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	INTRODUCTION
� ��Sialolithiasis is the most common disease of the salivary glands and the major cause of salivary gland dysfunction [1]. It is a disease that may cause partial or total obstruction and has a tendency to recur. Sialolithiasis comprise the majority of salivary gland diseases (over 50% of reported cases,) and is accepted as the most frequent cause of acute or chronic infection of the salivary gland.  More than 80% of the sialoliths occur in the submandibular gland or its duct, 6% in the parotid gland [2]. The incidence of stones in the sublingual gland is much lower, varying from 0�6.4%. While sialoliths can occur in any salivary gland or at any age, they generally develop after the age of 25, with prevalence in adult population of 12/1000 [3]. Many studies reveal that males are more commonly affected than females. Children are very rarely affected. The sialoliths are usually observed as round or ovoid shaped, smooth or rough surfaced, and yellow in color. Intra-ductal stones, which are frequently smaller in size, are more common compared to intra-glandular stones. In the acute phase, the collected saliva forms retrograde pressure in the gland and duct ; thus symptoms of pain and swelling usually increase with salivation during or after eating. Patients with sublingual sialoliths can also experience pain, swelling and/or speech difficulty, but dysphagia is normally present only in extended lesions [4]. It may be palpated in the salivary duct only when it is of considerable size.  
       During acute phase, the advised treatment modality is to use antibiotherapy; than when acute phase passed it becomes to excise surgically the gland. In some situations, the sialolitechtomy can be also performed during acute phase by sharply decreasing the intraglandular pressure after discharging the collected exuda or saliva [5]. 
������ ��The sialoliths are usually composed of inorganic materials like calcium phosphate or carbonate associated with other salts and organic materials like mucopolysaccharides, glycoproteins and cellular residue [6]. 
���� �  As the Wharton duct is longer and with larger caliber and lies against gravity, the saliva, which has high mucin and calcium content, flows with slow rates. This, in addition to the higher alkalinity of the saliva found there, facilitates stone formation in the submandibular gland, but it is rarely seen in the sublingual gland [7]. The sialoliths of the major salivary glands usually measure between 5 and 10 mm in size. Regarding the literature, large sialoliths are rare and most are found in the submandibular gland [8].
����   We review the literature and report a case of a sialolith in the sublingual gland measuring 23 mm in length with review of the various available diagnostic modalities and treatment options.

�CASE REPORT
���   A 41-year-old female with a previous submandibular gland excision due to left submandibular sialolithiasis, described a left side pain related with eating, which began 5-6 months prior to her visit and increased in a stepwise fashion in the postoperative period. The patient began to feel stiffness in tongue movement over the preceding few weeks. There was no mouth dryness or systemic disease in the anamnesis. Upon extra-oral examination, the patient showed a swelling over the submental region. There were no signs of a cutaneous sinus, fistula, or ulceration in the affected region. Per-oral examination and bimanual palpation revealed a large, tender, yellowish and firm mass, which had displaced the tongue laterally, in the left floor of the mouth over the sublingual region. The ductal opening was normal and no discharge was detected, however the sialolith was seen protruding from ductal opening. There was no lymphadenopathy upon physical examination. On the basis of clinical findings, a diagnosis of left sublingual gland sialolith was made.
�� �� This left sublingual gland sialolithiasis was treated with excision of both the left sublingual gland and stone via a transoral approach under general anesthesia (Figure 1). The sialolith recovered measured approximately 23 mm in length. The calculus was oval, tan in color and had a granular surface. It was surrounded by white fibrous tissue. (Figure 2) �The one-year follow-up was unremarkable. No postoperative complications were noted. This study features human subjects, we have read the  HYPERLINK "http://www.wma.net/en/30publications/10policies/b3/index.html" \n _blankHelsinki Declaration and have followed the guidelines in this investigation. We explained the scientific importance of the patient�s disease to her, and she provided consent for publication of her case in scientific literature. 

�DISCUSSION
� ���� �The etiology and pathogenesis of the sialolithiaisis are still unclear [8]. The defects in mucin formation, electrolyte concentration disorder, hyposalivation, increase the viscosity of saliva, ductal dilatation, metabolic disorder, infection and rise in the mucin part of secretion are all contributing factors to the etiology. The pathogenesis of sialolithiasis consists of the stasis of calcium rich saliva. It is thought that the sialoliths are formed as a result of accumulation of the calcium salt around the organic core, which is composed of mucin, bacteria and epithelial cells [9,10].  The stasis, which occurs at intervals, leads to gel formation regarding to alteration in the mucoid content of saliva. As the organic elements and the salts collect over this gel formation, the sialolith develops [10]. 
������ ��Sialolithiasis is a common disease of the major salivary glands and is frequently seen in submandibular gland, but�sublingual sialoliths are very rarely reported in the literature [11]. They are usually small, but multiple sialoliths can combine to form a large mass, and can develop and cause symptoms such as pain, swelling, dysphagia and/or speech difficulty. Complete calcification of the entire gland may be constituted via long-standing sialolithiasis.  Clarence P. et al reported an unusual case of sialolithiasis of the sublingual gland presenting in a 35-year-old female. They removed the right sublingual salivary gland, together with the five stones (the largest measuring 7x6x4 mm) via an intraoral approach under general anesthesia preserving Wharton�s duct and lingual nerve [11]. The case report prepared by Choi JH et al discusses a multiple-stone sialolithiasis of 22 silaoliths with diameters ranging from 1 mm to 5 mm in the sublingual and the minor salivary glands; they were removed using transoral sialolithotomy with sublingual sialadenectomy [12]. Liao LJ et al studied a case of a 50-year-old male with left sublingual gland sialolithiasis who was treated with excision of the left sublingual gland and the stone via a transoral approach 6 weeks after the infection was controlled [13].  Ki Hwan�Hong et al evaluated a case of multiple sialoliths arising in the sublingual gland in a 14-year-old female; the diameter of the largest one was 9 mm [14].  Eyigor H et al �presented a 67-year-old woman who had 268 sialoliths, whose largest diameter was approximately 4 mm, forming a very large mass in the sublingual gland and causing the patient to have dysphagia [15]. G�ng�rm�_ et al reported the largest single sublingual gland calculus in the literature. In this case, the giant sialolith was completely encased in the glandular substance, and the stone was a single calcified mass about 32 mm in diameter [16].
��� ����Sialoliths may consist of a single stone or multiple stones in any major salivary gland, however 70�80% of reported patients present with a single stone sialolith. Lustmann et al indicated that one sialolith was present in 75.3% of cases, two in 15.6%, three in 2.9% and four to eight in 6.2% [9]. In our study, the patient had one single sialolith measuring 23mm in size.
��� �� Sialoliths grow, on average, 1-1.5mm per year; regarding this, �zden et al emphasized that a period of more than 10 years should pass in order to observe a sialolith with 1.5 cm size. They also revealed that can explain how a sialolith can grow intraductally and remain asymptomatic during an extended period of time [9]. Larger sialoliths may cause salivary gland dysfunction and at the same time may be a source of infection, either acute or chronic bacterial infection. Due to the aforementioned reasons, it is recommended that they be excised upon finding. 
������ Salivary gland stones become symptomatic when the growing stone causes increasing obstruction of salivary secretion, which leads to swelling accompanied by trismus and usually associated with eating, pain and infection of the gland [2].  The degree of ductal obstruction, intraductal pressure and the continuation of the saliva flow determine the intensity of pain and swelling in sialolithiasis [4].  In cases where the duct adjacent to the sialolith allows normal secretion of saliva around the stone, the stone can remain asymptomatic for longer periods of time. The sialolith can grow to the point of extending the dilatation capacity of duct, and diffuse edema and hyperemia on the floor of the mouth (along the line of Wharton�s duct) can also be seen and it may result in a sialooral fistula, thus the sialolith may partially protrude into the oral cavity [18,19].  In the case of addition of the secondary infection, fever, erythema and purulent secretion from duct after manual massage can be seen. Our patient described a left side pain, related with eating, which began 5-6 months ago and increased stepwise.
����  A careful anamnesis and physical examination are usually enough to diagnose the sialolithiasis. Sialoliths can be palpated via�bimanual examination of the floor of mouth. If the gland is palpated as fixed and stiff, it may be considered as hypofunctional or nonfunctional. it may result in a sialooral fistula, thus the sialolith may partially protrude into the oral cavity [20].  In our case report, on extraoral examination, the patient showed a swelling over the left submental region. There were no signs of cutaneous sinus, fistula, or ulceration in the affected region. Per-oral examination and bimanual palpation revealed a large, tender, yellowish and firm mass in the left floor of the mouth over the sublingual region.
������  Differential diagnosis of sialolithiasis from other calcified structures beyond the salivary gland should also be made. These include: torus mandibularis, osteoma, calcified lymph nodes, phlebolith and other vascular calcifications, tuberculosis of lymph nodes or salivary glands, calcified atheroma plaques in major blood vessels, and very rarely myositis ossificans and distant calcified neoplasms [21].  
����  The treatment of sialolithiasis varies depending on the location of stone,�duration of symptoms, frequency of the recurrence and size of the sialolith. Some conservative methods like proper hydration of the patient, moist warm heat application and massaging the gland in combination with sialogogues can be performed for treatment of small sialoliths. They can also be milked out through ductal orifices by bimanual palpation [22]. If the secondary infection is present, penicillinase resistant anti-staphylococcal antibiotherapy can be added to the treatment. Whenever the stone can be palpated intraorally, it is best to remove it through an intraoral approach from the duct [22,23]. The duct should be isolated and a longitudinal incision made into the duct (over the stone) to retrieve it.  Sialolith that are located more distally can be taken out after dilatation of the ductal orifice by dilatators or lacrimal probes. The stones located more deep can be excised with transoral approach by performing an incision along ductus. After the stone is taken out, the incision should be left open to permit drainage.�Excision of the salivary gland with sialoliths is indicated when there is a stone of substantial mass within the gland itself that is not surgically accessible. 
���� � The choice of surgical approach to access the sialolith and the consideration for preserving the salivary gland requires careful evaluation when dealing with large sialoliths of a remarkable size [18].  Newer treatment modalities such as extracorporeal shock wave lithotripsy and more recently the use of endoscopic intracorporeal shockwave lithotripsy are effective alternatives to conventional surgical excision for smaller sialoliths. However, for large sialoliths, transoral sialolithotomy with sialodochoplasty or sialadenectomy remains the primary mode of management [24]. The sialolith of our case was also treated with excision of the left sublingual gland and the stone via transoral approach under general anesthesia.

Conclusion
�� ��  In general, sublingual gland sialoliths are considered rare. Patients with sublingual sialolith e��	!	�	�	�	



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