Ranula successfully treated by botulinum toxin type A: report of 3 cases

Tam-Lin Chow, FRCS(Edin), FHKAM (Surgery),a Sharon W. W. Chan, FRACS, FHKAM (Surgery),b and Siu-Ho Lam, FRCS(Glas), FHKAM (Surgery),c Kwun Tong, Kowloon, Hong Kong Special Administrative Region
UNITED CHRISTIAN HOSPITAL

The conventional treatment of ranula is surgical procedure. We report an innovative method for ranula by using botulinum toxin type A on 3 patients. All 3 cases of ranula resolved after this minimally invasive therapy. The treatment complication was minimal. (Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2008;105:41-2)

CASE REPORT Case 1

A 25-year-old woman underwent a transoral removal of a stone impacted in the left submandibular duct in December 2003 in another hospital. Marsupialization of the sialodochotomy was also carried out. Initial postoperative recovery was unremarkable. However, a painless swelling developed at the operative site 2 weeks later. The swelling persisted for 1 month, without any sign of resolution. At the time of consultation on January 30, 2004, a 2-cm submucosal cystic swelling (Fig. 1) was evident on the left side of the anterior oor of the mouth. The marsupialization orice was patent and located on the surface of the swelling. No residual stone was found, and there was no submandibular swelling. Based on the history and the ndings, the diagnosis of ranula resulting from sublingual gland injury was made. The patient was informed about the pathophysiology of a ranula and conventional therapy of a sublingual sialoadenectomy. However, she desired an alternative nonsurgical treatment. Based on our past encouraging experience1,2 of managing parotid salivary stula and sialocele, she consented to our proposal of using botulinum toxin type A for her ranula. After needle aspiration of 1-mL clear viscous uid from the cyst, 75 units of botulinum toxin type A (Botox, Allergan Botox Ltd., Westport, Ireland) were injected into the cyst as well as into the left sublingual gland on January 30, 2004.

Fig. 1. Ranula resulting from sublingual gland injury.

Senior Medical Ofcer, Division of Head and Neck Surgery, Department of Surgery, United Christian Hospital, Kwun Tong, Kowloon, Hong Kong Special Administrative Region. b Resident Specialist, Division of Head and Neck Surgery, Department of Surgery, United Christian Hospital, Kwun Tong, Kowloon, Hong Kong Special Administrative Region. c Consultant, Division of Head and Neck Surgery, Department of Surgery, United Christian Hospital, Kwun Tong, Kowloon, Hong Kong Special Administrative Region. Received for publication Oct 31, 2006; returned for revision Mar 7, 2007; accepted for publication Apr 9, 2007. 1079-2104/$ - see front matter 2008 Mosby, Inc. All rights reserved. doi:10.1016/j.tripleo.2007.04.007

No local anesthetic was required. The cyst instantaneously decreased in size but recollected to the original size the next day. However, the cyst markedly attenuated 2 days following injection and ceased to recollect. There was no evidence of recurrence at 28 months after therapy (Fig. 2). In the rst month following treatment, the patient experienced a mild degree of difculty during mastication, which is plausibly related to inadvertent injection to the genioglossal muscle. Thereafter, she adapted to this mild disability quickly and currently enjoys a regular diet. There was no evidence of lingual nerve injury resulting in tongue paraesthesia. She is satised with the outcome of the nonoperative treatment.

Case 2
A 25-year-old woman presented to us in May 2005 with a 2-month history of spontaneous swelling under the tongue. A bluish 2-cm cystic swelling was found under the right side of the tongue, and the diagnosis of ranula was made. She consented to Botox injection, which was carried out with the technique described in Case 1. There was complete resolution at 12-month follow-up.

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Fig. 2. Resolution of ranula after injection of botulinum toxin type A.

Case 3
A 33-year-old woman presented to us on July 28, 2006, with a painless oor-of-mouth cystic swelling, on the right side, which appeared 4 months earlier. On physical examination, a 3-cm submucosal cystic mass was located on the right anterior oor of the mouth. The clinical diagnosis was ranula. The patient preferred Botox injection treatment to surgical procedure. Botox (50 units) was given to the cystic swelling, once on November 10, 2006 and once on January 5, 2007. The oor-of-mouth swelling disappeared clinically when the patient was followed up on March 2, 2007. The patient was very satised with the result.

for better treatment alternatives continues. OK-432 (Picibanil, Chygai Pharmaceutical Co, Tokyo, Japan) has been demonstrated to be useful in some cases, but this drug is not widely available. Adverse effects are considerable; fever and pain at the injection site occurs in half of the patients.5 For these reasons, OK-432 has never gained popularity. This article reports the rst 3 cases of clinical use of botulinum toxin for ranula. The drug acts by the chemical denervation of the secretomotor parasympathetic nerve endings responsible for salivation.4 This medication has been used for a variety of clinical problems for the past 2 decades, and its safety as well as tolerability are well documented.6 The local incidence of ranula is rare. Only these 3 cases of ranula were retrieved from the outpatient database in the past 3 years. Nevertheless, these case reports illustrate a simple, nonsurgical method to treat the ranula successfully by using botulinum toxin type A.
REFERENCES
1. Chow TL, Kwok SPY. Successful use of botulinum toxin type A in a case of parotid sialocele. Hong Kong Med J 2003;9:293-4. 2. Lai ATY, Chow TL, Kwok SPY. Management of salivary stula with botulinum toxin type A. Ann Coll Surg H K 2001;2:65-9. 3. Iida S, Kogo M, Tominaga G, Matsuya T. Plunging ranula as a complication of intraoral removal of a submandibular sialolith. Br J Oral Maxillofac Surg 2001;39:214-6. 4. Yoshimura Y, Obara S, Kondoh T, Naitoh SI. A comparison of three methods used for treatment of ranula. J Oral Maxillofac Surg 1995;53:280-2. 5. Fukase S, Ohta N, Inamura K, Aoyagi M. Treatment of ranula with intracystic injection of the streptococcal preparation OK-432. Ann Otol Rhinol Laryngol 2003;112:214-20. 6. Blitzer A, Sulica L. Botulinum toxin: basic science and clinical uses in otolaryngology. Laryngoscope 2001;111:218-26. Reprint requests: Tam-Lin Chow, FRCS(Edin), FHKAM Department of Surgery United Christian Hospital 130 Hip Wo Street Kwun Tong, Kowloon, Hong Kong SAR tamlinc@yahoo.com

DISCUSSION Ranula, oral or plunging, is a pseudocyst due to the extravasation of saliva from the sublingual gland. Accidental creation of ranula as a result of intraoral removal of the submandibular sialolith has been reported.3 Sublingual gland excision is considered the most reliable method of treatment.4 Nevertheless, general anesthesia is required for this mode of therapy. Moreover, injury to the lingual nerve and submandibular duct are potential complications. Thus, the quest