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.

Prognostic factors for sublingual gland carcinoma: a population-based Surveillance, Epidemiology and End Results database study.

OBJECTIVE: To identify the clinical features and prognostic factors for sublingual gland carcinoma. METHODS: This was a case-control study. Patients with surgically treated sublingual gland carcinoma were retrospectively included in the Surveillance, Epidemiology and End Results database and divided into adenoid cystic carcinoma (ACC) and non-ACC groups. Primary outcome variables were disease-specific survival (DSS) and overall survival (OS). Prognostic factors for each group were analyzed using Cox models. RESULTS: We included 251 patients (115 men and 136 women). Compared with the non-ACC group, the ACC group had a larger average tumor size and received more adjuvant radiotherapy. In patients with ACC, the pathologic grade had an independent impact on DSS and OS, and patients who were undergoing adjuvant chemotherapy had worse DSS than those who were not receiving chemotherapy. In the non-ACC group, pathologic grade IV, lymph node metastasis, and adjuvant chemotherapy were associated with poor DSS and OS, and tumor extension predicted worsening DSS. CONCLUSIONS: In sublingual gland carcinoma, the pathologic grade and adjuvant chemotherapy were the most important prognostic factors, whereas lymph node metastasis had a negative impact in non-ACC patients but not in ACC patients.

Palisading Adenocarcinoma: A Morphologically Unique Salivary Gland Tumor With a Neuroendocrine-like Appearance and a Predilection for the Sublingual Glands of Women.

Adenocarcinoma, not otherwise specified (NOS) is a heterogenous group of salivary gland tumors that likely contains distinct tumors that have not yet been characterized. Indeed, in recent years, cases previously diagnosed as adenocarcinoma, NOS have been recategorized into novel tumor designations such as secretory carcinoma, microsecretory adenocarcinoma, and sclerosing microcystic adenocarcinoma. We sought to describe a distinctive, hitherto-undescribed salivary gland tumor encountered in the authors' practices. Cases were pulled from the surgical pathology archives of the authors' institutions. Histologic, immunohistochemical, and clinical findings were tabulated, and targeted next-generation sequencing was performed on all cases. Nine cases were identified, arising in 8 women and 1 man ranging from 45 to 74 years (mean, 56.7 y). Seven tumors (78%) arose in the sublingual gland, while 2 (22%) arose in the submandibular gland. The cases shared a distinctive morphologic appearance. They were biphasic, with ducts scattered among a predominant polygonal cell with round nuclei, prominent nucleoli, and pale eosinophilic cytoplasm. These cells were arranged as trabeculae and palisaded as pseudorosettes around hyalinized stroma and vessels, resembling a neuroendocrine tumor. Four of the cases were well-circumscribed, while the remaining 5 showed infiltrative growth including perineural invasion in 2 (22%) and lymphovascular invasion in 1 (11%). Mitotic rates were low (mean, 2.2/10 HPFs); necrosis was absent. By immunohistochemistry, the predominant cell type was strongly positive for CD56 (9 of 9) and variably positive for pan-cytokeratin (AE1/AE3) (7 of 9) with patchy S100 (4 of 9), but negative for synaptophysin (0 of 9) and chromogranin (0 of 9), while the ducts were strongly positive for pan-cytokeratin (AE1/AE3) (9 of 9) and CK5/6 (7 of 7). Next-generation sequencing did not reveal any fusions or obvious driver mutations. All cases were resected surgically, with external beam radiation also done in 1 case. Follow-up was available in 8 cases; there were no metastases or recurrences after 4 to 160 months (mean, 53.1 mo). A dual population of scattered ducts with a predominance of CD56-positive neuroendocrine-like cells characterizes a unique salivary gland tumor which is often encountered in the sublingual glands of women, for which we propose the term "palisading adenocarcinoma." Although the tumor was biphasic and had a neuroendocrine-like appearance, it lacked convincing immunohistochemical evidence of myoepithelial or neuroendocrine differentiation. Although a subset showed unequivocally invasive growth, this tumor appears to behave in an indolent manner. Moving forward, recognition of palisading adenocarcinoma and its separation from other salivary adenocarcinomas, NOS will facilitate a better understanding of the characteristics of this previously unrecognized tumor.

Non-neoplastic salivary gland diseases in children: a 10-year review at a tertiary center.

OBJECTIVES: Non-neoplastic salivary gland diseases are rare in children. This paper aims to present the spectrum of encountered non-neoplastic salivary gland diseases at a tertiary center by describing the demographics, clinical characteristics, and outcomes in this patient population. METHODS: A review of electronic medical records was performed from 2010 until 2020. Relevant data were retrieved and charted according to the type of salivary gland disease. A comparison between diseases was made for demographics, presenting symptoms, treatment, and outcomes. RESULTS: Fifty patients with 11 different non-neoplastic salivary gland diseases were identified. Sialolithiasis was the most prevalent condition (12/50), with 83% localized in the submandibular gland. In contrast to gender, age, and symptoms, the location of pathology was significantly associated with the diagnosis (p < 0.001). In patients with sialolithiasis, a hybrid procedure (combined endoscopy and lithotomy) resulted in 100% resolution of symptoms. For (plunging) ranula, marsupialisation had a relative risk of recurrence of 9.6 compared to (partial) extirpation of the sublingual gland. CONCLUSIONS: Children with salivary gland diseases present with overlapping symptoms, making clinical diagnosis challenging. The present study may aid physicians and specialists in diagnosing the most prevalent conditions in children. Although no gold standards exist for their treatment, hybrid procedures (sialolithiasis) and subglingual gland extirpation (ranula) showed superior results over alternatives.

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.

Lymphomas Affecting the Sublingual Glands: A Clinicopathological Study.

BACKGROUND: Lymphomas affecting the sublingual glands are extremely rare and very few case reports are currently available. Therefore, the aim of the current study is to describe the clinicopathological features of a series of lymphomas involving the sublingual glands. METHODS: Cases diagnosed in four pathology services were assessed and the formalin-fixed paraffin-embedded tissue blocks were retrieved for diagnosis confirmation. Clinical data were obtained from patients' medical files. RESULTS: We obtained seven cases of lymphomas in the sublingual glands, representing two follicular lymphomas, two diffuse large B cell lymphomas not otherwise specified (DLBCL NOS), two extranodal marginal zone B cell lymphoma of mucosa-associated lymphoid tissue (MALT lymphomas) and one mantle cell lymphoma (MCL). In all cases the tumor cells infiltrated the glandular parenchyma, although in two of them the neoplastic cells were located more superficially and permeated the glandular acini and ducts. Clinically, the tumors presented as asymptomatic nodules and two patients (affected by DLBCL NOS and MCL) died, while the other five patients remained alive at last follow-up. CONCLUSION: Lymphomas affecting the sublingual glands are usually of the mature B cell lineage, often represent low-grade subtypes and may clinically resemble other more common lesions in the floor of the mouth like salivary gland tumors.

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.

Modified micromarsupialization technique as an alternative primary treatment for ranulas: A case series in a resource-challenged economy.

OBJECTIVES: Ranula is one of the commonest salivary gland cysts that mostly occur due to mucus extravasation from the sublingual salivary gland. Treatment of this lesion is still somewhat shrouded in controversy and varies from conservative treatment to surgical excision of the causative gland. MATERIALS AND METHODS: This was a case series in quasi-experimental design that evaluated the outcome and complications of the modified micromarsupialization technique as a newly introduced treatment at our center for simple sublingual ranula over a 2-year period. RESULTS: Twenty patients were treated by the modified micromarsupialization technique and followed up for 1 year. There was complete resolution following this technique in 17 patients (85%) with no evidence of recurrence or complications, whereas 2 patients (10%) showed partial resolution and the remaining 1 patient (5%) showed a failure and recurrence. The age of the patient, the size of the ranula, and the retention of sutures throughout the study period did not significantly affect the treatment outcome. CONCLUSIONS: The modified micromarsupialization technique was a simple and effective treatment that should be used as a primary treatment option for simple ranulas and we recommend it to be the first-choice treatment before surgical excision of the sublingual gland, especially in a resource-challenged economy like ours.

Adenocarcinoma not otherwise specified (NOS) arising in the sublingual gland: Rare case report and follow-up.

BACKGROUND: Adenocarcinoma not otherwise specified (AdCaNOS) is a rare malignant salivary gland tumor that occurs with higher frequency in the parotid gland of male patients. In this study, we described a second case of AdCaNOS arising in the sublingual gland, in a female patient with 39 years of age, highlighting the clinical, radiographic, microscopic, treatment features and follow-up. CASE REPORT: A 39-year-old female patient presented a fibroelastic nodule with a yellowish coloration, in the left region of the floor of mouth, measuring about 4.0 cm in its largest diameter. An incisional biopsy was performed and the main microscopic features revealed an infiltrative lesion with glandular differentiation organized in cystic spaces, where neoplastic cells secreting eosinophilic material were observed. The tumor showed immunopositivity for pancytokeratin (AE1/AE3), keratins 7 (CK7) and 14 (CK14), and negativity for p63. The proliferation level measured by Ki-67 marker was considered higher. The patient underwent radical surgical resection, but unfortunately, she developed local recurrence, lymph node mestastasis and died 1.5 year after diagnosis. CONCLUSION: Although rare in the sublingual gland, particularly at this age, AdCaNOS can occur and early diagnosis and early treatment are essential for a better prognosis and survival rates of the patients.

Lymphoepithelial carcinoma in the sublingual gland.

Lymphoepithelial carcinoma is rare in the salivary glands, with an incidence of 0.4%. The most commonly affected site is the parotid gland, followed by the submandibular gland. Lymphoepithelial carcinoma in the sublingual gland has been reported only four times in the existing English-language literature. Such tumours are characterized by the presence of a poorly differentiated carcinoma that is surrounded and infiltrated by lymphocytes, and they are strongly associated with Epstein-Barr virus infection, patient ethnicity, and prominent radiosensitivity. Wide surgical excision combined with adjuvant therapy has been suggested as the first-choice therapeutic regimen. This report describes the case of a 34-year-old Indonesian woman who was evaluated and treated in Taipei Medical University Hospital. She had a tumour that presented as a painless swelling on the floor of the mouth. The diagnosis was confirmed by conducting an incisional biopsy, and a wide surgical excision with bilateral supraomohyoid neck dissection and free flap reconstruction was performed. The patient also underwent adjuvant chemoradiotherapy. No evidence of local recurrence or distant metastasis was detected during the 6 months of follow-up. Subsequently, the patient returned to her home country, and further follow-ups were not conducted.

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

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

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.

Dry Mouth Caused by Facial Nerve Ischemia due to Subarachnoid Hemorrhage: An Experimental Study.

OBJECTIVE: Parasympathetic network damage results in facial nerve damage, sublingual ganglion degeneration, sublingual gland dysfunction, and dry mouth. In this study, subarachnoid hemorrhage (SAH) was considered to be the cause of dry mouth. METHODS: We assessed 23 hybrid rabbits, including 5 control (group 1, Control). One milliliter of serum saline was injected into the cisterna magna of 5 animals (group 2). SAH was induced by injecting 1 mL of autologous blood into the cisterna magna of 13 animals (group 3). The animals were killed after 3 weeks of induction. The animals' sublingual ganglion and sublingual gland were excised for histopathological examination. The number of degenerated cells in the sublingual ganglion, secretory vesicles, and secretory granules in the sublingual gland that contain salivary components were estimated using Sequential Window Acquisition of All Theoretical Mass Spectra data analysis. The values were compared by the Mann-Whitney U-test. RESULTS: The numbers of secretory vesicles in the sublingual gland were 5.3 ± 1.1 × 103 (group 1), 4.23 ± 0.45 × 103 (group 2), and 1.56 ± 0.22 × 103 (group 3); the numbers of secretory vesicles containing saliva in the sublingual gland were 324 ± 12.18 (group 1), 263 ± 36.23 (group 2), and 114 ± 23.14 (group 3); and the numbers of degenerated cells in the sublingual ganglion were 11 ± 3/mm3 (group 1), 98.43 ± 15.54/mm3 (group 2), and 346 ± 12.28/mm3 (group 3) (P < 0.05). CONCLUSIONS: Clinical findings in infection and diseases such as Sjögren syndrome, aseptic meningitis, and SAH are similar. However, until now, SAH has not been demonstrated experimentally to cause dry mouth. Discovering that SAH might cause dry mouth might prevent unnecessary use of antibiotics and decrease morbidity due to the wrong or late diagnosis.

Comprehensive histochemical and stereological study of the parotid and sublingual salivary glands in Caucasian squirrel (Sciurus anomalus).

The present investigation tries to reflect histochemical and stereological properties of the parotid gland (PG) and sublingual gland (SLG) in the Caucasian squirrel. Totally, five male Caucasian squirrels were used. The glands were examind histochemical using PAS, Alcian blue (AB, pH = 2.5) and Aldehyde fucshin (AF) stainings. Total volume of the glands, acini, ducts as well as total length of ducts were estimated using streological procedures. The PG had a unique morphology and were connected to each other with an intermediated isthmus. The SLG was oval-shaped. Serous acini of PG exhibited strong, negative and weak reaction with AF, AB and PAS, respectively. While, mucous acini of SLG showed positive staining for AB and PAS, and negative staining for AF. The ductal system was consisted of intercalated (Id), striated (SD) and excretory ducts (Ed). The Sd in PG was lined with simple cuboidal to the columnar epithelium, while it was lined with simple cuboidal epithelium in SLG. The Ed was lined with stratified cuboidal epithelium in both glands. The striated duct was longest duct in both glands, the excretory and intercalated ducts were the shortest ones in PG and SLG, respectively. The maximum total volume in both gland were blonged to the acini and straited ducts. It concluded that PG and SLG in the Caucasian squirrel have some distinct structural properties in comparison to other rodents and mammalis species.

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.

Comparison of outcomes of treatment for ranula: a proportion meta-analysis.

Treatment for ranula is classified into three categories depending on how the leaking saliva is managed: removal of the leaking site by intraoral or transcervical resection of the sublingual gland; formation of a drainage tract through the wall of the pseudocyst by micromarsupialisation or marsupialisation; or sealing of the site of the leak by inducing fibrosis with a sclerosing agent. Resection of the sublingual gland is probably the option most likely to be curative for both oral and plunging ranula considering their pathophysiology. Although alternative treatments have been introduced to avoid the invasiveness of resection of the gland, their outcomes have been inconsistent. The objective of this study therefore was to help decision-making by providing more integrated rates of cure, consistency of treatment, and morbidity, depending on types of treatment used in previous series of cases. We used proportion meta-analysis of 39 such published series, and the most curative treatment for oral ranula was intraoral resection of the sublingual gland. Micromarsupialisation and its modification showed cure rates comparable with those of resection of the gland, but these were moderately inconsistent. In the treatment of plunging ranula, there was no significant difference in cure rate between the intraoral and transcervical approaches, although they both showed higher cure rates than injection of OK-432. Comparisons of morbidity were available for patients who had developed nerve dysfunction and haematoma after the intraoral and transcervical approaches and there was no significant difference between the two, though the morbidity was higher after the transcervical than that after the intraoral approach. In conclusion, intraoral resection of the sublingual gland is sufficient treatment with a tendency to have fewer complications than that in the transcervical approach.

Protective Effect of Increased Zinc Supply against Oxidative Damage of Sublingual Gland in Chronic Exposure to Cadmium: Experimental Study on Rats.

Cadmium is one of the main chemical pollutants found in the daily environment of developed countries. Cigarettes are a significant source of that metal, which makes it important in terms of oral cavity health. The aim of this study was to determine if increased supply of zinc in chronic exposure to cadmium might protect the sublingual gland structure against oxidative damage. The experiment took 12 months and was conducted on 72 adult male rats. They were randomized into 9 groups. Eight groups received cadmium in drinking water (as CdCl2) at 5 or 50 mg Cd/dm3 and/or zinc (as ZnCl2) at 30 or 60 mg Zn/dm3. The control group received regular water. In the sublingual gland of all animal groups, levels of oxidative parameters were measured. The oxidative stress index was calculated as a TOS/TAS ratio. Cadmium exposure at 5 mg and 50 mg Cd/dm3 induced oxidative stress in the sublingual glands of the rats. Cadmium reduced the TAS and GSH levels and increased LPO, H2O2, TOS, and OSI. In cadmium exposure conditions, increasing the supply of zinc by 79% or 151%, as compared to the standard dietary intake of this microelement, completely prevented the reduction of TAS and GSH levels and accumulation of LPO, H2O2, and TOS in the examined gland at both exposure levels to that metal. The outcome data confirm the protective effect of increased zinc intake on the sublingual gland tissue in chronic cadmium exposure.