtained by biopsy. Radical surgery was performed with the patient under general anesthesia. The resected specimen was finally diagnosed as MEC. Histopathologically, hyalinization, and dystrophic calcification were conspicuous within the stromal components of the tumor. Interestingly, ectopic bone formation was also found in the tumor tissues. Bony materials with osteocyte-like cells were also observed in a series of dystrophic calcification. CONCLUSION: These findings suggest that bony materials are associated with dystrophic calcification.

Types of sublingual gland herniation observed during sonography of plunging ranulas.

OBJECTIVES: Plunging ranulas have been shown to have a common trio of mylohyoid defects, sublingual gland herniation, and submandibular space fluid collections. The herniated sublingual gland may be susceptible to subclinical trauma. The purpose of this study was to investigate the frequency of occurrence of various types of active sublingual gland herniation observed during diagnostic sonographic studies. METHODS: Sonographic findings of sublingual gland herniation from 76 patients with a total of 80 plunging ranulas are presented. All sublingual gland herniations, including those on the contralateral side, were documented at the time of the examinations and reviewed later. The sonographic appearances of active sublingual gland herniation were given the names "slide," "wobble," "mushroom," and "retrusion" to reflect the observed movement. RESULTS: Mylohyoid defects were found in 98% of plunging ranulas. The different types of sublingual gland herniation observed were as follows: slide in 77.8%, wobble in 11.1%, mushroom in 9.7%, and retrusion in 1.4%. The unaffected contralateral side showed a wobble in 44.8% of cases and slide in 8.9%. CONCLUSIONS: Sublingual gland herniation through mylohyoid defects is easily missed if one is unaware. Prior knowledge of the various types of sublingual gland herniation and their frequencies of occurrence are very helpful during diagnostic sonographic examinations.

Dynamic sonography and CT findings of unilateral submandibular gland agenesis associated with herniated hypertrophic sublingual gland.

Congenital agenesis of the submandibular gland is uncommon. We report dynamic sonography and CT findings of the unilateral submandibular gland agenesis associated with herniated hypertrophic sublingual gland tissue through the mylohyoid gap. The dynamic sonography examination applied at rest and during the modified Valsalva maneuver demonstrated hypertrophied sublingual gland prolapsing to the anterior part through the mylohyoid gap. This may be the first reported case describing dynamic sonography findings of this entity. CT confirmed the agenesis of the left submandibular gland and compensatory hypertrophy of the ipsilateral sublingual gland.

Bilateral plunging ranula: two case reports and a review of the literature.

AIM: Presentation of two bilateral plunging ranula cases and then review of the plunging ranula literature to understand current concepts on aetiology, imaging for diagnosis and management. METHOD: A literature review using PubMed (92 papers) and Google Scholar (18 papers) has revealed only 18 cases of bilateral plunging ranulas ever published and with the addition of the two cases presented this makes a total of 20. RESULTS: These cases are reviewed and information related to aetiology and management is presented. The cause appears to be a combination of mylohyoid dehiscence, racial predisposition and previous trauma to the mouth/face or previous oral surgery. CONCLUSION: Plunging ranula are a rare cause of bilateral and unilateral neck swellings but more common in Maori, Polynesian and Asian people. Their cause is multifactorial and ultrasound scan (USS) is the current investigation of choice. Management relies on excision of the sublingual gland with the cystic contents via a trans-oral approach.