Oral Biol Res 2019; 43(4): 340-348  https://doi.org/10.21851/obr.43.04.201912.340
Sialolithiasis in children: Three case reports
Su-Young Park1 , Sang-Ho Lee1 ,2, Nan-Young Lee1 ,2, and Myeong-Kwan Jih1,2*
1Department of Pediatric Dentistry, School of Dentistry, Chosun University, Gwangju, Republic of Korea
2Oral Biology Research Institute, Chosun University, Gwangju, Republic of Korea
Correspondence to: Myeong-Kwan Jih, Department of Pediatric Dentistry, School of Dentistry, Chosun University, 309 Pilmun-daero, Dong-gu, Gwangju 61452, Republic of Korea.
Tel: +82-62-220-3868, Fax: +82-62-225-8240, E-mail: mdenti@chosun.ac.kr
Received: October 23, 2019; Accepted: October 29, 2019; Published online: December 31, 2019.
© Oral Biology Research. All rights reserved.

This is an open-access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0) which permits unrestricted noncommercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
Abstract

Sialolithiasis is a disease in which calcified substances accumulate in the ducts or parenchyma of the salivary glands. This disease can occur in people of any age, but it is predominantly seen in middle-aged adults and is rare in children. Owing to their anatomical and physiological characteristics, sialoliths commonly begin to form in the submandibular gland. The main symptoms of sialolithiasis are pain and swelling. Asymptomatic cases have also been reported. Clinical examinations and radiological examinations are used to diagnose sialolithiasis. Moreover, therapeutic methods comprise conservative treatments to promote salivation, surgical removal, and extracorporeal shockwave lithotripsy. Further, three pediatric patients visited the Department of Pediatric Dentistry, Chosun University Dental Hospital owing to sialolithiasis. All patients were asymptomatic, and clinical examination revealed a yellowish substance located near the Wharton’s duct in the oral cavity in each case. The sialoliths were removed by transoral sialolithotomy under local anesthesia.

Keywords: Children, Salivary duct, Sialolithiasis, Submandibular gland
INTRODUCTION

Sialolithiasis is a disease in which calcareous concretions occur in the ducts or parenchyma of the major and minor salivary glands [1]. Sialolithiasis can occur at any age, but is seen predominantly in adults aged 30–60 years [2]. It occurs in about 1% of the total population, and pediatric cases are very rare, accounting for about 3% of all sialolithiasis cases [3-5]. Most sialoliths occur in the submandibular gland, but these concretions also occur in the parotid, sublingual, and minor salivary glands [6-8]. Sialolithiasis in the submandibular gland is attributable most commonly to the anatomical and physiological characteristics of this gland, such as the longer duct, flow of saliva against gravity, and discharge of alkaline saliva with high viscosity [5,9].

Sialoliths are associated with many clinical symptoms, but the main symptoms are pain and swelling. Retention of saliva resulting from partial or complete obstruction of the duct by sialoliths, and subsequent expansion of the duct, cause pain and swelling. These symptoms occur during or after meals. Symptoms may include fever, pus discharge, and trismus, but asymptomatic cases have also been reported [5,10].

Sialolithiasis is diagnosed based on radiographic and clinical examinations, including medical anamnesis, ocular inspection, and bimanual palpation of the affected salivary gland and duct [11]. Although occlusal and panoramic radiographs may aid in rapid detection of the presence of radiopaque sialoliths, insufficient calcification of some sialoliths may result in false-negative findings based on radiolucency [5]. Sialography is useful in the detection of sialoliths, and high-frequency ultrasound, computed tomography (CT), magnetic resonance imaging (MRI), and sialendoscopy can be used to diagnose sialolithiasis [12].

Treatment is determined by the size, location, and number of sialoliths, and the degree of interference with excretion from the duct. Conservative treatments, such as hydration, massage of the affected salivary glands, warming, and administration of sialagogues, antibiotics, and non-steroidal anti-inflammatory drugs (NSAIDs), can be used in cases in which sialoliths are smaller than the diameter of the duct. Sialoliths that cannot be removed by opening the duct can be removed by trans-oral or extra-oral sialolithotomy, extracorporeal lithotripsy, sialendoscopy, or laser treatment. Sialoadenectomy is performed in cases in which sialoliths are located in the salivary gland or proximal portion of the duct, calcification is scattered, or sialoadenitis has developed due to prolonged duct obstruction [13-21].

We report the clinical symptoms and surgical treatment of sialoliths in the submandibular duct in patients who visited the Department of Pediatric Dentistry, Chosun University Dental Hospital, Gwangju, Korea.

CASE

Case 1

A 10-year-old boy visited the Department of Pediatric Dentistry at Chosun University with the chief complaint of sialoliths at the base of the tongue. The sialoliths were observed during oral examination at a private dental clinic and the child was referred to our hospital. The patient’ s history included kidney surgery at birth, and he had no symptoms such as discomfort or pain. Clinical examination revealed a yellowish mass on the surface of the submandibular duct opening on the left side of the oral cavity (Fig. 1A). The patient had no facial asymmetry or lymph node enlargement. The lesion was painful on palpation, and characterized by fluidity and firmness. On mandibular occlusal radiographs (Fig. 1B), radiopaque masses were detected in the left anterolingual region. Although the presence of the mass was confirmed visually and fluidity was present, removal of the mass by manual manipulation through the duct orifice would have been difficult; thus, surgical sialolithotomy was planned.

Fig. 1.

(A) Initial intraoral photograph. A 3.0×3.0 mm of yellowish mass was observed on the left of middle side of the mouth floor. (B) Initial occlusal radiograph. Two radiopaque masses were observed in the left side of the mandible.


Under local anesthesia, two sialoliths were removed through a minimal incision in the upper mucosa of the duct in which they were located (Fig. 2A). The margin of the incision was unclear due to the irregular surface patterns of the sialoliths (Fig. 2B), and a cut-down tube (poly-ethylene tube) was inserted to prevent stenosis of the duct and ranula development. After confirmation of saliva flow into the tube, the length of the tube was adjusted and it was fixed in place using Vicryl 5-0 (Fig. 2C). However, the child’s constant tongue movement due to tube insertion caused repeated tube dropouts. One month later, normal salivary flow was evident when the salivary gland was stimulated, and the patient showed good healing and no inflammation (Fig. 2D).

Fig. 2.

(A) The two removed sialolith with size of 2.5×3 mm and 3×4 mm showed a rough surface. (B) After removal of the sialolith. The margin of incision was not clear. (C) A cutdown-tube was inserted to prevent the stenosis of the duct. (D) One month later, the surgical site was healed well.


Case 2

A 5-year-old boy was referred to our hospital because of a lump that had developed under his tongue. The lump had first been observed 1 month previously during treatment of dental caries in a private dental clinic, and had since increased in size threefold. The patient had no spontaneous pain, but pain occurred on palpation. The patient’s medical history was unremarkable.

On clinical examination, a yellowish lesion about 3× 5 mm in size was observed in the vicinity of the submandibular opening on the right side of the oral cavity. No facial asymmetry or sign of infection in the salivary gland was observed (Fig. 3). On mandibular occlusal radiographs and CT images, a radiopaque mass was detected in the right anterolingual region (Fig. 4). Surgical sialolithotomy was planned because removal of the mass through the duct orifice by manual manipulation was considered impossible.

Under local anesthesia, an oval sialolith with a smooth surface was removed through a minimal incision in the upper mucosa of the duct in which it was located (Fig. 5A). Minimal bleeding occurred at the incision site, and the margin of the incision was smooth. Sutures were not placed due to the risk of duct obstruction during suturing (Fig. 5B). After 1 week, the child had normal salivary flow and no inflammation (Fig. 6A). At 8 months later, no recurrence was observed and normal salivary flow was evident upon palpation (Fig. 6B).

Fig. 3.

Intial intraoral photograph. A 3×5 mm of yellowish solid mass was observed on the right side of the mouth floor.


Fig. 4.

On occlusal radiographs (A) and computed tomography scan (B), a radiopaque mass located on the lingual side of the mandibular incisor region was observed.


Fig. 5.

(A) The removed sialolith with size of 3×5 mm showed an oval smooth surface. (B) After removal of the sialolith. The margin of incision was clear.


Fig. 6.

(A) One week after the operation, well-healed state of the affected area was observed. (B) Eight months after the operation, no signs of recurrence or complications were observed.


Case 3

A 7-year-old girl visited the hospital clinic with the chief complaint of a mass in the lower part of the tongue. The parents had first identified the mass 1 month previously while brushing the child’s teeth, and they reported no change in size since that time. The patient had no spontaneous pain or pain on palpation. The medical history was unremarkable. On clinical examination, a yellowish mass about 2×2 mm in size was observed near the submandibular duct orifice on the right side of the oral cavity, with no facial asymmetry or sign of infection in the salivary gland (Fig. 7A). On mandibular occlusal radiographs, a radiopaque object was seen in the right anterolingual region (Fig. 7B). Surgical sialolithotomy was planned because removal by manual manipulation of the sialolith was considered impossible.

Fig. 7.

(A) Initial intraoral photograph. A 2×2 mm of yellowish mass was observed on the right side of the mouth floor. (B) Initial occlusal radiograph. A radiopaque mass was observed on the left side of the mandible.


Under local anesthesia, the sialolith was removed through a minimal incision in the upper mucosa of the duct in which it was located, and suturing of the incision site was not performed (Fig. 8A, B). After 1 week, the child had recovered well, with no inflammation and normal salivary flow (Fig. 9A). After 6 months, no symptom of recurrence was observed and normal salivary flow was evident upon palpation (Fig. 9B).

Fig. 8.

(A) A 3×3 mm size of removed sialolith with a smooth surface. (B) After removal of the sialolith. The margin of incision was clear.


Fig. 9.

(A) One week after the operation, well-healed state of the affected area was observed. (B) Six months after the operation, no signs of recurrence or complications were observed and the saliva flow was observed.


DISCUSSION

Sialoliths are calcified accumulations that occur in the glandular tissue parenchyma of the major and minor salivary glands and in ducts. The central part of a sialolith consists of materials such as bacteria, foreign substances, and exfoliated epithelial cells on which a calcium salt is deposited concentrically to form a lamellar structure [22,23]. Sialoliths occur mainly in middle-aged adults, and are rare in pediatric patients. McCullom et al. [23], Waseem and Forte [24], Trivedi [21], and Shinohara et al. [25] reported cases of sialolithiasis in children. In Korea, Lee et al. [26] reported sialolithiasis in a 5-year-old boy in 1997, and Lee et al. [11] reported sialolithiasis in a 5-year-old girl in 2009. The prevalence of sialolithiasis in children is low because the condition requires a considerable amount of time to develop, and the sublingual papillae and cross sections of the salivary glands are very small, making invasion by foreign substances difficult [21]. In addition, concentrations of calcium and phosphorus in the saliva increase with age, facilitating sialolith formation in adults. In children, saliva flow is rapid; thus, most sialoliths are located distally. Due to the smaller size of these sialoliths, obstruction is shortlived and quickly results in the patient visiting the hospital [27,28]. Conflicting information on the incidence rates in male and female has been presented. Shinohara et al. [25] reported that sialolithiasis occurs more frequently in girls than in boys, with a rate of 1:1.6 in children younger than 10 years. However, Nahlieli et al. [29] reported a higher incidence of sialolithiasis in boys than in girls. Sialoliths are round or oval, and range in size from a few millimeters to a few centimeters [30]. Most are unilateral; bilateral occurrence is rare, accounting for only 0.5%–2.2% of all sialolithiasis cases [5].

Approximately 80%–90% of sialoliths occur in the submandibular gland, 5%–15% occur in the parotid gland, and the remainder occur in the sublingual and minor salivary glands [6,7]. The amount of saliva discharged by the submandibular gland into the oral cavity is about 40% of the total amount of saliva secreted into the oral cavity. The submandibular gland is about 5 cm in length and 2–4 mm in diameter, and it excretes saliva through Wharton’s duct [31,32]. Sialoliths occur more commonly in the submandibular duct than in the parotid duct because the former is longer and located below the excretion port. These factors cause the saliva to move against gravity and undergo reversal at a right angle to the posterior border of the mylohyoid muscle, facilitating retention of saliva in the submandibular duct. In addition, high concentrations of calcium and phosphate in the saliva in the submandibular gland allow formation of apatite. The saliva secreted from the submandibular gland is more alkaline and mucinous than that secreted from the parotid gland, and the rate of saliva movement is slow; thus, sialolithiasis occurs frequently in the submandibular gland [21,24,33]. Unilateral sialoliths were observed near the submandibular duct opening in the cases reported here.

No general agreement has been reached regarding the causes and mechanisms of sialolith occurrence, although several hypotheses have been proposed. Lustmann and Shteyer [34] suggested that sialolith formation is initiated by increases in saliva pH and organic substances due to bacterial infection. In addition, morphoanatomical factors (salivary duct stenosis, inflammation or injury of the ductal epithelium, foreign body inflow) cause calcium-rich saliva deposition, nucleus formation for calcium deposition, and an increase in saliva bicarbonate by an unknown metabolic process, and a decrease in the solubility of calcium phosphate and precipitation of calcium and phosphorus are believed to contribute to the formation of sialoliths [9,35]. Changes in salivary factors (high supersaturation or crystallization inhibitor deficits) disturb the internal state of the salivary duct [9,35]. Waseem and Forte [24] reported a case of sialolithiasis due to a high-calcium diet and calcium supplement usage. Grases et al. [35] reported that salivary calcium concentrations were high and magnesium and phytate concentrations were low in patients with sialolithiasis. The first of our cases had a history of kidney surgery at the time of birth, and we cannot exclude the possibility of recurrence due to the effect of this procedure on calcium metabolism. Therefore, regular follow-up will be necessary in this case.

The clinical symptoms of sialoliths vary. The most common symptoms are swelling and pain in the affected salivary gland. Pus discharge, fever, and trismus have also been reported, but sialoliths may also be asymptomatic [5,10]. Continuous edema in patients with chronic obstruction of a duct by a sialolith may lead to chronic sialoadenitis. When a sialolith blocks a duct and pressure increases, but the saliva can flow around the sialolith, or when a branch of the duct, rather than its main body, is blocked, partial obstruction occurs. Pain usually increases intermittently after a meal (colicky postprandial pain) and is relieved at rest due to the increased secretion of saliva into a duct obstructed partially or completely by a sialolith [12,36-38]. In the present case report, two of the children were referred to the hospital clinic because sialoliths were observed during treatment in private dental clinics, and a child visited our hospital because the parents observed sialoliths during oral hygiene management. All three children were asymptomatic, and no previous history of edema or pain was noted during anamnesis. We inferred that these sialoliths did not completely block the ducts and were not able to inhibit saliva excretion.

In the diagnosis of sialolithiasis, radiological and clinical examinations, including ocular inspection and palpation, sialography, sialendoscopy, high-frequency ultrasound, CT, and MRI, can be performed. Bimanual palpation of the oral cavity is useful for the examination of sialoliths in the submandibular gland ducts [39]. Radiographic examinations consist of the acquisition and evaluation of panoramic and occlusal radiographs. However, some sialoliths are not sufficiently calcified to display radiopacity. Lustmann et al. [5] reported that 2% of sialoliths were radiolucent. Williams [9] reported that 80%–94.7% of submandibular sialoliths were radiopaque. Blatt [40] reported that 20% of submandibular sialoliths were radiolucent and could be detected only by sialography. Sialography is used to examine the position and condition of the salivary gland via injection of radiopaque contrast agent into the gland. However, sialography is not recommended when a sialolith is located anteriorly due to the risk of pushing it into the posterior part of the duct under the force of contrast agent injection [41]. In the cases reported here, the diagnosis of sialolithiasis was based on panoramic and occlusal radiographic features, and the size and location of the sialoliths were confirmed by ocular inspection and palpation.

Sialolith elimination is essential for the treatment of this condition. Treatment is determined by the size, location, and number of sialoliths, and the degree of duct obstruction [42]. Surgical treatment is required when conservative treatments, such as sialagogues, hydration, salivary gland massage, warming, antibiotics, and NSAIDs, fail and sialoadenectomy can be performed when necessary [23,24,43]. A sialolith located distal to the duct can be removed easily through an intraoral approach. When a sialolith is located in the proximal portion of the duct or in the parenchyma of the salivary gland, sialoadenectomy may be performed concurrently with sialolith removal, and submandibular gland excision may be performed through an extraoral approach [44]. Sialendoscopy, sialolithotripsy, and laser treatment have been introduced as noninvasive methods for the removal of sialoliths. In the cases reported here, all of the sialoliths were removed by transoral sialolithotomy, as conservative approaches were not considered effective. Although not included in the present case report, sialoliths were observed near the submandibular duct orifice in two children during clinical examination, and conservative treatments, such as hydration and the consumption of lemon candy, were applied. At the next visits, the sialoliths were not observed. To maintain the patency of the duct after surgery, insertion of a cut-down tube has been recommended, but no criteria have been established [45]. Insertion of a cut-down tube may reduce the need for additional procedures, such as reoperation, in cases with edema, abscess, or possible duct stenosis [44]. However, in recent years, satisfactory results have been obtained without insertion of a cut-down tube in several cases. Although the duct is unlikely to be obstructed, due to persistent saliva flow, in cases with long-term sustained duct inflammation, stimulation of the duct wall by the sialolith, and poor incision and suturing of the duct wall during removal of the sialolith may result in fibrotic stenosis [45,46]. In the 1st case reported here, we placed a polyethylene tube because the margin of the incision was not clear and we were concerned about stenosis of the duct. However, frequent dropouts occurred due to the poor coordination of the pediatric patient. In the remaining cases, polyethylene tube insertion and suturing were not applied, but healing was good and normal saliva flow was observed at regular follow-up visits. When a sialolith is located at the orifice and inflammation at the incision site is absent, the requirement for polyethylene tube insertion or suturing should be considered carefully.

In three pediatric patients who visited the hospital clinic with rare sialolithiasis, surgical treatment was performed using an intraoral approach to remove the sialoliths, yielding successful results. The sialoliths were removed through a minimal incision in the duct mucosa above the sialoliths under infiltration anesthesia. All three cases showed good healing of the surgical sites, without duct obstruction. In the 1st case, a cut-down tube was inserted due to the possibility of duct stenosis because the incision line was not clear. However, the child was unable to tolerate the discomfort of the cut-down tube, which caused the boy’s tongue to move constantly, resulting in repeated dropouts. In the remaining two cases, no suturing or cut-down tube insertion was performed, and good healing results were obtained with no complication such as duct stenosis. In pediatric patients, insertion of a cut-down tube is not essential because most sialoliths are located in the submandibular duct opening and the patients do not tolerate tube insertion well. In addition, suturing is not necessary unless the duct is inflamed due to persistent salivary flow.

ACKNOWLEDGEMENTS

This study was supported by research fund from Chosun University Dental Hospital, 2019.

CONFLICTS OF INTEREST

The authors declare that they have no competing interests.

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