Ranula Formation After Lingual Frenectomy in a Newborn.

Ankyloglossia is a developmental anomaly characterized by a shortened lingual frenulum, which can range from cases with no clinical relevance to severe cases, where frenectomy may be indicated. In newborns, this indication has increased exponentially. The purpose of this report is to discuss obstruction of the sublingual caruncle, with a recurrent ranula, as a post-surgical complication of laser lingual frenectomy performed in a newborn. The treatment provided is also discussed (marsupialization and removal of the affected sublingual glands). Lingual frenectomy should be performed with great care, especially in newborns, because the sublingual caruncles can be affected and complications can occur.

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.

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.

Treatment of oral ranula in HIV-positive patient.

HIV-associated salivary gland disease refers to the pathology in head and neck lesions such as ranula, salivary gland swelling, xerostomia, and benign lymphoepithelial cysts in the parotid gland. Here, we present a unique case of the ranula patient with HIV infection treated with OK-423 sclerotherapy. Case report: The patient was a 42-year-old Japanese male with a few months history of oral floor swelling. Computed tomography (CT) showed a low-density area limited within the right floor of the mouth. Magnetic resonance imaging (MRI) revealed a distinct T2-high intensity area localized on the same location. The puncture fluid was bloody mucus, and the cytology was no malignancy. We diagnosed a simple ranula. He was, however, found to be HIV-antibody positive at the examination before treatment by chance. He was referred to the department of infectious diseases and definitively diagnosed HIV infection by western blot. We chose OK-432 sclerotherapy because of its minimally invasive and the risk of HIV infecting medical staff. Two times OK-432 injection made the lesion disappear. Conclusion: The case indicated that OK-432 sclerotherapy could be effective for ranula related to HIV.

Major complications after tongue-tie release: A case report and systematic review.

INTRODUCTION: The diagnosis of ankyloglossia, or tongue-tie, and the number of frenotomies performed has increased over 10-fold from 1997 to 2012 in the United States. The sharpest increase has been in neonates. For parents considering frenotomy for their breastfeeding newborn, there is controversy surrounding the evaluation of tongue-tie and the benefit of a frenotomy. Complications from tongue-tie procedures are thought to be low, though it is not well reported nor studied. OBJECTIVES: The aim of this study is to describe a case of a sublingual mucocele after laser frenotomy in a neonate with tongue-tie and to investigate major complications reported after tongue-tie release in pediatric patients through a systematic review of the literature. CASE REPORT: We present a 6-week-old female who underwent a laser frenotomy procedure performed by a dentist who presented with a new cyst under her tongue. MATERIAL AND METHODS: A systematic literature search of articles published from 1965 to April 2020 was conducted in Ovid MEDLINE(R), Ovid EMBASE, and Scopus. Citations were uploaded into a systematic review software program (DistillerSR, Ottawa, ON, Canada), followed by full text screening. RESULTS: 47 major complications were reported in 34 patients, including our patient. Most of the cases were located in the United States and Europe. The most frequent indications for the procedure were breastfeeding problems (n = 18) and speech impediment (n = 4). The procedure was performed by dentists (n = 6), lactation consultants (n = 5), and otolaryngologists (n = 4). The bulk of the major complications after frenotomy included poor feeding (n = 7), hypovolemic shock (n = 4), apnea (n = 4), acute airway obstruction (n = 4), and Ludwig angina (n = 2). CONCLUSIONS: Reporting of complications after frenotomy is lacking. Risks to neonates may be different than risks to older children and adults. Practitioners across different specialties should be monitoring and studying this more rigorously to better guide patients and families on the risks and benefits of this procedure.

The anatomical basis and rational for the transoral approach during the surgical excision of the sublingual salivary gland for the management of plunging ranula.

PURPOSE: The aim of this study was to illustrate and confirm that the complete intraoral excision of the sublingual gland alone, is anatomically the most rational approach, for the management of plunging ranula. MATERIAL AND METHODS: We retrospectively reviewed clinical charts of diagnosed HIV-infected patients presenting with plunging ranula. The intraoral approach was used for the complete excision of the sublingual gland with evacuation of the pseudocystic content only. Neither extra oral approach, nor ranula dissection / drainage was performed. The surgical procedure was performed using local anesthesia. Pre- and postoperative MRI-scan investigations were recorded. Histological reports were documented to confirm the diagnosis of oral mucocele. Patients were clinically monitored. RESULTS: We identified 90 adults presenting with oral mucocele, type ranula. Seventy (77%) of them were diagnosed with HIV infection. Plunging ranula was recorded in 35 (50%) patients from the latter group. The study enrolled 11 operated patients whose files contained useable data, including an acceptable follow-up period. The postoperative follow-up period ranged from three to 15 months. The clinical and postoperative MRI-scans of operated patients demonstrated satisfactory results. There were neither postoperative complications nor recurrence of ranula reported. CONCLUSION: The location of the sublingual gland in the floor of the mouth coupled with the physio-pathogenesis of the plunging ranula, makes the transoral complete excision of the offending gland, with the intraoral evacuation of the pseudocyst, anatomically the most rational approach for plunging ranula management. There is no need for cervical approach, ranula dissection and/or postoperative placement of drainage.

Sjögren's syndrome and ranula development.

OBJECTIVES: We hypothesized that the extravasation of saliva from damaged ducts with lymphocytic infiltration in patients with Sjögren's syndrome causes ranulas. There are too many uncertainties to support this hypothesis. The aim of this study was to investigate whether there is an association between Sjögren's syndrome and ranulas. MATERIALS AND METHODS: We observed three cases of patients with ranulas who were also diagnosed with Sjögren's syndrome at the same facility. These cases led to the question of whether there are other such cases, and thus, an exhaustive literature search was conducted. RESULTS: Three cases in two case reports of mucocele of the floor of the mouth associated with adult Sjögren's syndrome were noted. Including our cases, until now, there have been six cases of ranula with adult Sjögren's syndrome. CONCLUSIONS: It could be useful to investigate whether patients presenting with a ranula are also affected by Sjögren's syndrome and, conversely, investigate patients with Sjögren's syndrome longitudinally to see whether they develop ranulas.

Pediatric primary Sjögren syndrome presenting with bilateral ranulas: A case report and systematic review of the literature.

CONTEXT: Primary Sjögren syndrome is uncommon in children, and the standard clinical criteria used in diagnosis of adult Sjögren syndrome will miss many children with the disease. Floor of mouth ranulas have not been described in Sjögren syndrome. OBJECTIVE: This study aims to describe a novel presentation of juvenile primary Sjögren syndrome, and to present a comprehensive systematic review of the literature regarding the presentation and diagnosis of Sjögren syndrome in children. DATA SOURCES: Ovid MEDLINE. STUDY SELECTION: A MEDLINE literature search was performed using the following search terms: primary, Sjögren, disease, and children. Results were limited to human subjects and articles written in English between 1981 and 2014. Applicable articles were reviewed and qualitatively summarized. DATA EXTRACTION: Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines (PRIMA). RESULTS: Initial MEDLINE search yielded 146 articles, 80 of which were excluded as not clinically pertaining to Sjögren syndrome. An additional 25 were excluded due to lack of pediatric-specific data. Systematic review of the literature revealed no reports of ranula in association with Sjögren syndrome. 6 papers were manually included from review of reference lists of included articles. Our review indicated that recurrent parotitis is the most commonly reported presenting symptom in children, followed by ocular and oral symptoms, musculoskeletal, and renal symptoms. Compared to adults, children are less likely to present with dry eyes and mouth. LIMITATIONS: All studies were retrospective chart reviews, case series or case reports. CONCLUSION: This is the first report of a child presenting with floor of mouth ranulas in association with Sjögren syndrome. While recurrent parotitis is the most common presentation in children, other salivary gland and extra-salivary manifestations may be seen, and the clinician must maintain a high index of suspicion for underlying Sjögren syndrome.

Are the patients with anatomic variation of the sublingual/Wharton's duct system predisposed to ranula formation?

OBJECTIVE: To evaluate ranula development according to anatomic variation of the ductal system of sublingual gland (SLG), especially the presence of Bartholin's duct. METHODS: The anatomic variation of SLG duct was prospectively investigated and compared between 55 consecutive patients with ranulas treated by SLG excision (group 1) and another 15 consecutive patients undergoing similar surgeries for other conditions (group 2). The ductal structures of SLGs and submandibular glands (SMG) were also compared between the pediatric patients and adult patients with ranulas. RESULTS: In 32 of 55 patients with ranulas (58.2%) and 1 of 15 patients without ranulas (6.7%), the SLG showed an anatomic variation of the main duct called Bartholin's duct structure (P<0.01). Seventeen of 22 (77.3%) pediatric patients with ranulas had Bartholin's ducts and 15 of 33 (45.5%) adult patients with ranulas had Bartholin's ducts (0.01

Modified L-shaped surgical approach to excision of the sublingual gland.

We present a series of 1131 patients who were treated between 1981 and 2013 using the modified L-shaped angular surgical approach to excision of the sublingual gland, and describe the risks and benefits compared with the conventional approach. A total of 365 patients were treated with the conventional approach, while the other 766 have been treated since 1991 using the modified approach. There were no serious complications or recurrences during or after the operation in the modified series. There were significant differences between the two approaches in terms of injuries to the submandibular duct (p=0.03), incidence of ranula on the opposite side (p=0.02), amount of intraoperative bleeding (p=0.000), and duration of operation (p=0.000). The modified L-shaped incision is both viable and safe, and simplifies excision of the sublingual gland while protecting the submandibular duct.

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.

Oral mucocele/ranula: Another human immunodeficiency virus-related salivary gland disease?

OBJECTIVES/HYPOTHESIS: To describe clinical characteristics of oral mucoceles/ranulas, with a focus on human immunodeficiency virus (HIV)-related salivary gland diseases. STUDY DESIGN: A descriptive and clinical study, with review of patient data. MATERIAL AND METHODS: We reviewed 113 referred cases of oral mucocele. The following anatomical sites were identified: lip, tongue, and floor of the mouth (simple ranulas), as well as plunging ranulas. The age and gender data of the patients with oral mucoceles were recorded. The HIV status of the patients and other information were reviewed. RESULTS: There were 30 (26.5%) males and 83 (73.5%) females. Most patients were below 30 years of age, with the peak frequency in the first and second decade. Ranula (simple and plunging) represented 84.1% of the mucocele locations. Mucocele on the lips represented 10.6%. Seventy-two (63.7%) patients were HIV positive; and 97.2% of them had ranulas. Thirty-eight (33.6%) patients presented with plunging ranulas; and 92.1% of them were HIV positive, compared with two patients presenting with plunging ranulas in the HIV-negative group. These results strongly suggest that an HIV-positive patient is statistically (P < 0.001) more at risk of presenting with not only a simple, but also a plunging ranula type. CONCLUSION: This study presents a different clinical picture of oral mucoceles/ranulas, as observed in HIV-positive patients. Additionally, it suggests a possible clinical link between the two pathologies. The authors strongly support the suggestion that oral mucocele/ranula is an HIV-related salivary gland disease. LEVEL OF EVIDENCE: 4.

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.

Salivary gland diseases: infections, sialolithiasis and mucoceles.

The three most frequently diagnosed salivary gland diseases are salivary gland infections, sialolithiasis and mucoceles. Salivary gland infections are usually of bacterial or viral etiology and can be divided into acute and chronic types. Occasionally they can result from obstruction of the salivary duct, an autoimmmune disease or cancer therapy. Infections can occur in all types of salivary glands and are observed at all ages. Sialolithiasis is characterized by the development of calcified structures in the salivary glands, especially in the submandibular gland. Sialoliths are generally attributed to retention of saliva and are usually accompanied by swelling and pain when a salivary stimulus is applied. Mucoceles can be differentiated into mucus extravasation phenomenon or mucus escape reaction, mucus retention cysts and ranulas. They result from extravasation of saliva into the surrounding soft tissues or from retention of saliva within the duct.

Ductal variation of the sublingual gland: a predisposing factor for ranula formation.

BACKGROUND: The purpose of this study was to evaluate ranula development according to anatomic variation of the sublingual gland (SLG). METHODS: We conducted a prospective, consecutive case series considering other clinical conditions. Twenty-five cases treated by SLG excision were enrolled in this study. The ductal structures of the SLGs of another 11 patients undergoing similar surgeries for other conditions were compared. RESULTS: In 19 of a total of 25 ranulas (76.0%) and 16 of 18 oral nonplunging ranulas (88.9%), the SLG showed an anatomic variation of the main duct called Bartholin's duct structure. Meanwhile, only 3 of 7 plunging ranulas (42.9%) had Bartholin's ducts. Bartholin's duct structure of the SLG was not found in the 11 control cases. CONCLUSION: Anatomic variation of the ductal system of the SLG might be a possible cause of ranulas. Surgical resection of the SLG is a better treatment choice for ranulas than other, more conservative treatments.

The use of sialendoscopy for the treatment of multiple salivary gland stones.

OBJECTIVE: The purpose of this study is to document our experience with sialendoscopy for the treatment of multiple calculi of the salivary ducts, as well as to discuss the technique and indications for the procedure. PATIENTS AND METHODS: In this retrospective non-interventional study, 530 consecutive cases of sialolithiasis were investigated, and the cases with multiple calculi were selected for further analysis. These selected cases were analyzed from clinical and surgical aspects. RESULTS: Multiple calculi were detected in 37 of 530 consecutive investigated sialolithiasis cases. The gender distribution was 15 men and 22 women, with a mean age of 40.4 years. Among these patients, the submandibular gland was affected in 33 cases (right in 13 and left in 20) and the parotid gland was affected in 4 cases (right in 3 and left in 1). As for complications, 1 patient had a postoperative infection and 2 other patients had postoperative ranulas that were considered minor complications. Among all 37 cases, we had 3 cases in which preoperative imaging (sonography in 1 and radiography in 2) showed a single sialolith in the duct, but failed to show further sialoliths that were discovered later by sialendoscopy. CONCLUSIONS: The use of sialendoscopy allows a better diagnosis and minimally invasive treatment for multiple calculi. Interventional sialendoscopy allows the extraction of sialoliths in most patients, preventing open gland excision or surgical removal of the gland even in cases with multiple calculi. Sialendoscopy allows exploration of the ductal system in detail. This technique helps to discover additional stones in the ductal system that could not have been identified with the conventional imaging modalities available.

The potential impact of highly active antiretroviral therapy on the treatment and epidemiology of ranula in human immunodeficiency virus-positive patients.

OBJECTIVE: The study's aim was to assess the potential therapeutic effect of highly active antiretroviral therapy (HAART) on ranulas in human immunodeficiency virus (HIV)-positive patients. STUDY DESIGN: The study includes a retrospective observation of 3 patients who were initially part of a prospective study on the comparative effect of HAART on ranulas in 14 HIV-positive patients. These patients were clinically monitored while pursuing the medical treatment with HAART. Neither a fine needle aspiration nor a surgical procedure was performed. Clinical photographs were used for monitoring of any reduction in the ranula size. The effect of HAART on ranula was assessed at 3, 6 and 12 month. RESULTS: A complete resolution of the ranula lesion was noticed in the 3 HIV-positive selected patients. These results were observed between 6 and 12 months period. CONCLUSION: This study suggests that HAART might present a potential therapeutic effect on ranula in HIV-positive patients.

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.