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Table of Contents
ORIGINAL ARTICLE
Year : 2020  |  Volume : 22  |  Issue : 2  |  Page : 50-56

Oral ranulas: A 15-year retrospective clinico-surgical evaluation in a major referral hospital in the Southern Province of the Kingdom of Saudi Arabia


1 Department of Maxillofacial Surgery, King Khalid Hospital, Najran, Kingdom of Saudi Arabia
2 Department of Oral and Maxillofacial Surgery, Specialty Regional Dental Center, Najran, Kingdom of Saudi Arabia

Date of Submission21-Feb-2020
Date of Decision12-Mar-2020
Date of Acceptance17-Apr-2020
Date of Web Publication30-Dec-2020

Correspondence Address:
Dr. Ramat Oyebunmi Braimah
Department of Oral and Maxillofacial Surgery, Specialty Regional Dental Center, Najran
Kingdom of Saudi Arabia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/SJOH.SJOH_4_20

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  Abstract 


Background: A ranula is a mucous extravasation phenomenon involving the sublingual gland. The main objective of the current study was to present a clinic-surgical evaluation of ranula in our center. Materials and Methods: This is a 15-year retrospective analysis of ranulas in the main referral hospital in the Najran Region of the Kingdom of Saudi Arabia. Data collected includes demographics, type of ranula, modality of occurrence, laterality, surgical approach, and type of treatment. Data were stored and analyzed using IBM SPSS Statistics for IOS Version 25(IBM Corp., Armonk, NY, USA). Results were presented as simple frequencies and descriptive statistics. Results: Fifty-five cases of oral ranula were recorded out of a total of 75 ranulas during the study with a prevalence rate of 0.14 cases/1000 persons. There were 27 males and 28 females with a male-to-female ratio of 1:1.03. Their ages ranged from 2 years to 45 years with mean ± standard deviation (22.2 ± 10.7). The age group from 11 to 20 years had the highest frequency with 19 cases, representing 34.5% followed by the age group of 21–30 years with 16 cases, representing 29.1%. Of the total of 55 cases of oral ranula, 53 (96.4%) had the lesions excised together with the sublingual gland through intraoral approach, while only 2 (3.6%) cases were marsupialized. Conclusions: We have reported that oral ranulas represent about 73.3% of the ranulas. Surgical removal of the lesion and the sublingual gland is essential for the prevention of recurrence. Loose intraoral suturing in the floor of the mouth is advised to prevent saliva refilling.

Keywords: Excision, marsupialization, mucocele, ranula, sublingual gland


How to cite this article:
Daniels JS, AlBakry I, Braimah RO, Samara MI. Oral ranulas: A 15-year retrospective clinico-surgical evaluation in a major referral hospital in the Southern Province of the Kingdom of Saudi Arabia. Saudi J Otorhinolaryngol Head Neck Surg 2020;22:50-6

How to cite this URL:
Daniels JS, AlBakry I, Braimah RO, Samara MI. Oral ranulas: A 15-year retrospective clinico-surgical evaluation in a major referral hospital in the Southern Province of the Kingdom of Saudi Arabia. Saudi J Otorhinolaryngol Head Neck Surg [serial online] 2020 [cited 2021 Jan 19];22:50-6. Available from: https://www.sjohns.org/text.asp?2020/22/2/50/305467




  Introduction Top


A ranula is a mucous extravasation phenomenon involving the sublingual gland. It is also regarded as a mucocele seen on the floor of the mouth, although a ranula is often larger than a mucocele. The name came from the Latin word rana which means “frog” (ranula = “little frog”) because the swelling resembles the vocal or air sacs of the frog.[1],[2]

Ranula usually occurs as a result of mucin collection in the surrounding connective tissue from a ruptured salivary gland due to local trauma. The local trauma injures the fragile ducts (ducts of Rivinus) that drain saliva from the sublingual gland into the floor of the mouth.[3] When it occurs, it is usually seen as a bluish, transparent dome-shaped fluctuant swelling in the floor of the mouth. The deep blue color results from tissue cyanosis and vascular congestion associated with the stretched overlying tissue and translucent character of the accumulated fluid beneath.[1] However, if the extravasation occurs deep into the tissues, the bluish appearance is usually nonexistent but acquires the same color as the oral mucosa [Figure 1]. The variation in color depends on the size of the lesion, its proximity to the surface and the elasticity of the overlying tissue.[1] Ranula is common in children and it is also the most common lesion associated with sublingual gland.[3] It has been reported that the major salivary glands have penchant for specific disease conditions: pleomorphic adenoma is associated with parotid gland, submandibular gland is the seat of sialoliths, and ranula is associated with sublingual gland.[4]
Figure 1: Photograph of oral ranula showing: Bluish appearance suggesting proximity of the ranula to the oral mucosa

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It is usually soft and painless unless secondarily infected and commonly found lateral to the midline although it can present centrally. The central variety, also referred to as superficial dissecting ranula, is a rare occurrence with bilateral presentation which can be exceptionally large in size.[1] Normally, the lingual frenum acts as a restraining barrier, preventing the ranula from spreading across the midline. However, this may be overridden by excessive amount of mucus leakage and more resistance inferiorly, resulting in excessive superior pressure. The origin of ranula is always unilateral and arises from deep medial surface of the sublingual gland. Even when there is bilateral enlargement, the side of origin is always a bit bigger than the contralateral side.[1] Radiological imaging of the central variety supports the view that even the central ones have their origin in one or the other of the sublingual glands. This helps to distinguish it from dermoid cyst. Histologically, ranulas are pseudocysts and, unlike dermoid cysts, have no epithelial lining but surrounded by nonepithelial layers, filled with mucus, and lined by granulation tissue.[5]

Several surgical modalities have been described in the literature in the management of ranulas and recurrent rates have been based on these different treatment modalities.[3] They include excision of the ranula, excision of ranula and the ipsilateral sublingual gland, marsupialization and its modifications, cryotherapy, hydrodissection, laser ablation, and sclerotherapy.[5],[6] Removal of sublingual gland, and sometimes the submandibular gland, along with the lesion in the management of ranulas have also been debated.[3] Some have argued that removal of sublingual gland will reduce the recurrent rate, while others have disapproved its removal, preferring to do excision only if there is recurrence.[7]

This study was designed to highlight our experience in the management of oral ranulas vis-a-vis the recent concept of the etiopathogenesis that may influence surgical management and outcome of surgery.


  Materials and Methods Top


This was a retrospective study of ranulas seen and managed in King Khalid Hospital, the main referral hospital in the Najran Region of the Kingdom of Saudi Arabia, over a 15-year period from 2004 to 2018. King Khalid Hospital, Najran, has a huge catchment area spanning the length and breadth of the Region.

Data collected include demographics such as age and gender of patients, type of ranula (oral or plunging), modality of occurrence (first time, recurrence or contralateral site), laterality (right, left or bilateral), surgical approach (intra-oral or extra-oral), and type of treatment (marsupialization or excision). Ethical clearance was obtained from the Ethics and Research Committee of King Khalid Hospital, Najran, Kingdom of Saudi Arabia with IRB number H-11-N-081.

Data were stored and analyzed using IBM SPSS Statistics for IOS Version 25 (IBM Corp., Armonk, NY, USA). Results were presented as simple frequencies and descriptive statistics. Statistical significance was set at P ≤ 0.05.


  Results Top


Seventy-five patients with ranula, out of a total of 212 salivary gland lesions (both major and minor), were treated during the review period. Out of the 75 cases of ranulas, 55 were cases of oral ranula, representing 73.3% of the total number of ranulas in this study. The overall prevalence rate of ranula, in a population of 402,000, was 0.19 cases/1000 persons. A prevalence rate of 0.14 cases/1000 persons was observed for oral ranulas only. There were 27 males and 28 females with an M: F ratio of 1:1.03. Their ages ranged from 2 years to 45 years with mean ± standard deviation (22.2 ± 10.7). The age group from 11 to 20 years recorded the highest frequency of 19 cases, representing 34.5% followed by the age group from 21 to 30 years with 16 cases representing 29.1% of ranulas [Table 1].
Table 1: Distribution of age groups of patients with ranula according to their gender

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Of the 55 cases of oral ranula, 27 (49.1%) occurred on the left, 23 (41.8%) on the right while only 5 (9.1%) cases were bilateral [Table 2]. Forty-nine (89.1%) cases presented for the first time, while 2 (3.6%) of the cases were recurrent and 4 (7.3%) cases with involvement of the contralateral side were detected [Table 2].
Table 2: Distribution of laterality, modality of occurrence and approach

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A total of 53 (96.4%) cases underwent excision through intraoral approach together with sublingual gland removal, while only 2 (3.6%) case were marsupialized [Table 3].
Table 3: Distribution of treatment modality according to age group of patients

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  Discussion Top


The sublingual salivary gland is the smallest of the paired major salivary glands. It has a flattened almond shape, weighing about 2 g and measuring about 2.5 cm in anteroposterior dimension. Obstruction of any of its numerous short ducts of Rivinus (12–20) that open along its pinnacle on the sublingual fold results in retention of mucous, leading to ranula.[3] Its main duct, the duct of Bartholin's opens directly onto the summit of the sublingual fold in the floor of the mouth.

Ranulas represent about 1%–10% of all pathologies in the oral cavity, a prevalence rate of 0.2% cases/1000 persons in the population[8] and are ranked 41st in the Minnesota Oral Disease Prevalence Study. This rate is consistent with our current study in which a prevalence rate of 0.19 cases/1000 persons, in a population of 402,000 was observed. However, the result of our study is in contrast with the reported prevalence in Swedish (0.11%)[9] and Brazilian (0.08%)[10] studies. These reported studies only gave the overall prevalence rates of ranulas. However, our study also reported the prevalence of oral ranula of 0.14 cases/1000 persons. A search of the English literature did not reveal any study reporting the prevalence of oral ranulas.

Ranulas have no racial predilection. Female gender predilection has been reported for oral ranula,[11],[12] while other studies have reported no gender preference.[3] Our study showed no clear gender predilection with a male to female (M:F) ratio of 1:1.03, which is consistent with other studies which have reported no gender predilection.[3] No specific age is spared for oral ranulas. Our current study has reported age range from 2 to 45 years while other studies that have reported age range from 5 months to 39 years.[3] A rare case of congenital ranulas in a neonate[13] and another case involving an infant[14] have been reported. Recently, some cases of prenatal ranula have been diagnosed in utero by ultrasonography and managed by ex utero intrapartum treatment (EXIT) at the time of delivery.[3],[15],[16]

Oral ranulas have been reported to occur predominantly in children and young adult.[12] This is in tandem with our current study where most of them were seen in the younger age group. When they present in children and young adults, the symptoms are usually shorter than when they present in adults. From our series, most of the patients presented early between ages 1 and 20 years, corroborating previous reports.

The oral cavity, which has unyielding boundaries, is vital for swallowing and respiration. These vital functions can be easily compromised by large lesions in the floor of the mouth hence the reason for this early presentation.[3] Large oral ranulas can cause discomfort, interference with speech, mastication, swallowing, and produce external swelling.[1] With large oral ranulas, the tongue may compress the ranula to such an extent that during eating and swallowing that there is interference with submandibular salivary flow resulting in obstructive symptoms and enlargement of the ipsilateral submandibular gland.[1] The resultant external submandibular swelling may give the false impression of a plunging ranula so radiological examination such as computed tomographic (CT) scan of magnetic resonance imaging (MRI) usually confirm whether ranula is above the mylohyoid muscle or extends beyond it. Other reported symptoms include failure to thrive, breathing difficulties including snoring, obstructive sleep apnea, and impending airway obstruction.[3]

Difficulty with eating and deglutition has been the major presenting complaints of patients with intraoral ranulas from our study. We have also found out in this study that most of the intraoral ranulas occurred on the left side of the floor of the mouth [Figure 2]. The reason for this site inclination is unknown. Although no study has reported the left side as the most common site of presentation of oral ranulas, plunging ranulas have been reported to occur more on the right side.[11],[12]
Figure 2: Axial view of computed tomographic scan of the ranula showing the source of the ranula

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The possible pathogenesis of ranula is still intricate. Apart from the disruption of the excretory duct resulting in extravasation of mucus from the sublingual gland into surrounding soft tissue, rupture of an acinar structure caused by the ductal obstruction, and damage to the glandular parenchymal cells of the salivary gland lobules have been reported as alternative possible mechanisms.[17] Harrison et al.[18] suggested that the reason why majority of ranulas develop without the history of trauma is that duct obstruction leads to extravasation of mucus from ruptured acini rather than from duct leakage and that the mucus passes through the imperfect sublingual gland capsule into the tissues.

In our series, possible trauma and irritation from chronic oral habits could have been partially responsible for the development of these lesions. Indeed, some of the patients gave a history of habitual use of “shammah” which is also referred to as smokeless tobacco. These substances are placed below the tongue and kept there for some time before being spat out. To buttress our suspicion, it has been reported that mucosal inflammation that involves the minor salivary gland duct may result in blockage, dilatation, and rupture of the duct with subepithelial spillage of fluid. The obstruction may also be due to mucus plugs and debris in Bartholin's duct resulting in superficial ranula.[1] Another cause is iatrogenic as a complication of removing sialolith from the submandibular duct.[1] The submandibular duct passes medially to the sublingual gland so it can be damaged during ranula surgery and conversely a ranula can be initiated during submandibular duct sialolithotomy procedures.[19]

It has also been reported that immunologic responses from content analysates of mucoceles may contribute to the invasive nature of extravasated mucus.[20] Increased levels of proteolytic enzymes such as metalloproteins, tumor necrosis factor-alpha, type IV collagenase, and plasminogen were hypothesized to cause tissue destruction resulting in the expansion of mucoceles.[20],[21] Although the present study did not analyze the contents of these ranulas, future study on chemical analysis of this lesion is underway to test this hypothesis. Other documented risk factors for the development of ranula include variation in the anatomy of the ductal system of the sublingual gland especially where the Bartholin's duct empties into the Wharton duct[22] Sjogren syndrome, sarcoidosis, and HIV infection.[11],[23]

Oral ranula is a clinical diagnosis. Its diagnosis is aided by imaging studies and fine-needle aspiration of the lesion. The imaging studies which have been carried out to determine the extent of the lesion include ultrasound, CT scan, and MRI scan. Some researchers have reported ultrasonography use in the evaluation of oral ranulas,[24],[25] however, this modality of investigation was used in our plunging ranulas cases to ascertain the submandibular or cervical swellings. Ultrasonography is especially important in prenatal diagnosis of congenital ranula.[3],[15],[16] CT scans with intravenous contrast as well as MRI scans have also been used to determine the extent of oral ranulas especially the fairly big ones.[24],[26] These are useful not only in diagnosis but also to determine the size, extent, and relationship with adjacent structures. For our study of oral ranulas, CT scan with intravenous contrast was the modality of imaging used in our series. The axial views of CT scan reveal which sublingual gland is the source of the ranula while the coronal and sagittal views determine whether the ranula is above the mylohyoid muscle as in the case of oral ranula or extends below the muscle and extend into the neck in the case of plunging ranula [Figure 3]a and [Figure 3]b. Fine-needle aspiration of ranula reveals mucus content which has high protein and amylase levels compared to serum.
Figure 3: (a) Axial computed tomographic scan of the oral ranula crossing the midline. (b) Sagittal view of oral ranula displacing the tongue superiorly and posteriorly but confined entirely above the mylohyoid muscle

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Several preferences exist in the management of oral ranulas as the controversies in the literature persist. Such options include: observation,[27] needle aspiration,[28] marsupialization, micro-marsupialization,[29] marsupialization with packing,[1] sclerotherapy[30],[31] laser excision or cryosurgery,[32] surgical excision of the cyst with or without sublingual and/or submandibular gland excision,[33] partial sublingual glandectomy with ranula excision[34] and hydrodissection.[5] However, the gold standard in the treatment of ranula is excision of the ranula with the sublingual gland as this approach to treatment is the key to minimizing recurrence.[1] This is usually achieved through transoral approach.

The advocates of “observation” have based their argument on the fact that ranula can resolve spontaneously after 3 months especially when it occurred in infants.[8] Pandit and Park[35] reported that the optimal management of pediatric oral ranulas may include observation for 5 months for spontaneous resolution. If the lesion persists, then needle aspiration can be done in this pediatric age group. If it does not resolve after this period, surgery is recommended. However, Singh et al.[13] reported a rare case of a large congenital oral ranula in a neonate which was excised directly. Recently, Mneimneh et al.[14] reported a case of huge congenital oral ranula in a 4-month-old patient which was excised after recurrence following marsupialization. When prenatal ranulas have been diagnosed, EXIT has been performed during delivery to decompress the cystic lesion before the umbilical cord was clamped.[3],[15],[16]

Two of our patients presented with impending airway obstruction that required immediate drainage of the cystic content and placement of corrugated rubber drain [Figure 4]a, [Figure 4]b, [Figure 4]c. This was followed later by excision of the remnants of the ranula together with the offending sublingual gland through transoral approach. Similar clinical presentation of impending airway obstruction was reported by Kokong et al.[3] in their series. Sometimes, needle aspiration method is used in the management of such patients as has been reported in the literature.[28]
Figure 4: (a) Extraoral photograph showing patient with a submental swelling and unable to close his mouth properly. (b) Photograph showing very large oral ranula displacing the tongue superiorly and posteriorly. (c) Photograph showing drained ranula kept patent by insertion of corrugated drain temporarily until definitive surgery was undertaken

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Micro-marsupialization has been suggested as an alternative technique in the management of ranula in pediatric age group in the literature.[29] It involves the placement of a 4.0 silk suture through the largest diameter of the lesion (vault of the lesion) without engaging the underlying tissue. A surgical knot is then made, and the suture is left in situ for 7 days and thereafter removed. The reason for this technique is to allow re-epithelization of the severed ducts or the formation of new epithelial-lined ducts that will allow free flow of saliva into the oral cavity.[29] However, this technique is indicated for lesions < 1 cm. The modification of the micro-marsupialization technique has been reported by Sandrini et al.[36] who used the same size of sutures but placed multiple number sutures placed in the lesion, reduced the distance between the entry and exit points of the needle and left the sutures for a longer period of 30 days. Marsupialization with packing utilizes a pressure pack whose continued presence elicits inflammatory response severe enough to initiate fibrosis to permanently seal the leak leading to acinar atrophy. This method has been used in children and the elderly with great success.[1]

These surgical procedures have been fraught with different outcomes of which recurrence of the lesion has been identified by different scholars as the main complication of these techniques.[37] The different recurrence rates, reported for these procedures, include; incision and drainage (71-100%), ranula excision only (0%–25%), marsupialization only (61%–89%), marsupialization with packing (0%–12%), and complete excision of the ranula with the sublingual gland (0%–2%).[37] This low recurrence rate has been reported when total ranula and sublingual gland excisions were made. We have carried out total excision of ranula together with the sublingual gland, transorally in 53 (96.4%) of the patients in our series with a recurrent rate of 3.6% [Figure 5]a and [Figure 5]b. In addition, loose suturing [Figure 6] of the floor of the mouth was also carried out after surgical removal in order to allow free flow of saliva into the floor of the mouth so as to prevent re-accumulation of any residual mucus. Perhaps, this loose suturing technique might have also contributed to the low recurrence rate observed in the current study.
Figure 5: (a) Photograph showing dissection of the ranula and the sublingual gland. (b) Photograph of the surgical field with preservation of the lingual nerve (shown by the white arrow) and the Wharton's duct (shown by the metal hook)

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Figure 6: Photograph showing the loose interrupted sutures used to close the surgical wound

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Although excision of ranula and the sublingual gland cures all oral and plunging ranulas and therefore minimizes risk of recurrence,[12],[38] the morbidity associated with this method is relatively high with the risk of bleeding (1%–2%), infection (1%–2%), lingual nerve injury (2%–12%), and injury to Wharton's duct (2%)[14] with possibility of duct stenosis leading to obstructive submandibular sialadenitis and ductal leakage leading to extravasation of saliva. These complications are largely avoided by employing laser in the treatment of oral ranula. Lai and Poon[38] reported three cases of oral ranula treated by excision with carbon dioxide laser. This is usually achieved by either vaporizing the lesion with defocused beam or using a combination of focused beam to excise the dome and defocused beam to vaporize the wall and base of the lesion. The advantages of laser excision are; good hemostasis, less postoperative swelling, less pain, and less scarring. Because the laser energy penetrates no more than 0.1 mm beyond the cut and the zone of necrosis and thermal damage below the cut is <0.5 mm deep, the risk to the submandibular duct and lingual nerve are minimized.[38] The waterlase laser, an Erbium Chromium Yttrium, Scandium, Gallium Garnett has also been used in the management of ranula.[39]

When surgery is contraindicated or nonsurgical approach is contemplated, sclerotherapy is used as a treatment modality for ranulas. The sclerotherapy agents, which have been found to produce good sclerosing effect, include intralesional steroids, bleomycin which is an antineoplastic antibiotic of Streptomyces verticillus, OK-432 (Picibanil),[30],[40],[41] pingyangmycin (PYM),[42] and botulinum toxin type A.[43]

OK-432 (Picibanil), originally developed as an immunotherapy agent for cancer, is a lyophilized mixture of a low virulent strain of  Streptococcus pyogenes Scientific Name Search ted with benzyl penicillin. In a recent study, Kono et al.[41] in 2017, reported a study of 23 cases of ranula treated with OK-432 and concluded that it was a safe and effective method of treatment compared with other surgical treatments. This procedure causes adverse effects including fever and pain. OK-432 sclerotherapy is contraindicated in patients with penicillin allergy.

PYM, the single component of bleomycin A5, is an anticancer agent that is refined from Streptomyces pingyangensis, with strong damaging effect to vascular endothelial cells, has been used effectively for sclerotherapy of oral ranulas.[42] Chen et al.[42] in 2013, reported the results of treatment of 3 cases of ranula with pingyamycin and concluded that this agent is safe, significantly effective with minimal complications and potentially a better sclerosing agent than OK-432.

Chow et al.[43] reported the first three cases of oral ranula which were successfully treated with intralesional injection of botulinum toxin type A. Oral administration of Nickel Gluconate- Mercurius Heel-Potentised Swine Organ Preparations D10/D30/D200, a homotoxicological agent has been used in the treatment of pediatric ranula.[8],[44] The duration of administration of this drug varies from 6 weeks to a few months and works by accelerating pseudocyst resolution, glandular repair and restoration of function. Garofalo et al.[44] observed a higher success rate in large lesions and attributed it to the increased sensitivity to the action of the drug due to their high vascularity.


  Conclusions Top


In this study, we have reported that oral ranulas represent about 73.3% of the ranulas. Based on the evidence of the possible pathogenesis of ranulas and low recurrence rate from the current study, surgical removal of the lesion and the sublingual gland is essential for the prevention of recurrence. The authors recommend the use of loose suturing in the floor of the mouth following removal of the ranula and sublingual gland to allow free drainage of residual saliva into the floor of the mouth and avoid refilling of the potential space left after the surgery to prevent recurrence.

Acknowledgments

The authors are grateful to the medical record department of King Khalid Hospital, Nahran, for the assistance in retrieving patients' data.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]
 
 
    Tables

  [Table 1], [Table 2], [Table 3]



 

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