PEDIATRIC DENTAL JOURNAL 15(1): 143–146, 2005 143

Case Report Large sialolith in the of a child

Ikuri Konishi*1,2, Satoshi Fukumoto*2, Aya Yamada*2, Kazuaki Nonaka*2 and Taku Fujiwara*1

*1 Division of Pediatric Dentistry, Department of Developmental and Reconstructive Medicine, Nagasaki University Graduate School of Biomedical Sciences 1-7-1 Sakamoto, Nagasaki 852-8588, JAPAN *2 Section of Pediatric Dentistry, Division of Oral Health, Growth and Development, Faculty of Dental Science, Kyushu University 3-1-1 Maidashi, Higashi-ku, Fukuoka 812-8582, JAPAN

Abstract is a disorder encountered by oral surgeons that is Key words rarely seen in children, although it is rather common in adults. Most sialolith Child, found in children are smaller than 5 mm in diameter, and the majority of Sialolithiasis, reported cases have been treated by surgically. We report a 9-year-old boy with Submandibular gland mm, which had developed in Wharton’s 3ן3.5ןa sialolith that measured 12 duct and was then spontaneously passed.

2,3) Introduction than 5 mm in children . Children who present with sialolithiasis are A sialolith is a calcareous concretion that may generally healthy and without systemic illness, occur in the ducts of the major or minor salivary except for symptoms of acute inflammation. Most glands, or within the glands themselves. The of their chief complaints regard intermittent and submandibular gland is most commonly affected, unilateral pain and swelling in the submandibular in most cases, the calculus is found close to the region, usually associated with eating. orifice or in the anterior two-thirds of Wharton’s We describe here a case of submandibular gland duct. Formation of a sialolith is considered to be the sialolithiasis in a 9-year-old child, which resulted in result of calcium salt around a central nidus, which the formation of a calculus greater than 10mm in may consist of desquamated epithelial cells, bacterial diameter that was located in Wharton’s duct on the or micro-organismal decomposition products, or right side. foreign bodies. The high incidence of submandibular calculi in this gland has been explained by the Case report high concentration of calcium salts, pH, and mucin content located there1). Although sialolithiasis is not A 9-year-old boy was referred to our hospital with uncommon in adults, only about, 3% of cases have the chief complaints of tenderness and swelling been reported in children. This low frequency of of the floor of the mouth with spontaneous pain, sialolithiasis in young patients is due to the long which had occurred over a period of 3 days. The period required for sialolith formation, faster salivary patient was first seen by a general practitioner, flow rate, lower concentrations of calcium and who made a diagnosis of acute sialodochitis, and phosphate in saliva, and smaller orifice size for prescribed a 2-day regimen of an antibiotic (cefotiam foreign body entrance. In adults, average calculus dihydrochloride). The past medical history of the size is 6.3 mm (range, 2–30 mm), but most are less patient was unremarkable. Physical examination revealed no acute distress, and body temperature, Received on September 29, 2004 pulse, and blood pressure findings were within normal Accepted on January 7, 2005 ranges. Clinical examination revealed swelling in

143 144 Konishi, I., Fukumoto, S., Yamada, A. et al.

Fig. 1 Diffuse swelling in the area of the right submandibular Fig. 2 Image of mouth floor showing edema, swelling, pus gland discharge, and erythema of Wharton’s duct orifice

Fig. 3 Occlusal view showing the sialolith in the anterior Fig. 4 The calculus following passage from Wharton’s duct portion of Wharton’s duct on the right side

the right submandibular region, and diffuse swelling and erythema of the orifice (Fig. 2). Radiographic of the right submandibular gland was palpable with examination revealed a radiopaque mass in the associated tenderness (Fig. 1). In addition, the right anterior one-third of Wharton’s duct, close to the submandibular nodes were palpable and about the orifice (Fig. 3). A diagnosis of submandibular size of the little finger, and showed mobility and acute sialodochitis caused by a sialolith was made. significant pain. was not present. Intraoral One day after beginning the prescribed antibiotics, examination revealed that the left-side Wharton’s a sialolith spontaneously migrated from the gland duct was normal, with clear salivary flow produced (Fig. 4). The calculus was an ash gray-colored oval, by gentle manipulation of the gland. In contrast, and measured approximately 12mm long and 3.5mm Wharton’s duct on the right side exhibited a in diameter. prominent edema, with swelling, discharge of pus, Physicochemical chemical analysis revealed the LARGE SIALOLITH IN A CHILD 145 presence of 67% calcium phosphate, 28% protein calcium carbonate may provide evidence that this and 5% calcium carbonate. The patient had an sialolith was made of saliva, but not other hard uncomplicated recovery and was released 1 day after tissue or foreign material. Approximately 40% of the migration of the calculus. He was asymptomatic, all submandibular stones are found in the distal and a subsequent follow-up examination 1 month portion of Wharton’s duct or at the orifice, and later revealed the disappearance of submandibular can be removed by simple intraoral procedures swelling. Four months later, there were no signs or performed under local anesthesia. For calculi that symptoms of recurrence. lie in the proximal duct or gland, the treatment of choice has been sialoadenectomy, which is effective Discussion in eradicating symptoms, but carries the risk of nerve injury. Recently, several new minimally invasive Although sialolithiasis accounts for 50% of the techniques have been introduced for the treatment of major diseases localized to the head sialolithiasis, such as extracorporeal sonography and and neck region, individual reports of management of intracorporeal endoscopically controlled lithotripsy, pediatric patients with sialoadenitis or sialolithiasis which have completely changed therapeutic methods are limited. In children, 80% to 90% of cases are utilized7,8). In the case of parotid duct stones, the found in the submandibular gland, compared with long-term outcome with extracorporeal lithotripsy 5% to 10% in the , and approximately has been quite satisfactory, with 50% of all patients 5% in the sublingual and other minor salivary reported to be free of stones and 80% free of glands3–6). The submandibular gland salivary outflow symptoms9). In comparison, fewer than 30% of includes large amounts of calcium and phosphorus, patients who suffer from sialolithiasis of the sub- compared with that of the parotid gland, and includes mandibular gland and receive lithotripsy treatment mucin, which has a high viscosity. Further, the are reported to be free of stones8,10–12). Therefore, the submandibular duct has an opening on the floor of benefits of a minimally invasive technique, compared the month that can easily retain saliva, and Wharton’s to those of other moderately invasive or other duct is longer than other sublingual ducts. These invasive surgical and gland-preserving techniques, characteristics may account for the preponderance must be considered for these patients. of reports of occurrence in this gland. Various techniques of sialodochotomy have Most sialoliths previously reported were removed been described in the literature, with the major using a surgical procedure, while in a few cases they point of concern being the risk of injury to the spontaneously migrated out. In pediatric patients lingual nerve, which passes in close proximity to with sialolithiasis, it is easy for the sialolith to be Wharton’s duct13–17). Surgical excision of the gland is passed from the duct, since most are located near the recommended in cases with extreme proximal stone orifice of Wharton’s duct, salivary flow is faster than localization, due to the anatomical circumstances that from other salivary glands, maturation of the and the assumption that the submandibular salivary sialolith is generally poor, the tissues surrounding gland will not tend to exhibit improvement after the ducts are soft, the size of the sialolith is usually years of obstruction and recurrent inflammation14,18). less than 5 mm in size, and swelling and pain are Diagnosis is very important for correct selection milder than in adults. For these reasons, the first of treatment of sialolithiasis in children. As in most choice of treatment for pediatric sialolithiasis in the cases with spontaneous passage of a sialolith, in submandibular gland may be to wait for spontaneous the present case the stone was localized in anterior migration. In fact, a sialolith with a diameter greater potion of Wharton’s duct, and its passage did not than 10 mm in size passed from the duct 1 day depend on its size. Preservation of gland function after starting antibiotic treatment observed in the in conjunction with low-level risk and minimal present case, to our knowledge, is the largest. The discomfort for the patient should be the primary sialolith is generally containing calcium phosphate, objectives of treatment of sialolithiasis. Apart from protein, and calcium carbonate. In adult cases, the problems such as scar formation, disturbances of ratio of calcium phosphate is high compared with skin sensation, and injury to the gustatory nerves, pediatric cases. In present case, the composition transient functional disturbances of the marginal of sialolith is approximately same as the previous branch of the facial nerve are most often encountered report in child. Additionally, the concentration of in up to 12.5% of open gland excisions. Further, 146 Konishi, I., Fukumoto, S., Yamada, A. et al. permanent lesions have been reported in 7% of 5) Yoel, J.: Pathology and Surgery of the Salivary Glands. these cases19,20). In addition, unilateral excision of Charles C. Thomas, Springfield, 1975, pp.364–383, 854–901, 1148. the submandibular gland also leads to substantial 6) Mela, F., Berrone, S. and Giordano, M.: Considera- reduction of nonstimulated flow of saliva, which zioni clinicostatistiche sulla scialolitiasi sottoman- may significantly affect oral hygiene, risk of caries, dibolare. Minerva Stomatol 35: 571–573, 1986. and the development of xerostomia21). Conservative 7) Zenk, J., Benzel, W. and Iro, H.: New modalities treatment should therefore be selected for sialo- in the management of human sialolithiasis. Minim lithiasis in children. invasive Ther 3: 275–284, 1984. 8) Iro, H., Schneider, H.T., Fodra, C. et al.: Shockwave A few reports have noted submandibular calculi lithotripsy of salivary duct stones. Lancet 339: 1333– that passed out of the duct spontaneously, 1–2 1336, 1992. days following stimulation. These were successful 9) Iro, H., Zenk, J., Waldfahrer, F., Benzel, W., Schneider, results of initial management protocols that included T. and Ell, C.: Extracorporeal shock wave lithotripsy instructing the child to suck on a sour lemon or of parotid stones: results of a prospective clinical trial. Ann Otol Rhinol Laryngol 107: 860–864, 1998. orange candy to stimulate salivary flow. In adults 10) Ottaviani, F., Capaccio, P., Campi, M. and Ottaviani, with long-standing stones, exfoliation of the calculus A.: Extracorporeal electromagnetic shockwave litho- through a perforation of the overlying mucosa has tripsy for salivary gland stones. Laryngoscope 106: been reported, but not a spontaneous passage through 761–764, 1996. the duct. 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