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.

Structural analysis of the mylohyoid muscle as a septum dividing the floor of the oral cavity for the purposes of dental implant surgery: variety of muscle attachment positions and ranges of distribution.

OBJECTIVES: The objective was to investigate the details of the attachments of the mylohyoid muscle to the mandible anterior to the hyoid and mylohyoid lines to understand the positional relationship between the sublingual space and the mylohyoid, knowledge that is essential for dental implant surgery in the incisal region, as well as the routes of communication between the sublingual space and other spaces. METHODS: While evaluating the presence or absence of an anterior mylohyoid muscle fiber attachment to the mandible, sublingual gland herniation, spaces between muscle fascicles were also recorded as sites of penetration. The mean muscle thickness in each of these areas was also calculated. RESULTS: In all specimens, the mylohyoid originated not only from the mylohyoid line but also from the lingual surface of the center of the mandibular body (the mandibular symphysis) below the mental spines. The mylohyoid muscle fascicles were thickest in the posterior region, and further anterior to this, they tended to become thinner. Sublingual gland herniations passing through the mylohyoid were noted in the anterior and central regions, but not in the posterior region. Penetration between the muscle fascicles was most common in the central region, and no such penetration was evident in the posterior region. CONCLUSIONS: These results suggest that the mylohyoid functions only incompletely as a septum, and that routes of communication from the sublingual space to the submandibular space may be present in both the anterior and central muscle fascicles of the mylohyoid. Therefore, bleeding complications during dental implant placement in the anterior mandible can be serious issues. There is a potential for sublingual hematoma that could compromise the airway by pressing the tongue against the soft palate into the pharynx.

New Conservative Approach for the Management of Recurrent Sublingual Ranula-A Case Report.

Ranula is a rare and benign extravasation mucocele that is clinically characterized by asymptomatic sublingual or submandibular masses. Surgical excision is considered the most effective treatment approach, but it has been associated with high invasiveness and several complications (hemorrhage, damage to Wharton's duct, and lingual nerve injury). Over the past decade, more conservative therapies have been rapidly disseminated into clinical practice to seek a more effective and less traumatic approach for young patients. In this report, an 8-year-old female with an asymptomatic, recurrent sublingual ranula was treated using a conservative approach with marsupialization and an intracystic injection of a plaque remover (Hybenx® gel). After incision of the cystic dome, Hybenx® gel was applied into the cystic lumen for 20 seconds and then aspirated; next, the area was rinsed thoroughly with sterile saline solution before suturing. Ultrasound re-evaluation at 10 months and intraoral clinical examination at 24 months confirmed the absence of relapse. Our results support the hypothesis that marsupialization combined with intracystic injection of Hybenx® gel could be an encouraging conservative treatment alternative for recurrent sublingual ranula in children. Further randomized controlled trials are needed to test this hypothesis.

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.

[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.

Ránula cervical gigante: a propósito de un caso / Huge plunging ranula: about a case

La ránula es una lesión pseudoquística causada por la retención de contenido salival de la glándula sublingual en el tejido conjuntivo subyacente, siendo la patología más frecuente de esta glándula. Su localización es a nivel del suelo de la boca, pudiendo en ocasiones extenderse a través del músculo milohioideo hacia la región submaxilar o cervical (ránula plunging o cervical), presentándose clínicamente como una tumoración laterocervical de crecimiento lento. El apoyo de imágenes mediante ecografía, tomografía computarizada, o RM (resonancia magnética) es fundamental para un correcto diagnóstico. Respecto al tratamiento, el procedimiento más aceptado y con menos tasas de recurrencia es la escisión de la ránula cervical por abordaje laterocervical, asociada a la extirpación de la glándula sublingual implicada vía transoral. Aquí presentamos el caso clínico de un varón de 25 años quien presenta una masa laterocervical derecha de seis meses de evolución de crecimiento progresivo e indoloro. La ecografía y RM confirman una ránula cervical gigante de 62x45x101 mm, que se localiza en espacio submandibular derecho, alcanzando el espacio parafaríngeo en su vertiente más craneal. Debido a las características de la lesión y su anatomía se decide tratamiento quirúrgico.

The ranula is a pseudocystic lesion caused by the retention of salivary content of the sublingual gland in the underlying connective tissue, being the most frequent pathology of this gland. Its location is at the level of the floor of the mouth and can sometimes extend through the mylohyoid muscle towards the submaxillary or cervical region (plunging or cervical ranula), clinically presenting as a slow-growing laterocervical tumor. The support of images by ultrasound, computed tTomography or MRI (magnetic resonance imaging) is essential for a correct diagnosis. Regarding treatment, the most accepted procedure, and with the lowest recurrence rates is excision of the cervical ranula by the laterocervical approach, associated with the transoral removal of the involved sublingual gland. Here, we present the clinical case of a 25-year-old man who presented a six-month-old right laterocervical mass of progressive and painless growth. Ultrasound and MRI confirmed a giant cervical ranula measuring approximately 62x45x101 mm, located in the right submandibular space, reaching the parapharyngeal space in its most cranial aspect. Due to the characteristics of the injury and its anatomy, surgical treatment was decided.

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.

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.

Transoral Complete vs Partial Excision of the Sublingual Gland for Plunging Ranula.

OBJECTIVES: Plunging ranula is a pseudocyst of saliva extravasated from the sublingual gland (SLG) to the submandibular space. This is treated by transoral excision of the SLG or transcervical cyst excision that might differently affect surgical morbidity and recurrence. This study compared the clinical outcomes of complete vs partial excision of the SLG for plunging ranula. STUDY DESIGN: A nonrandomized comparative study. SETTING: Academic medical center. METHODS: This study included 42 patients with plunging ranula who underwent complete or partial excision of the SLG with the evacuation of cystic content. Two surgical methods of complete or partial SLG resection were alternatively allocated to consecutive patients without randomization. The primary outcome was a postoperative recurrence. Secondary outcomes were operation time and complications. RESULTS: Complete and partial excision of the SLG was performed in 22 and 20 patients, respectively, without injury to the Wharton's duct or the lingual nerve. Postoperative complications in 42 patients were minor with temporary events: hematoma, 1 (5%); tongue numbness, 2 (5%); dysgeusia, 4 (9%); and dysphagia, 2 (5%), which did not differ between patients with complete and partial excision of the SLG (P > .1). However, recurrence occurred in only 5 of 20 patients with partial SLG excision but none of 22 patients with complete SLG excision for a median follow-up of 36 months. CONCLUSIONS: Complete SLG excision is preferred over partial SLG excision to treat plunging ranula for reducing postsurgical risks of complications and recurrence.

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).

Excision of Sublingual Gland.

There are 2 types of ranulas: oral ranulas and plunging (cervical) ranulas. The management of the cervical ranula involves surgical excision of the oral portion of the ranula along with the associated sublingual salivary gland. The sublingual gland is easily removed from an intraoral approach. Significant anatomic structures associated with the removal of the sublingual gland are the submandibular duct, lingual nerve, and sublingual artery. Knowledge of the anatomy makes the surgery easier and without complications.

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.

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.

Primary sialorrhea and its surgical treatment with denervation of the submandibular glands in combination with sublingual gland excision.

OBJECTIVES: The purpose of this study was to evaluate the effectiveness of ablating postganglionic parasympathetic fibers of the submandibular ganglion in combination with sublingual gland excision in the treatment of primary sialorrhea. MATERIALS AND METHODS: Healthy volunteers were initially chosen to determine a basic saliva flow rate value by saliva collection. Next, unstimulated and stimulated saliva flow rates, and a visual analog scale index for sialorrhea were recorded in all the patients pre- and postoperatively. In addition, the generalized anxiety disorder scale was used to assess preoperative anxiety symptoms. The follow-up durations ranged from 24 to 36 months after surgery. RESULTS: A total of 10 patients were included in this study. Preoperatively, the mean unstimulated saliva flow rate for the 10 patients was 11.26 ± 4.19 ml/10 min, the stimulated saliva flow rate was 16.76 ± 3.49 ml/6 min, and the visual analog scale index was 66.29 ± 14.86. Postoperatively, the mean unstimulated and stimulated saliva flow rates were significantly reduced to 5.99 ± 1.33 ml/10 min (p = 0.001) and 13.28 ± 1.86 ml/6 min (p = 0.013), respectively, and the visual analog scale index was 25.41 ± 5.6 (p < 0.001). No complications were found after operation. CONCLUSION: This study demonstrates that ablation of postganglionic parasympathetic fibers of the submandibular ganglion in combination with sublingual gland excision by an intraoral approach is a simple, safe, and efficient approach for treating primary sialorrhea.

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.

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.

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.

Long-term impact of saliva control surgery in children with disability.

Sialorrhea is a common problem in children with disability, often negatively affecting socialization, self-esteem, and burden of care. Saliva control surgery is an available option to manage this problem, particularly when other conservative methods have failed. As little is known about the long-term impact of surgery, we followed up 62 patients who had combined bilateral submandibular duct translocation and bilateral sublingual gland excision at our pediatric hospital between 1994 and 2014. Eligible individuals were identified through a search of ICD procedure codes. When families of patients were contacted successfully, they were invited to complete a 14-item questionnaire designed specifically for this study. The results indicated that long-term outcomes of surgery were very good; 13/62 (21%) individuals no longer had a drooling problem and another 30 (48%) experienced only mild to moderate drooling. Although 84% families reported some or major improvement in drooling, 9 families reported that they would not go through the experience again because of a difficult recovery period, lack of effectiveness of the intervention, changes in saliva consistency that caused coughing and gagging, and dental decay. None of the collected variables were predictive of good or poor outcome. The study indicated that surgical intervention is effective in the long term in the majority of cases and can be recommended to other families who attend our saliva control clinic.