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.

[Little frog in the mouth: a visual diagnosis?] / Fröschlein im Mund: eine Blickdiagnose?

Ranula, from the Latin "little frog", is a retention cyst filled with saliva in the oral cavity. Simple ranulas most commonly affect the sublingual gland and typically present as a hemispherical bluish cyst on the floor of the mouth, making it a visual diagnosis. A 7-year-old girl presented with a swelling on the underside of the tongue, an uncommon location for a ranula that made diagnostic assignment difficult. The optimal treatment of a ranula is still controversial in the literature. Many authors favor surgery as the treatment of choice. Our case shows that a watch and wait approach with simple mechanical pressure on the cyst can be sufficient.

Anatomical Relationship Between the Lingual Nerve and Submandibular Duct.

ABSTRACT: The purpose of this study was to investigate the anatomical relationship between the lingual nerve and submandibular duct. This study included 1403 patients with submandibular or sublingual gland diseases who underwent intraoral removal of submandibular gland sialoliths, submandibular glands, or sublingual glands. Of all patients, 33 patients underwent bilateral surgeries. All surgeries were performed a single surgeon, and the anatomical relationship between the lingual nerve and submandibular duct was always identified intraoperatively and recorded in the operation recorded. The anatomical relationship was investigated based on the intraoperative findings. The lingual nerve which crosses above the submandibular duct was detected in 8 of 1436 sides (0.6%). There were 4 in the right sides and 4 in the left sides. The lingual nerve below the submandibular gland was seen in 99.4%. Although the lingual nerve crosses above the submandibular duct with a rarer incidence, surgeons should beware of injuring the lingual nerve during intraoral salivary gland surgery.

Subtotal functional sialoadenectomy vs four-duct ligation for the treatment of drooling in neurologically impaired children: Long-term follow-up.

OBJECTIVES: The best surgical option to treat drooling in neurodisabilities is still under debate. The aim of this study was to describe the technique of subtotal functional sialoadenectomy (SFS) (ie four-duct ligation (4-DL) together with bilateral sublingual gland excision) and its long-term outcomes, in comparison with 4-DL. DESIGN: Retrospective observational cohort study. SETTING: Unit of Pediatric Surgery of Bambino Gesù Children's Hospital (Rome). PARTICIPANTS: Seventy-five patients surgically treated for drooling between 2002 and 2012, with at least five years of follow-up, divided into two groups: 4-DL group (19 patients) underwent four-duct ligation, and SFS group (56 patients) underwent subtotal functional sialoadenectomy. MAIN OUTCOME MEASURES: Primary end points were the evaluation of drooling improvement after surgery (parameters: Drooling Severity and Frequency Scale, DSFS; no of bibs/day; no of shirts/day; no of pneumonia/year; use of antidrooling drugs) and the comparison between two different surgical techniques. RESULTS: Median age at surgery was 10 years (1-35). Long-term outcomes showed significant improvement in DSFS and in no of shirts/day in both groups. Significantly better results were found in the SFS group than in the 4-DL group as far as DSFS (P value .045), no of bibs/day (P value .041), no of shirts/day (P value .032) are concerned. Reoperation rate for recurrence was 42% in the 4-DL group and 0% in the SFS group (P value < .0001). Six patients (8%; 2 in the 4-DL group and 4 in the SFS group) experienced perioperative complications, while 4 patients (5%; 2 in the 4-DL group and 2 in the SFS group) recorded long-term complications, with no difference between groups neither need for surgical treatment. No surgery-related mortality was recorded. CONCLUSIONS: In our experience, subtotal functional sialoadenectomy ensured significantly greater long-term effects than four-duct ligation for drooling treatment in neurologically impaired child, with equal complication rate.

Transoral approach for mandibular and sublingual sialoadenectomy in a cat.

Sialocele is an uncommon condition in cats. The treatment of choice for sublingual sialocele is excision of the ipsilateral mandibular and sublingual salivary gland/duct complex. Lateral and ventral cervical approaches have been described for mandibular-sublingual sialoadenectomy; however, the transoral approach, described here, has never been reported in cats. Ranula in the present case was likely caused by an inadvertent trauma of the sublingual duct during resection of a sublingual lesion performed by the referring veterinarian. The definitive surgery consisted of mass removal and sialoadenectomy through a unique oral approach. The surgery was effective without complications encountered after 6 months of follow-up. Key clinical message: This article reports a novel, transoral approach, for mandibular and sublingual sialoadenectomy in the cat. This approach decreases the surgical time and prevents recurrence of the mucocele.

Approche trans-orale pour la sialo-adénectomie mandibulaire et sublinguale chez un chat. La sialocèle est une maladie rare chez les chats. Le traitement de choix pour la sialocèle sublinguale est l'excision du complexe glandes salivaires/canal salivaire ipsilatéral mandibulaire et sublingual. Des approches cervicales latérales et ventrales ont été décrites pour la sialo-adénectomie mandibulaire-sublinguale; cependant, l'approche trans-orale, décrite ici, n'a jamais été rapportée chez les chats. Dans le cas présent, la ranula a probablement été causée par un traumatisme involontaire du canal sublingual lors de la résection d'une lésion sublinguale réalisée par le vétérinaire référent. La chirurgie définitive consistait en un enlèvement de masse et une sialo-adénectomie par une approche orale unique. La chirurgie a été efficace sans complications rencontrées après 6 mois de suivi.Message clinique clé :Cet article rapporte une nouvelle approche trans-orale pour la sialo-adénectomie mandibulaire et sublinguale chez le chat. Cette approche diminue le temps chirurgical et empêche la récidive de la mucocèle.(Traduit par Dr Serge Messier).

The importance of sublingual gland removal in treatment of ranulas: A large retrospective study.

PURPOSE: Investigate the efficacy of sublingual gland removal for the treatment of simple and plunging ranulas. MATERIALS AND METHODS: After IRB approval, a retrospective review was performed on patients treated for a ranula from February 2013 to May 2018 at Texas Children's Hospital in Houston, TX. Clinical data was collected from medical records and patients were contacted in November 2018 to obtain additional information about recurrences or complications. RESULTS: This study included 52 patients with ranulas (10 plunging, 42 simple; M:F 21:31) with a mean age of 9.68 years. Eighteen patients provided information in the extended follow-up period. Simple ranulas were treated with intraoral excision of the cyst and the sublingual gland (27 cases), marsupialization (7 cases), intraoral excision of the cyst alone (7 cases), and intraoral excision of the cystic component and subsequent marsupialization after recurrence (1 case); intraoral excision of the sublingual gland was not associated with any recurrence. Plunging ranulas were treated with intraoral excision of the cyst and/or sublingual gland (7 cases) or with a transcervical approach (3 cases). One patient was initially treated with sclerotherapy before undergoing intraoral excision of the sublingual gland. Two patients treated with transcervical excision of the cyst experienced recurrence compared to no recurrence with intraoral excision of the sublingual gland. CONCLUSION: Intraoral removal of the sublingual gland is the most effective treatment for both simple and plunging ranulas. Plunging ranulas must be considered in patients presenting with a submandibular and submental cystic mass given intraoral extension may not be apparent.

Management of Intraoral Surgical Defects Using Sublingual Gland Flap.

The aim of the study was to evaluate the effectiveness of sublingual gland flap in the reconstruction of surgical defect following sequestrectomy in medication induced osteonecrosis of jaws (MRONJ), osteomyelitis (OML), and osteoradionecrosis (ORN) of mandible. A total of 6 patients with MRONJ (n = 4) osteomyelitis (n = 1) and ORN (n = 1) underwent sequestrectomy and reconstruction with sublingual gland flap. There were 03 males and 03 females. The age range was 45-71-70 years. All defects were present in the posterior mandible. All patients' undergone sequestrectomy, debridement, and reconstruction with sublingual gland flap under local anesthesia. Postoperatively patients were followed up at 1, 3, and 6 months. Patients were monitored for complete epithelization of defect, infection, pain, and recurrence of the lesion. Complete epithelization with closure of the defect was achieved in all cases. None of the patients had residual pain or inflammation at the surgical site. None of our patients experienced any donor site morbidities. There was no case of postoperative infection. Reconstruction of intraoral defects using sublingual gland flap is an effective treatment modality to reconstruct small to medium sized defect of oral cavity.

Trans-Oral Robotic Surgery of Submandibular Gland Removal With Preservation of Sublingual Gland and Wharton's Duct.

In surgical removal of a submandibular gland, trans-cervical approach has esthetic problem and existing trans-oral approaches are extensively invasive. The authors have used trans-oral robotic surgery to remove the submandibular gland with preservation of the sublingual gland and the Wharton's duct, and hereby report the case and discuss our preliminary experiences.

Susceptibility of partially desalivated rats to erosive tooth wear by calcium-supplemented beverages.

OBJECTIVES: To investigate the susceptibility of partially desalivated rats to erosive tooth wear (ETW); the anti-erosive effect of a calcium-supplemented beverage; and the quantification of ETW by microcomputed tomography (micro-CT). METHODS: The study population consisted of thirty-eight rats, divided into partially desalivated (n = 19) and normal salivary flow (n = 19). They were randomly allocated into three subgroups (n = 6-7): A-diet soda, B-diet soda + calcium, C-water (control). Solutions were provided ad libitum for 28 days, and the rats were euthanized afterwards. Each left hemi-mandible was scanned using micro-CT for enamel volume (three molars) calculation. Visual analysis of photographs of the lingual surface of first molars was performed independently by three blinded examiners. Data were statistically analysed (α = .05). RESULTS: Micro-CT revealed no significant differences between partially desalivated or normal groups. Rats consuming A had more enamel loss than those consuming B or C, which did not differ from each other. For visual analysis, desalivation did not affect ETW. Rats consuming C showed the lowest ETW, followed by B and then A, for both partially desalivated and normal rats. Spearman correlation between the two ETW quantification methods was -.65. CONCLUSIONS: Partial desalivation did not increase ETW. Ca-containing beverage prevented ETW. Micro-CT quantified ETW, although it was not as sensitive as visual analysis.

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.

Ranula: Current Concept of Pathophysiologic Basis and Surgical Management Options.

BACKGROUND: There is no consensus opinion on a definitive surgical management option for ranulas to curtail recurrence, largely from the existing gap in knowledge on the pathophysiologic basis. AIM: To highlight the current scientific basis of ranula development that informed the preferred surgical approach. DESIGN: Retrospective cohort study. SETTING: Public Tertiary Academic Health Institution. METHOD: A 7-year 7-month study of ranulas surgically managed at our tertiary health institution was undertaken-June 1, 2008-December 31, 2015-from case files retrieved utilising the ICD-10 version 10 standard codes. RESULTS: Twelve cases, representing 0.4 and 1.2% of all institutional and ENT operations, respectively, were managed for ranulas with a M:F = 1:1. The ages ranged from 5/12 to 39 years, mean = 18.5 years, and the disease was prevalent in the third decade of life. Main presentation in the under-fives was related to airway and feeding compromise, while in adults, cosmetic facial appearance. Ranulas in adults were plunging (n = 8, 58.3%), left-sided save one with M:F = 2:1. All were unilateral with R:L = 1:2. Treatment included aspiration (n = 2, 16.7%) with 100% recurrence, intra-/extraoral excision of ranula only (n = 4, 33.3%) with recurrence rate of 50% (n = 2, 16.7%), while marsupialisation in children (n = 1, 8.3%) had no recurrence. Similarly, transcervical approach (n = 5, 41.7%) with excision of both the ranula/sublingual salivary gland recorded zero recurrence. Recurrence was the main complication (n = 4, 33.3%). CONCLUSION: With the current knowledge on the pathophysiologic basis, extirpation of both the sublingual salivary gland and the ranula by a specialist surgeon is key for a successful outcome.

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.

Plunging Ranula Occurring Without its Oral Counterpart: A Case Report.

A ranula is a lesion that arises from mucin spillage from the sublingual or submandibular gland ducts, beneath the mucosa of the oral floor. A plunging or cervical ranula dissects the mylohyoid muscle and appears as a submandibular swelling. Rarely, a plunging ranula is encountered without its oral counterpart. Here, we report a rare case of a plunging ranula that occurred without its oral counterpart in a 11-year-old male patient. We have also discussed the pathogenesis and treatment options for the ranula.

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.

Complications of sialendoscopy: personal experience, literature analysis, and suggestions.

PURPOSE: This article describes our experience combined with analysis of the emerging literature, presenting suggestions of how to avoid the complications that may arise during or after endoscopic or endoscopy-assisted surgery of the salivary glands. MATERIALS AND METHODS: In a retrospective study, the surgical data of 498 consecutive patients who underwent surgery for calculus removal from the parotid, submandibular, and sublingual glands from 2010 to 2012 were collected and analyzed. The analysis was concentrated on specific complications of endoscopic or endoscopy-assisted operations. RESULTS: The patients were operated on by various sialendoscopy-involved techniques that included intraductal endoscopy and an endoscopy-assisted extraductal approach. The total percentage of complications associated with endoscopy was 3.23% (n = 17). In 1 case (0.2%) (submandibular gland surgery), severe bleeding occurred that required immediate gland resection. CONCLUSIONS: At present, sialendoscopic and endoscopy-assisted techniques produce a minimal number of postsurgical endoscopy-related complications, but they are not complication-free operations. Strictures, ranulas, and lingual nerve paresthesias are the most frequent of these complications. Further reduction of the number of complications is possible with careful preprocedural imaging analysis and correct endoscopic techniques.

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.

Management of ranula: 9 years' clinical experience in pediatric and adult patients.

PURPOSE: The aim of the present study was to evaluate the long-term results in the authors' department in the management of patients with oral and plunging ranulas. A specific diagnostic and therapeutic approach is suggested based on these data and data from the literature. MATERIALS AND METHODS: A retrospective analysis of 65 patients with a final diagnosis of ranula was carried out. The medical records were evaluated for the principal demographic, clinical, diagnostic, and therapeutic data. Statistical analysis was used to compare outcomes of the different treatments. RESULTS: Treatments that included complete sublingual gland excision were associated with the lowest recurrence rate (3.6%), followed by partial sublingual gland excision (9.1%), marsupialization (13%), and ranula excision (36.7%). Nine patients (13.8%) had recurrences. A statistically significant difference was observed (P = .01) in the recurrence rate between complete sublingual gland excision and ranula excision alone. Complications developed in 7 patients (10.8%). Ultrasonography performed in all patients showed an intimate relation among the cyst, mylohyoid muscle, and sublingual gland. Dehiscence of the mylohyoid muscle was noted in some cases. CONCLUSIONS: The surgical experience in the authors' department confirms the different treatments that are offered for ranula. Sublingual gland excision should be the preferred treatment, but marsupialization may be useful as an alternative minimally invasive procedure, with a success rate higher than 85% and no risk of increased complications if revision surgery is needed. Ultrasound is the recommended diagnostic tool, in addition to the clinical examination, for the differential diagnosis and during follow-up.

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.

[Transoral approach for sublingual-plunging ranula]. / Transoraler Zugang bei sublingualer Tauchranula.

The case of a 7-year-old boy suffering from progressive submental/submandibular swelling is reported. Following clinical and imaging diagnostics (MRI), the suspected diagnosis of a sublingual-plunging ranula was made. Surgery was performed with transoral excision of the sublingual gland in combination with excision of the ranula. Additional submandibular gland excision should be avoided.