Management of infant ranula

Management of infant ranula

International Journal of Pediatric Otorhinolaryngology (2008) 72, 823—826 www.elsevier.com/locate/ijporl Management of infant ranula Keqian Zhi a,*,...

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International Journal of Pediatric Otorhinolaryngology (2008) 72, 823—826

www.elsevier.com/locate/ijporl

Management of infant ranula Keqian Zhi a,*, Yumin Wen b, Wenhao Ren a, Yincheng Zhang a a

Department of Oral and Maxillofacial Surgery/Oncological Head and Neck Surgery, College of Stomatology, Xi’an Jiaotong University, Number 98, Xiwu Road, Xi’an, ShaanXi 710004, China b Department of Oral and Maxillofacial Surgery, West-China College of Stomatology, Sichuan University, Chengdu, Sichuan, China Received 13 January 2008; received in revised form 10 February 2008; accepted 11 February 2008 Available online 2 April 2008

KEYWORDS Ranula; Sublingual gland; Management

Summary Objective: Many surgical techniques to manage ranulas have been described in the literature. Ranula of infant patients was rare. Few studies have described the approach toward management in infant patients. Methods: Eleven infant patients were treated for intraoral ranulas. The methods of treatment included aspiration of mucus, marsupialization and excision of the ranula and the ipsilateral sublingual gland. All cases were performed aspiration of mucus and observed for 6 months; and the marsupialization were recommended if the ranula recurred; the surgical resection of ipsilateral sublingual gland were performed if the ranula recurred when infant patients was about 1-year-old. These patients were followed up at least 24 months. Results: Age of presentation ranged from 2 days to 3 months. There were six females (54.55%) and five males (45.45%). All cases presented simple (introral) ranula. Excision ranula with sublingual gland was performed on seven patients (63.64%) while marsupialization was performed on two patients (18.18%) and two patients (18.18%) were aspiration of the mucus of ranula and no recurrence. There were no recurrent lesions in all cases. Conclusion: Conservative treatment of infant ranula maybe includes observation for 6 months for spontaneous resolution. The methods for observation is performed the aspiration of mucus and marsupialization. The resection of ipsilateral sublingual gland is recommended if ranula recurred for infant patients about 1-year-old. We believe that it is safe that the submandibular duct and complete sublingual nerve are dissected before the sublingual gland is removed. # 2008 Elsevier Ireland Ltd. All rights reserved.

1. Introduction * Corresponding author. Tel.: +86 29 81015479. E-mail addresses: [email protected], [email protected] (K. Zhi).

Ranula is reported by Hippocrates and Celsius [1]. Ranula is derived from the Latin word rana which

0165-5876/$ — see front matter # 2008 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2008.02.012

824 means ‘‘frog’’, because the lesions in the floor of the mouth resemble the bulging underbelly of a frog [2]. Theoretically, the ranula formation is excretory duct rupture followed by extravasation and accumulation of saliva into the surrounding tissue [3]. The accumulation of mucus in the surrounding connective tissue forms a pseudocyst that lacks an epithelial lining [4]. The analysis of the saliva reveals high protein and amylase concentration consistent with secretions from the mucinous acini in the sublingual gland. The high protein content maybe produces a very intense inflammatory reaction and mediate pseudocyst formation. Many methods of treatment for ranulas have been described in literature, including excision of the ranula only, excision of the ranula and the ipsilateral sublingual gland, marsupialization and cryosurgery [4,5], the definitive treatment is now considered to be surgical excision of the ipsilateral sublingual gland, which is supported by the recent studies [6]. The lingual nerve and submandibular duct, which are close to the sublingual gland, will be at risk for injury from this approach, especially in infant patients. This report summarizes our experience with the management of 11 infant patients with oral cavity ranulas.

2. Patients and methods A total of 11 children referred to stomatological Hospital of Xi’an Jiaotong University and Sichuan University between 1995 and 2005 were assessed and treated for oral cavity ranula. Typically, the patients presented with painless, a slight blue, translucent swelling in the unilateral floor of mouth. There were no precipitating factors such as trauma or infection. All patients underwent clinical assessment followed by fine needle aspiration cytology (FNAC) and Biochemical analysis revealed high protein and amylase concentrations and subjected to definitive diagnosis. Radiological investigations such as ultrasonography, computerized tomography (CT) scanning, or magnetic resonance imaging (MRI), were not performed in all cases. Informed consent was obtained from their parents prior to the commencement of treatment. This study was approved by Institutional Review Board of our hospital. The methods of treatment included aspiration of mucus, marsupialization and excision of the ranula and the ipsilateral sublingual gland. All cases in this series were performed the aspiration of mucus at first. The spontaneous drainage was formed after the aspiration of mucus is performed on the ranula. The infant patients were observed for 6 months and repeated aspiration of mucus if the ranula recurred.

K. Zhi et al. Six months would provide an adequate time for resolution based on our results. The presence of amylase from an aspirated cyst sample will confirm the diagnosis. If the lesion did not resolve within this period or repeatedly recurred after spontaneous drainage, the marsupialization was performed for all of recurrence. The excision of the ranula and the ipsilateral sublingual gland was recommended if it recurred again after the marsupialization at about 1-year-old. All specimens showed a thin walled cyst with no squamous epithelial lining in histopathology. Following surgery the patient was placed on a normal diet, 5 days of therapy with antibiotic orally. The drain with rubber strip was removed after 48 h and the suture were removed 7 days for the cases with excision of the ipsilateral sublingual gland. All cases followed up at 24 months.

3. Results Table 1 summarized the clinical course of 11 infant patients with oral cavity ranulas. Age of presentation ranged from 2 days to 3 months (Figs. 1 and 2). There were six females (54.55%) and five males (45.45%). All cases presented simple (introral) ranula. All cases were performed aspiration of mucus, marsupialization and resection of ipsilateral sublingual gland because the spontaneous resolution and so younger on infant patients. Ranula of two infant patients (18.18%) disappeared and not recurred by aspiration of mucus. Two infant patients (18.18%) was performed marsupialization and ranula did not recur. Seven infant patients (63.64%) were recommended the resection of ipsilateral sublingual gland and total excision of the pseudocyst is probably unnecessary when infant patients were about 1-year-old. We did not encountered recurrence in all cases. Followed up for all cases was 24 months.

4. Discussion Ranula can be classified into two groups, simple (intraoral)) and the plunging (cervical) type. Simple ranula is much more common than plunging type. A simple ranula represents a localized collection of mucus within the floor of the mouth. In plunging ranula, the mucus collection is in the submandibular and submental space of the neck with or without an associated intraoral collection. In our study, all cases presented simple ranula. We think it is the reason the ranula were referred so early that the ranula did not developed to cervical.

Management of infant ranula

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Table 1 Patient summary Patient

Age

Treatment

1 2

3 days 3 days

3

1 month

4

2 months

5 6

7 days 1 month

7

2 months

8

3 months

9 10 11

4 days 3 days 7 days

Aspiration of mucus Aspiration of mucus + marsupialization + ipsilateral sublingual gland Aspiration of mucus + marsupialization + ipsilateral sublingual gland Aspiration of mucus + marsupialization + ipsilateral sublingual gland Aspiration of mucus + marsupialization Aspiration of mucus + marsupialization + ipsilateral sublingual gland Aspiration of mucus + marsupialization + ipsilateral sublingual gland Aspiration of mucus + marsupialization + ipsilateral sublingual gland Aspiration of mucus + marsupialization Aspiration of mucus Aspiration of mucus + marsupialization + ipsilateral sublingual gland

Age of resection

Outcome

1 year

No recurrence No recurrence

1.2 years

No recurrence

1.5 years

No recurrence

9 months

No recurrence No recurrence

9 months

No recurrence

1.2 years

No recurrence

1.2 years

No recurrence No recurrence No recurrence

Other differential diagnosis in this age group is a vascular malformation/lymphangioma and mucoceles of the minor salivary gland. Mucoceles are mostly due to extravasation of mucus from a minor salivary gland, although a few are true retention phenomena. The most common site is the lower lip. Mucoceles should not recur if the underlying damaged minor salivary gland has been removed. Lymphangiomas or lymphatic malformationsare defined as isolated regions of lymphatic tissue when developing lymphatic tissue fails to properly anastomose. Lymphangioma in histopathology can be classified as capillary, cavernous, or cystic. When aspirated, the cystic space fluid includes proteinaceous fluid with few lymphocytes. Diagnosis of lymphangioma is principally made on the basis of clinical appearance and imaging.

The causes of ranula formation were thought to be trauma or surgery to the floor of the mouth. Trauma to the floor of the mouth or neck region may rupture the sublingual gland acini or cause obstruction of the sublingual gland ducts which results in mucus extravasation [7]. In our study, all cases never had the history of trauma and the parents of infant patients were investigated the detail when the baby was born. Although the cause for newborn ranula is unknown now in literature, we suggest the ranula, maybe, congenital in origin and deep research will be done. Many surgical techniques to manage ranulas have been described in literature. Among these, there is excision of the cyst with or without excision of the ipsilateral sublingual gland, marsupialization, cryosurgery, and CO2 laser excision. Spontaneous resolution may be another option for pediatric population.

Fig. 1 Sublingual ranula of right floor mouth in 1-month infant.

Fig. 2 Sublingual ranula of right floor mouth in 3-day infant.

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K. Zhi et al.

If the lesion does not resolve after 6 months observation for spontaneous resolution or recurs repeatedly, surgical treatment is recommended [8]. In our study, two infant patients (18.18%) in this series had spontaneous resolution of their lesions after aspiration of mucus of ranula. We advocate that the oral cavity ranula of infant patients should be observed for 6 months. Six months is an adequate time for resolution based on our results. If the lesion does not resolve within this period or repeatedly recurs, the marsupialization is recommended. Marsupialization of the ranula can be treated with the placement of a silk suture or seton into the dome of the cyst [9]. It was not recommended because of the risk of the excessive failures and the high incidence of iatrogenically caused cervical ranula that may follow this procedure [10]. In this series, our study showed that two infant patients were performed the marsupialization and not recurred follow-up 24 months. We think that marsupialization should be chosen as a conservative treatment for infant ranula. Surgical removal of the sublingual gland and pseudocyst is recommended, if ranula in oral cavity does not resolve after marsupialization. We suggest excision of sublingual gland should be performed in case it repeated as other studies stated in their articles [10,11]. Total excision of the pseudocyst is probably unnecessary and places surrounding structures at risk of damage, but a biopsy of the pseudocyst wall is important to confirm the diagnosis [12]. The complications of excision of sublingual gland are injury to sublingual nerve and Wharton’s duct and resulted in subsequent nerve numbness and the possibility of obstructive sialadenitis. The submandibular duct was dissected and it was transposed to the floor of mouth to enhance exposure if it is necessary. The sublingual nerve was completely dissected and identified before sublingual gland was removed. In this series, we did not encountered complications during surgical treatment.

In conclusion, infant ranula is rare. Conservative treatment of infant ranula is recommended and it is adequate period of observation for 6 months for spontaneous resolution. The methods for observation are performed aspiration of mucus and marsupialization for spontaneous resolution. If the ranula in oral cavity does not resolute and repeatedly recurs, the resection of ipsilateral sublingual gland should be performed on patients about 1-year-old. We believe that it is safe that the submandibular duct and complete sublingual nerve are dissected before the sublingual gland was removed.

References [1] C.A. Quick, S.H. Lowell, Ranula and sublingual salivary glands, Arch. Otolaryngol. 103 (1977) 397—400. [2] W.S. Crysdale, J.D. Mendelsohn, S. Conley, Ranulasmucoceles of the oral cavity: experience in children, Laryngoscope 98 (1988) 296—298. [3] S.L. Bronstein, M.S. Clark, Sublingual gland salivary fistula and sialocele, Oral Surg. 57 (1984) 357—361. [4] R.P. Morton, J.R. Bartley, Simple sublingual ranulas: pathogenesis and management, J. Otolaryngol. 24 (1995) 253—254. [5] Y. Yoshimura, S. Obara, T. Kondoh, S. Naitoh, A comparison of three methods used for treatment of ranula, J. Oral Maxillofac. Surg. 53 (1995) 280—282. [6] Y.F. Zhao, J. Jia, Y. Jia, Complications associated with surgical management of ranulas, J. Oral Maxillofac. Surg. 63 (1) (2005) 51—54. [7] M. Mahadevan, N. Vasan, Management of pediatric plunging ranula, Int. J. Pediatr. Otorhinolaryngol. 70 (2006) 1049— 1054. [8] R.T. Pandit, A.H. Park, Management of pediatric ranula, Otolaryngol. Head Neck Surg. 127 (1) (2002) 115—118. [9] R.P. Morton, J.R. Bartley, Simple sublingual ranulas: pathogenesis and management, J. Otolaryngol. 24 (4) (1995) 253— 254. [10] H.D. Baurmash, Marsupialization for treatment of oral ranula: a second look the procedure, J. Oral Maxillofac. Surg. 50 (12) (1992) 1274—1279. [11] N.E. Langlois, P. Kolhe, Plunging ranula: a case report and a literature review, Hum. Pathol. 23 (11) (1992) 1306—1308. [12] M.J. Davison, R.P. Morton, N.P. Mclvor, Plunging ranula: clinical observations, Head Neck 20 (1998) 63—68.

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