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.

Response of the mouse sublingual gland to spaceflight.

The ultrastructure and immunohistochemistry of secretory proteins of sublingual glands were studied in mice flown on the US space shuttles Discovery [Space Transportation System (STS)-131] and Atlantis (STS-135). No differences in mucous acinar or serous demilune cell structure were observed between sublingual glands of ground (control) and flight mice. In contrast, previous studies showed autophagy and apoptosis of parotid serous acinar cells in flight mice. The expression of parotid secretory protein (PSP) in sublingual demilune cells of STS-131 flight mice was significantly increased compared with ground (control) mice but decreased in STS-135 flight mice. Similarly, expression of mucin (MUC-19) in acinar cells and expression of the type II regulatory subunit of protein kinase A (PKA-RII) in demilune cells were increased in STS-131 flight mice and decreased in STS-135 flight mice, but not significantly. Demilune cell and parotid protein (DCPP) was slightly decreased in mice from both flights, and nuclear PKA-RII was slightly increased. These results indicate that the response of salivary glands to spaceflight conditions varies among the different glands, cell types, and secretory proteins. Additionally, the spaceflight environment, including the effects of microgravity, modifies protein expression. Determining changes in salivary proteins may lead to development of non-invasive methods to assess the physiological status of astronauts.

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.

Transoral Robotic Surgery with Sialendoscopy for a Plunging Ranula.

BACKGROUND/AIMS: Plunging ranulas are oral mucoceles that often cannot be visualized directly and thus can be challenging to diagnose and differentiate from other neck lesions. Surgery is the preferred treatment option, but the optimal approach requires careful consideration. METHODS: We report the case of a plunging ranula presenting as an isolated neck mass in a 33-year-old patient. This patient was treated with a novel transoral robotic surgery (TORS)-assisted approach aided by sialendoscopy. RESULTS: TORS allowed for exceptional visualization and access to the plunging ranula and sublingual gland while avoiding critical neurovascular structures. The patient healed well without recurrence or complications. CONCLUSION: The TORS approach with sialendoscopy for plunging ranulas described in this case report shows promise and should be considered for certain clinical presentations. Additional studies evaluating the effectiveness of TORS for plunging ranulas may be indicated.

A cadaveric study on mylohyoid herniation of the sublingual gland.

The purpose of this study was to document the presence of a sublingual gland (SLG) herniating inferiorly through the mylohyoid muscle into the submandibular area. A total of 100 half-heads of 50 adult Korean cadavers were enrolled in this study. The floor of the mouth was dissected from the neck, and mylohyoid muscle patency and position of the sublingual gland were evaluated. Demographic factors of the donor and characteristics of the herniation were evaluated. Herniation was found in 29 (58.0 %) of the 50 cadavers or 42 of the 100 half-heads. Herniation was more frequently observed in females than in males (p = 0.009). However, no laterality was observed. Classifying the location of SLG herniation from the midpoint of the mandible to the hyoid bone into 3 regions, 32 (63 %) of herniations were found in the anterior one-third. No ranula formation was observed. The size and weight of normal glands tended to be larger than those of herniated glands, but no statistical significance was observed. An SLG hernia is a very common condition and is more frequently observed in females. As such, SLG herniation should be considered when a submental neck mass is evaluated.

Tumors of the sublingual gland: a national clinicopathologic study of 29 cases.

Tumors of the salivary glands are a heterogeneous group of diseases most often originating in the major salivary glands. Only a minor proportion of mainly malignant tumors arise in the sublingual gland. Due to the rarity of sublingual gland tumors (SGTs), little is known about the clinicopathologic characteristics, prognostic factors, and clinical course. We present a large national series of histopathologically revised SGTs from the past 35 years in Denmark with clinicopathologic correlation. Twenty nine cases were identified, of which 96.6 % were malignant and 16/28 (57.1 %) were adenoid cystic carcinomas (ACC). Patient demography was similar to salivary gland tumors in other locations. All fine needle aspiration cytologies (FNACs) interpreted as benign were from ACCs. Metastatic disease was found in 12.5 % of ACCs at diagnosis with one third of all ACC patients having metastases at the end of follow-up. Stage >II and T-stage >2 were significantly associated with shortened disease-specific survival (DSS) (p = 0.005 and <0.001, respectively), whereas perineural invasion and involved margins was not. No parameters were associated with disease-free survival. In conclusion, the majority of SGTs are malignant, most frequently ACC with a high rate of metastatic spread. The diagnostic value of FNAC in SGTs seems inferior to what is found for other major salivary glands. DSS is determined by stage and T-stage and not by histopathological parameters. International collaboration is warranted to confirm and elaborate these findings in larger materials.

[Surgery in Salivary Gland Diseases]. / Operative Therapie bei Erkrankungen der großen Kopfspeicheldrüsen.

Salivary gland surgery is a common procedure in otorhinolaryngology. Parotidectomy and submandibulectomy represent the treatment of choice in benign and malignant tumors of the salivary glands. However, as to this point there are no guidelines are concerning diagnostic and therapeutic approaches of benign and malignant tumors of the salivary gland. Since the introduction of sialendoscopy there are new diagnostic and therapeutic procedures available in treating obstructive sialadenitis. The present article provides an overview of anatomical principles, diagnostic approaches and the most important salivary gland surgeries for future otorhinolaryngologists under specialist training.

[Cerebral infarction in a patient with primary Sjogren's syndrome: a case report and literature review].

Sjogren's syndrome (SS) is a chronic autoimmune disorder characterized by lymphocytes infiltration in the exocrine glands. Central nervous system complications of primary SS are not rare, but ischemic stroke has been rarely reported. Here we report a 43-year-old female with a two-year history of primary SS, presenting with sudden cerebral infarction. Her primary SS was diagnosed on the basis of clinical features, high levels of serum anti-SSA and anti-SSB antibodies, salivary gland secretion evaluation and positive sublingual gland biopsy results. Magnetic resonance imaging revealed infarct lesions in the parietal and occipital lobes, as well as in the left basal ganglia. Magnetic resonance angiography showed a remarkable stenosis in the left middle cerebral artery. Other differential diagnoses were ruled out. Corticosteroid and immunosuppressor, together with anti-platelet and statin were effective, and the patient recovered quickly without sequelae. Based on these findings, vasculitis due to primary SS should be considered among the causes of stroke. The literature was reviewed and the relationship between primary SS and cerebral infarction explored. The pathogenesis of ischemic stroke in primary SS is still unknown and warrants further studies.

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.

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.

Plunging ranula: an unusual multilocular presentation.

A 32-year-old male patient who was HIV positive presented at the Wits Oral Health Centre complaining of a large swelling of the left submandibular region of three years' duration. The swelling was nontender, soft and doughy on palpation and appeared to be crossing the midline. Bilateral submandibular and submental lymphadenopathy was present. Intraorally the lesion caused considerable elevation of the floor of the mouth and impaired the flow of saliva. Fluid from the lesion was aspirated and the patient sent for MRI examination. These images revealed a multilocular cystic lesion causing disruption of the mylohyold muscle. The aspirate consisted of a thick, bloody fluid which tested positive for salivary amylase. A provisional diagnosis of plunging ranula was made. The multilocular nature of the lesion seen on MRI prompted a more extensive surgical approach in order to prevent recurrence. Consequently the sublingual gland was removed via an intraoral approach while the multilocular cyst was dissected by means of a submandibular approach in order to effect complete removal. Microscopic examination of the submitted specimen confirmed the clinical diagnosis of a plunging ranula.

[An evaluation of the radioprotective effect of vitamin E on the salivary glands of radioactive iodine in rats]. / Radyoaktif iyot verilen siçanlarda tükürük bezleri üzerinde E vitaminin radyasyondan koruyucu etkisinin degerlendirilmesi.

OBJECTIVES: This study aims to examine the acute effect of vitamin E on salivary gland of radioactive iodine (RAI; 131I)-induced rats and to evaluate whether vitamin E have a radioprotective effect. MATERIALS AND METHODS: Sixteen Wistar albino rats were used in the study. The rats were divided into two groups. The first group was administered (131)I orally and 1 ml physiological saline. The second group was administered (131)I and 1 ml vitamin E intraperitoneally. Vitamin E was started two days before the RAI therapy and continued for seven days. On the eighth day, salivary glands were removed and evaluated histopathologically. RESULTS: There was no statistically significant difference between the groups. The changes in the interstitial space of all glands and in acinar epithelial cells of parotid and sublingual glands were observed in a lower number of the rats of vitamin E group, compared to the controls. Except panacinar inflammation, histopathological changes in acinar epithelial cells of the submandibular gland were noticed in a lower number of the rats of vitamin E group. CONCLUSION: Considering the changes in the interstitial space and acinar epithelial cells in a lower number of the rats of vitamin E group, we conclude that vitamin E may have protective effects for interstitial space of all glands and acinar epithelial cells of the parotid and sublingual glands during the acute period.

One case of sublingual gland mucosa-associated lymphoid tissue lymphoma.

Mucosa-associated lymphoid tissue (MALT) lymphoma is a type of marginal zone B-cell lymphoma that occurs outside the lymph nodes in mucosal tissue. It accounts for 6-8 % of non-Hodgkin's lymphomas. MALT lymphoma of the salivary gland is a rare disease, with primary tumors in the salivary gland accounting for 2-5 % of salivary gland tumors. The most common site is the parotid gland (80 %), followed by the submandibular gland (14 %), minor salivary glands, and sublingual gland (5 %). Patients with salivary gland MALT lymphoma often have autoimmune diseases such as Sjogren's syndrome and rheumatoid arthritis. Primary malignant tumors of the sublingual gland account for less than 1 % of cases, and preoperative diagnosis is difficult, often requiring biopsy for confirmation. To our knowledge, there are no reports of MALT lymphoma arising from the sublingual gland. We report a case of MALT lymphoma originating from the sublingual gland in a patient with a history of hypertension, diabetes, cerebral infarction, and non-traumatic numbness of the right lower limb.

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.

Double thyroglossal duct cyst involving the floor of the mouth and sublingual gland region.

Thyroglossal duct cysts are the most common congenital neck masses that develop during childhood. The masses develop from remnants of thyroglossal ducts and typically appear as midline neck masses. Double thyroglossal cysts and the complete failure of the obliterative process of thyroglossal duct are very rare in the literature. This manuscript describes a rare case of double thyroglossal duct, localized above the geniohyoid muscles affecting the floor of the mouth region and sublingual gland. The diagnostic criteria of the tumor and surgical procedure are discussed. To our knowledge, only 3 similar cases were reported in the literature.

Postmortem investigation of mylohyoid hiatus and hernia: aetiological factors of plunging ranula.

The mylohyoid hiatus and hernia were discovered in the nineteenth century and were considered to explain the origin of the plunging ranula from the sublingual gland. This formed the rationale for sublingual sialadenectomy for the treatment of plunging ranula. However, a more recent, extensive histological investigation reported that hernias contained submandibular gland, which supported an origin of the plunging ranula from the submandibular gland and submandibular sialadenectomy for the treatment of plunging ranula. We therefore decided to investigate the occurrence and location of the hiatus and the histological nature of the hernia. Twenty-three adult cadavers were dissected in the submandibular region. The locations and dimensions of mylohyoid hiatuses were measured before taking biopsies of hernias. Hiatuses with associated hernias were found in ten cadavers: unilateral in six; and bilateral in four, in one of which there were three hiatuses. Sublingual gland was identified in nine hernias and fat without gland in six. This investigation supports clinical and experimental evidence that the plunging ranula originates from the sublingual gland and may enter the neck through the mylohyoid muscle. It confirms the rationale of sublingual sialadenectomy for the treatment of plunging ranula.

Temporal histological changes in lacrimal and major salivary glands in mouse models of Sjogren's syndrome.

BACKGROUND: Evidence in imaging studies suggests that there may be differences in glandular involvement in Sjogren's syndrome (SS) depending on the stage of the disease. No detailed histological studies are available to show if there are any such difference in glandular involvement at various time periods and stages of SS. This cross sectional study examines the inflammatory changes in mouse models of SS at various ages. METHODS: The histological changes in major salivary and lacrimal glands were studied at ages of 3, 6, 9, 12, 15 and 18 months in both sexes in well characterized mouse models of SS, non-obese diabetes mouse and Interleukin-14 alpha-transgenic mice. RESULTS: Our results indicate that early inflammation concurrently occur in submandibular and lacrimal glands around the age of 6 weeks. Parotid glands are involved much later in the course of SS with less severe inflammation. Sublingual glands are rarely involved. CONCLUSIONS: Our conclusions are that SS may be an organ specific disease with early inflammation occurring in submandibular and lacrimal glands, followed by the parotid. Non organ specific events occur in later courses of the disease. The understanding of the disease progression is important in tailoring early local therapeutic interventions before complete destruction of salivary and lacrimal glands.

[Sublingual gland amyloidosis causing obstructive sleep apnea hypopnea syndrome: a case report and review of the literatures].

OBJECTIVE: To improve the understanding of sublingual gland amyloidosis causing obstructive sleep apnea hypopnea syndrome(OSAHS). METHODS: A case of sublingual gland amyloidosis causing OSAHS diagnosed in april 2012 was reported and the related literatures were reviewed. The literature review was carried out respectively with "amyloidosis, sublingual gland, obstructive sleep apnea hypopnea syndrome", as the search terms in Wanfang Data and PubMed by November 2012. RESULTS: A case of 74 year-old male patient was admitted to the hospital because of snoring for 5 years, sleep apnea for 1 year and arousal for 1 month. After admission, polysomnography showed severe OSAHS, physical examination showed redundant the sublingual gland. Enhanced CT scanning showed soft tissue masses at the sublingual gland. Abdominal B ultrasonic and CT also showed a spaces-occupying lesion in the left retroperitoneal. B-guided core needle biopsy was performed in the left retroperitoneal. Pathology report showed amyloidosis. Subsequently, sublingual gland mass resection was performed. Pathology report after operation showed amyloid deposits staining with Congo red, which gives it a characteristic green birefringence in polarised light. Accordingly, it was diagnosed as sublingual gland amyloidosis. The symptoms of snore and sleep apnea were disappeared after operation.So far, there was no local recurrence with 10 months follow-up. A total of 3 literatures were received in Wanfang Data, including 2 of macroglossia amyloidosis causeing OSAHS case report and one of retrospective study. There were no reports about sublingual gland amyloidosis in Wanfang Data. A total of 5 literatures were received in Pubmed, including 2 of sublingual gland amyloidosis case report, 2 of macroglossia amyloidosis causeing OSAHS case report and one of retrospective study. However, there were no reports about sublingual gland amyloidosis causing OSAHS. CONCLUSIONS: Amyloidosis rarely occurred in the sublingual gland and is easy to be misdiagnosed and missed diagnosed, which can causing severe OSAHS. To make a definite diagnosis, histopathology and staining with Congo red are needed and a characteristic green birefringence in polarised light is a reliable marker for diagnosis. After sublingual gland mass resection, the patient had good prognosis.

Amylase as a Diagnostic Tool for Plunging Ranula: Clinical Series and Description of the Technique.

OBJECTIVES: This study describes a technique of measurement for neck cyst amylase content and reviews the experience of a tertiary referral center for cases of suspected plunging ranula. METHODS: A retrospective study was performed at the Manukau Surgical Center in Auckland, New Zealand. Patients with a possible diagnosis of plunging ranula based on clinical presentation and diagnostic aspiration of the cyst contents were included. Demographic data, imaging and laboratory findings were collected, along with findings from surgery and histology. The technique for measuring the amylase of the aspirated cyst contents was also carefully recorded. RESULTS: The 37 cases of confirmed plunging ranula included in this study had a submandibular cystic swelling that was aspirated. Imaging features consistent with a plunging ranula were seen in 89% of the study group. All cases had detectable levels of amylase of ≥3 U/L in the ranula contents. There was large variability (range: 5-560 U/L) in the concentration of amylase, with 70% of the cases demonstrating an amylase concentration below 200 U/L. Aspirates were typically described as viscous (87.5%) and yellow or straw-colored. CONCLUSION: The combination of clinical presentation, imaging and the presence of amylase in the cyst contents is diagnostic for plunging ranula. LEVEL OF EVIDENCE: 4 Laryngoscope, 133:535-538, 2023.