Annals of Pediatric Surgery, Vol 4, No 3&4 July-October, 2008 PP 89-93

Original Article

Surgical Treatment of Ranula: Comparison between Marsupialization and Sublingual Sialadenectomy in Pediatric Patients
Ehab A. Shehata 1, Hussam S. Hassan2
Maxillofacial & Plastic Surgery Department, Faculty of Dentistry- Alexandria University/ Oral and maxillafacial Unit, Al Hada Armed Military Hospital, Taif, KSA1, Pediatric Surgery Unit, Faculty of Medicine, Tanta University/ Al Hada Armed Military Hospital, Taif, KSA2 Abstract: Background/ Purpose: Several surgical techniques had been introduced to treat intraoral ranula. Marsupialization, excision of the sublingual gland or combined excision of both the ranula and the sublingual gland have been used with variable success rates. The optimal treatment option is still very controversial. This study aims at comparing two surgical methods for treatment of ranulas: marsupialization and sublingual sialadenectomy. Materials & Methods: This prospective randomized clinical study comprised a total of 24 patients with sublingual ranula during the period from 2005 to 2008. Patients were assigned to fit under two equal groups. In group (I), patients were treated by marsupialization while sublingual sialadenectomy was used to treat patients in group (II). Results: Early complications included infection in 2 cases in group I (16.7%) and 3 cases (25%) in group II, and lingual nerve paresis in one patient (8.3%) in sublingual sialadenectomy group. Recurrence occurred in 5 cases treated with marsupialization (41.6%), but no recurrence developed in patients treated by sublingual gland excision. Conclusion: Excision of sublingual gland is attended with minimal morbidity and no recurrence. The authors recommend sublingual sialadenectomy as the surgical treatment of choice for sublingual ranula. Index Word: ranula, marsupialization, excision sublingual gland.

INTRODUCTION

he term ranula generally applies to a bluish, translucent cystic mass in the floor of the mouth..1 It is derived from the Latin word rana, meaning frog as the shape of the cyst resembles the bulging underbelly of a frog.2

There are two different types of ranulas depending on the pathogenesis: It can be a true cyst with an epithelial lining, due to ductal obstruction of the sublingual gland or one of the minor salivary gland; or it can present as a pseudo cyst that originates from ductal injury, extravasation, and accumulation of saliva in the surrounding tissue. In this later type, the

pseudocyst wall lacks epithelial lining, and it consists of granulation tissue surrounded by condensed connective tissue.3 Clinically, ranula may be intraoral or plunging. An intraoral ranula is confined to the floor of the mouth and slowly enlarges to form a painless fluctuant swelling in the floor of the oral cavity. While a plunging ranula is a mucus extravasation pseudocyst arising from the sublingual gland located below the myelohyoid muscle and present as a swelling in the upper part of the neck.4

Correspondence to: Dr. Hussam S. Hassan, Pediatric Surgery Unit. Tanta University. E-mail: hassanh_30@hotmail.com

Shehata E & Hassan H.

Ranulas have been managed by various surgical methods: marsupialization,5 excision of the sublingual gland or combined excision of both the ranula and the sublingual gland.6 Other treatment modalities include intra-cystic injection with OK-432,7 hydrodissection,8 cryosurgery,9 Er,Cr:YSGG10 and carbon dioxide11 laser. The treatment of choice is still very debatable and controversial. The aim of this study is to compare two different surgical techniques for treatment for intraoral ranula: Marsupialization versus sublingual sialadenectomy.

carefully inspected and the orifices were marked by methylene blue.

PATIENTS AND METHODS

This randomized prospective study was conducted from August 2005 till January 2008. Twenty four patients were included in this study. The study was conducted at Al Hada Armed Military Hospital, KSA. All patients had the diagnosis of sublingual ranula by physical examination and CT scan. Inclusion criteria were children up to 12 years with intraoral ranulas and with no associated disease. The study protocol was approved by the research and ethics committee, Al Hada Armed forces hospitals. Written informed consent forms were obtained from parents of participating children. Once the patient was decided by the managing surgeon to be eligible for enrollment, patients were sent to a secretary who blindly assigned each patient into either treatment groups using closed identical envelops. Patients were randomly assigned to one of two equal groups: group I in which patients had been treated with marsupialization, while in group II sublingual sialadenectomy was the assigned surgical technique. All operations were carried out by a single team (the authors). The primary outcome of the study was to evaluate and to compare the efficacy of marsupialization versus sublingual sialadenectomy as different surgical modalities in the treatment of sublingual ranula. While the secondary end points were to detect the frequency of the treatment related complications. Follow up for both groups ranged from 2 to 12 months with the mean of 9 months. Surgical technique: Marsupialization: The patients were admitted in the day surgery unit. They were put asleep via nasal endotracheal intubation. Openings of submandibular ducts were Fig 1. Exposure of an intraoral ranula.

Fig 2. Marsupialization of an intraoral ranula.

Fig 3. Excised sub4mandibular gland.

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Probing of the submandibular ducts were not required. Marking the cystic swelling of ranula was followed by de-roofing of the cystic lesion. Hemostasis was achieved using bipolar diathermy. Under 2.5x magnification loupe, suturing the cystic wall to the mucosa of the floor of the oral cavity using vicryl 5/0. Patients were discharged with analgesic /antipyretic medication (Figs. 1-2). Sublingual sialadenectomy: Patients were anesthetized by nasal endotracheal anesthesia. A lacrimal probe was introduced into submandibular duct. Unilateral or bilateral duct probing was judged according to the proximity of the ranula to the duct course. The metal probe was kept intraductal by a purse string suture around, to facilitate dissection of the area with minimal risk of duct injury. Submandibular duct(s) was dissected using transverse incision parallel to the distal part of the duct. Small vessels in the area were coagulated carefully by bipolar diathermy. Sublingual gland was then freed from the submandibular duct and any branches of lingual nerve by blunt dissection. The most distal 2-4 mm of submandibular ducts were excised, the lumen was then incised vertically 2 mm to widen the opening which was then relocated and sutured to the edge of mucosal incision using vicryl 5/0. Mucosal approximation was then carried out using the same absorbable suture material aiming to minimize post operative pain and heamatoma formation (Fig. 3).

(25%) in group II. The cases were treated with antibiotics according to culture and sensitivity, and anti septic mouth gargle (in older children). Unilateral numbness and parasthesia of the tongue, a manifestation of lingual nerve paresis was documented in 1 patient (8.3%) in sublingual sialadenectomy group. The case was expectantly managed, and recovered spontaneously within 3 months. There was no statistically significant difference between the two groups with respect to early complications. Late complications included recurrence in 5 cases (41.6%) in group I, but none in group II developed recurrence (0 %). This difference was statistically significant. Table 1. presents early and late post operative morbidities Table 1. Early and late morbidity. Morbidity Early: Infection Lingual nerve paresis Late: Recurrence 5 (41.6%) 0 0.04(S) 2 (16.7%) 0 3 (25%) 1 (8.3%) 0.84 (NS) 1.0 (NS) Group I Group II P value

RESULTS
In three years period, we operated on 24 patients divided equally into both groups I & II, each group included 12 patients. There were 10 males and 14 females, their age ranged from 1 month to 12 years with mean age of 4.7 years. Univariant regression analysis was used to ensure that the two groups are comparable with respect to age and sex. The statistical test used (Fisher exact test) was 2- sided. P values less than 0.05 were considered to indicate statistical significance. Statistical analysis was performed using SPSS version 13, Chicago, USA. There were no mortalities. Early morbidity included: infection in 2 cases in group I (16.7%) and 3 cases
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DISCUSSION
In our study, we focused on ranula in pediatric age group. Surgical treatment of such cystic lesion in children poses more difficult situation compared to adult patients. This could be explained by smaller surgical field, thinner wall, greater friability and closer proximity of the cystic lesion to vital structures in the floor of the oral cavity. A variety of non surgical procedures have been proposed for therapy of ranula, all of them aim at avoiding surgery in the floor of the mouth, which may be complicated by the proximity to important structures like the submandibular duct and the
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lingual nerve and artery. Intra cystic injection of sclerotherapy agents like OK432 (a lyophilized mixture of low-virulence group A Streptococcus pyogens with penicillin G potassium) has been reported to be highly effective in management of intraoral ranula.14 Many researches recommend this technique as a primary treatment (done on an outpatient basis), while leaving surgical option for recurrent cases.15,16-17 Vaporization of ranula by various types of laser is also widely practiced. The minimal lateral tissue damage, seen with the laser, minimizes the risks. In addition, the bloodless nature of the surgery in this vascular area adds to increased safety by allowing more visibility of the surgical field.11,12 However, we believe that surgery is still the primary standard therapy of ranula. There are several surgical intervention methods. There is general agreement that incision only as a treatment should not be done because of rapid closure of the wound and recurrence of ranula.18 In practice, the two widely practiced techniques are marsupialization and sublingual gland excision. Marsupialization, which preserves the sublingual gland and adjacent tissue, is still practiced despite reported recurrence rates as high as 61 to 89%.19 In this prospective randomized study we compared both techniques with respect to short and long term morbidity. Short term complications included surgical site infection, which responded readily to medical treatment. Lingual nerve parasthesia eventually improved without intervention. There was no statistically significant difference between the two groups with respect to early morbidity. We did not encounter complications related to submandibular duct injury as we used a fine lacrimal probe to canulate it prior to dissection. Our complication rate is comparable to those reported in literatures.12 The recurrence rate after marsupialization was 41.6%. This is comparable to other reports 20, although some authors reported very high recurrence rate up to 89%.19 The recurrent cases in marsupialization group were treated successfully by sublingual sialadenectomy. None developed recurrence within the follow up period. The recurrence after excision of the sublingual gland was 0%, this is comparable to other researchers reporting recurrence rate 0-2%.13,19,21

CONCLUSION
We recommend excision of the sublingual salivary gland which is attended with morbidity rates statistically comparable to marsupialization, and has a recurrence rate approaching zero percent as a firstchoice treatment option for sublingual ranula. Marsupialization, although simple and fast, has high recurrence rate; and operating on a recurrent case of ranula has an increased risk of injury to important structures in the operative field such as the lingual nerve and artery, and the submandibular duct. It is safer to dissect submandibular duct(s) before removal of sublingual gland(s). loupe magnification improve dramatically visualization during dissection especially in pediatric patients.

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