| Abstract|| |
Sialolithiasis is often observed in the oral region, and is caused by the development of a calculus in the salivary gland or duct. This disease is mostly seen in adults or young adults, and seldom develops in children. Of all the cases of sialolithiasis, only 3% are seen in the pediatric population. The clinical presentation typically consists of a painful swelling of the involved salivary gland at meal times, as the obstruction is most acute at this time. The clinical signs often lead to an easy diagnosis. The salivary gland most commonly affected is the submandibular gland. In this paper, we have reported the case of a 10 year old female with sialolithiasis of the left submandibular duct. The treatment consisted of the use of lemon and orange drop candies, which stimulated the salivary flow and in turn resulted in the expulsion of stone.
Keywords: Children, salivary gland, sialolith, sialolithiasis, submandibular gland
|How to cite this article:|
Marwaha M, Nanda KS. Sialolithiasis in a 10 year old child. Indian J Dent Res 2012;23:546-9
Sialolithiasis is the most common cause of salivary gland obstruction, and is found in approximately 65% of the patients with chronic sialadenitis. Sialoliths are calcified structures that develop within the salivary ductal system. These are usually hard formations and maybe round or oval in shape and may have a variety of sizes.  80-90% of the salivary gland and duct calculi are found in the submandibular gland, 5-10% in the parotid gland and approximately 0-5% in the sublingual and other minor salivary glands.  The obstruction can be complete or partial, and may exhibit recurrence once removed. The incidence of sialolithiasis is shown to peak in the third to sixth decade of life.  Salivary calculi in the paediatric population comprise only 3% of all cases of sialolithiasis.  The presence of bilateral calculi in children is rare. Most calculi are relatively small (<1 cm, 93.1%) in children and located in the distal duct (62%). 
Submandibular gland sialolithiasis is more common because of the anatomical factors associated with formation of sialoliths in this gland. The Wharton's duct of the submandibular gland is the longest duct among all salivary gland ducts with the path of the duct going in an upward direction (antigravity flow). Also, the main portion of duct is wider than its orifice. Along with these anatomical factors,the submandibular gland saliva is alkaline in nature and rich in mucin, which can promote the formation of a sialolith. 20% of submandibular stones are not radiopaque, and sialography may be required to locate them.  The aim of this article is to report a case of sialolithiasis of the submandibular gland in a 10 year old girl, which was not detected in conventional radiograph, but was seen in an ultrasonography. A full diagnostic work up with the help of various radiological and histopathological techniques is required before making a confirmed diagnosis.
| Case Report|| |
A 10 year old female child presented to the department complaining of pain in the left lower molar tooth, and a swelling in the sublingual region from the past five months. She complained of mild pain which increased during meal times with dryness of the mouth since 2 months. She had already taken antibiotic therapy. Antibiotic prescribed was Tab Amoxycillin 250 mg TDS for 5 days; however, there was no improvement in the intensity of pain and size of swelling. Extra oral examination revealed a swelling in the sublingual region around 1.5 × 1 cm in size, which was soft on palpation and did not appear to be fixed to any underlying structures [Figure 1]. Intraoral examination revealed a stage of mixed dentition and fair oral hygiene. Caries were seen in 16, 26, 75, 36, 85 and 46 and fluorosis present in 11 and 21. Panoramic radiograph revealed unerupted 13, 15, 23, 24, 25, 35and 45, and missing 31, 32, 33, 41, 42and 43 [Figure 2]. Occlusal radiograph suggested the absence of any salivary calculus [Figure 3]. Intra oral examination revealed scanty salivary flow from the duct of the left submandibular gland. Ultrasonography was carried out in the pateient, which revealed a salivary calculus at the terminal end of the submandibular gland. Duct dilatation was also observed on the scan.[Figure 4].
|Figure 1: Extraoral swelling in sublingual region around 1.5 × 1 cm in size.|
Click here to view
|Figure 2: Panoramic radiograph revealing unerupted 13, 15, 23, 24, 25, 35, 45 and missing 31, 32, 33, 41, 42, 43.|
Click here to view
|Figure 3: Occlusal radiograph suggesting the absence of any salivary calculus.|
Click here to view
|Figure 4: An ultrasound showing the submandibular gland, a dilated duct and stone at the terminal end of the duct.|
Click here to view
A computed tomography (CT) and magnetic resonance imaging (MRI) were not possible in our steup, and the patient could not afford in in other laboratories, and hence was not carried out. Hematological investigations listed in [Table 1] show an increased erythrocyte sedimentation rate (ESR) and total lymphocyte count (TLC). Hepatitis C virus and human immunodeficiency virus (HIV) tests were negative. Patient was referred to the department of Oral Pathology for fine needle aspiration cytology (FNAC) and biopsy examination. FNAC showed an insignificant polymorphic population of lymphoid cells along with few macrophages. The histopathological examination showed a salivary parenchyma separated by connective tissue septae and exhibiting salivary acini and ducts [Figure 5]. Tissue section also revealed an infiltration of inflammatory cells, mainly the plasma cells and lymphocytes with atrophy of the acini and ductal dilation [Figure 6]. A diagnosis of sialolithiasis was rendered following confirmation by histopathology. Majority of the reported cases of sialolithiasis in children have been treated surgically, and only few authors have recommended the use of lemon or orange drop candies to increase the salivary flow rate resulting in expulsion of stone.  In the present case, we instructed the patient to use lemon or orange drop candy to stimulate salivary flow and also to apply moist heat to the left submandibular region. A follow-up appointment was given after every 5 days. On the 8 th day, the patient started feeling slight discomfort, and after 15 days, the stone was expelled from the salivary duct. The patient is still undergoing follow up at our department.
|Figure 5: Histopathological examination showed a salivary parenchyma separated by connective tissue septa and exhibiting salivary acini and ducts. H & E Staining ; Magnification 4×.|
Click here to view
|Figure 6: Tissue section showing infiltration of inflammatory cells,mainly plasma cells and lymphocytes with atrophy of acini and ductal dilation. H & E Staining; Magnification 10×|
Click here to view
|Table 1: Haematological investigations and their results carried out in the patient in the case study|
Click here to view
| Discussion|| |
Sialolithiasis is a condition that refers to the formation of calculi, more commonly known as stones, in the salivary gland or duct.  Sialoliths are usually composed of round organic cores which are intensely calcified, and surrounded by alternative layers of organic and inorganic substances.  The presence of a sialolith is characterized by a series of symptoms. The most characteristic symptom is the swelling of the salivary duct, usually at meal times or even without any stimulating factors. This symptom lasts for a relatively short period, not more than 2 hours,and disappears throughout the day. The swelling may be accompanied by pain, and the patient may present with an episode of salivary colic, which is an acute, lacerating, short duration pain which disappears after 15 or 20 minutes of its initial manifestation. 
In the present case, the patient complained of an intermittent and unilateral pain associated swelling in the region of the left submandibular salivary gland. Also, the symptoms were associated with eating. Bodner L, Fliss D (1995)  reported that only about 3% of cases of all cases of sialolithiasis are seen in children. The low incidence in children may be due to a relatively long time required for the formation of a salivary calculus, and also due to the fact that the sublingual papillae and cuts of the salivary glands are very small in children, making it difficult for foreign material to enter. Furthermore, a calculus is formed more easily in adults than in children as the concentration of calcium and phosphate ions in the resting saliva increases with age. Many theories have been postulated; however, the exact mechanism of calculus formation is yet unknown. In our literature search,  we found that the youngest patient reported was 13 months old, the oldest was 15 years old. The number of patients suffering from sialolithiasis and aged 10 years was largest (5 cases, 17%), followed by 4 patients aged 7 years (13%). Here, we have reported the case of a 10 year old female child with sialolithiasis of the left submandibular duct. In the present case, we have observed the presence of the calculus at the terminal end of duct, which may be due to a small calculus size, allowing it to move toward the sublingual papilla through the duct. For the diagnosis of sialolithiasis, radiographic examination is quite helpful, and an approximate detection rate of 90% for sialolithiasis in the submandibular gland has been reported. 
Panoramic and mandibular occlusal radiograph often reveal radiopaque calculus; however, approximately 20% of sialolithiasis cases in children show radiographic translucency,  as also seen in our case. This was probably because of the relatively small size of the salivary calculus, as well as an insufficient calcification process.  Other examinations such as sialography, scintigraphy, sono radiography, xeroradiography, endoscopy, CT, MRI can also be performed to detect or locate the calculi. Sialography is useful in patients showing signs of sialadenitis related to radiolucent stones or deep submandibular stones; however, it is contraindicated in acute infections or in patients with contrast medium allergy.  After clinical examination, ultrasonography should be the diagnostic method of choice. This is because it is uncomplicated, effective and inexpensive. In the present case, ultrasonography revealed a stone at the terminal end of submandibular gland and a dilated duct.
Along with radiographic and ultrasonography examinations, it is equally important to carefully perform manual palpation in the lower intraoral region for the possibility of detecting a calculus. This helps to enhance the effectiveness of diagnosis, since most of the times, the calculus is located in the submandibular duct itself. The treatment of submandibular sialolithiasis is by the surgical removal of the calculus or the complete excision of the gland. However, initial management consists of antibiotic therapy to reduce or eliminate the acute infection. The drug of choice is penicillin and for children allergic to penicillin, erythromycin or clindamycin can be used effectively as an alternate drug. In our case, we also instructed the child to suck on sour lemon or a lemon/orange candy to the stimulate salivary flow.  The calculus can be removed intraorally in many cases; however, in some cases the gland itself may require excision. Surgical management depends on the location of the sialolith in relation to the salivary gland and its duct. Other means of calculus removal are extracorporeal shockwave lithotripsy and endoscopic intracorporeal shockwave lithotripsy. In the present case, we instructed the patient to use lemon or orange drop candy to stimulate salivary flow, and also to apply moist heat to the left submandibular region. A follow-up appointment was given every 5 days. On the 8 th day, the patient felt slight discomfort and after 15 days, it resulted in the expulsion of the stone from the affected salivary gland.
| References|| |
|1.||Tandler B. Electron microscopical observations on early sialoliths in a human submaxillary gland. Arch Oral Biol 1965;10:509-22. |
|2.||Bonder L. Salivary gland calculi: Diagnostic imaging and surgical management. Compendium 1993;14:572,574-6. |
|3.||Williams MF. Sialolithiasis. Otolaryngol Clin North Am 1999;32:819-34. |
|4.||Bodner L, Fliss D. Parotid and submandibular calculi in children. Int J Pediatr Otorhinolaryngol 1995;31:35-42. |
|5.||Chung MK, Jeong HS, Ko MH, Cho HJ, Ryu NG, Cho DY, et al. Pediatric sialolithiasis: What is different from adult sialolithiasis? Int J Pediatr Otorhinolaryngol 2007;71:787-91. |
|6.||Blatt IM. Studies in sialolithiasis III. Pathogenesis, diagnosis and treatment. South Med J 1964;57:723-9. |
|7.||Cornell McC, Lee CY, Blaustein DI. Sialolithiasis in an 8-year-old child: Case report. Pediatr Dent 1991;13:231-3. |
|8.||Oka T, Nomura J, Matsumura Y, Yanase S, Nagata T, Uno S, et al. A case of sialolithiasis in a child. J Clin Pediatr Dent 2006;31:139-41. |
|9.||Scott J. The prevalence of consolidated salivary deposits in the small ducts of human submandibular glands. J Oral Pathol 1978;7:28-37. |
|10.||Epker BN. Obstruction and inflammatory diseases of the major salivary glands. Oral Surg Oral Med Oral Pathol 1972;33:2-27. |
Department of Pedodontics and Preventive Dentistry, SGT Dental College and Research Institute, Budhera, Tehsil-Gurgaon, Haryana
Source of Support: None, Conflict of Interest: None
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]