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 Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 22  |  Issue : 2  |  Page : 126-128

Giant submandibular sialolith: A case report and literature review


Natural Guard Hospital Alhasia, Resident Level 5, Dammam Medical Complex, Dammam, Eastern Province, Saudi Arabia

Date of Web Publication11-May-2016

Correspondence Address:
Zeinab AlQudehy
P. O. Box: 508, Dhahran 31311, Saudi Aramco
Saudi Arabia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0971-7749.182277

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  Abstract 


Submandibular sialolithiasis is the most common disease of salivary glands. Giant stone of more than 3 cm is extremely rare with only 17 cases reported in literature. In this short communication, we report a young gentleman, presented with an enlarged tender left submandibular gland and hard mass within Wharton duct. Neck ultrasound revealed left submandibular duct stone with an associated abscess confirmed by computed tomography scan. Medical management followed by left submandibular intraoral stone extraction with marsuplization of the duct 2 weeks later was done. The sialoliths was about 3.6 cm. The patient had smooth recovery with no complications in follow-up.

Keywords: Giant, Salivary glands, Sialolithiasis, Submandibular gland


How to cite this article:
Abdullah O, AlQudehy Z. Giant submandibular sialolith: A case report and literature review. Indian J Otol 2016;22:126-8

How to cite this URL:
Abdullah O, AlQudehy Z. Giant submandibular sialolith: A case report and literature review. Indian J Otol [serial online] 2016 [cited 2019 Nov 15];22:126-8. Available from: http://www.indianjotol.org/text.asp?2016/22/2/126/182277




  Introduction Top


Sialolithiasis is the most common disease of the salivary glands. Sialolith can form in any of the salivary glands in head and neck region, with submandibular gland is the most commonly affected site (80–92%).[1] It has been reported that the stones can form in the parotid gland in 6–20% of cases and in the sublingual and minor salivary glands in 1–2%.[1] Bilateral or multiple-gland sialolithiasis is occurring in fewer than 3% of patients. In cases with multiple stones, calculi may be located in different positions along the salivary duct and gland. Submandibular stone close to the hilum of the gland tends to become large before they become symptomatic. Male adults are more frequently affected than females, and children are rarely involved. Sialolithiasis occurs on right and left sides equally.

Several factors tend to favor submandibular gland stones that include longer submandibular duct and larger duct caliber, with slower salivary flow rate in the submandibular gland compared to the other ducts. In addition to the fact that saliva flows against gravity in the submandibular gland, the presence of more alkaline saliva in the submandibular gland, along with high mucin and calcium contents of the saliva, favor the higher incidence of silolithiasis in the submandibular gland.

Commonly, sialoliths measure from 1 mm to <1 cm in size. Giant salivary gland stones (GSGS) are defined in literature as those stones measuring over 1.5 cm and have been rarely reported in the medical literature.[2],[3] GSGS measuring over 3 cm are extremely rare, with only few reported cases.[4] The aim of this article is to present a case of a giant sialolith in the light of the literature on GSGS.


  Case Report Top


A 37-year-old gentleman presented to our ENT Department on June 2013. The patient was medically free and was complaining of hard swelling in the left submandibular area of about 2 years duration. He had no history of odynophagia, dysphagia, muffled voice, shortness of breath, or recent dental procedure. He gave a history of similar submandibular swelling episode one year ago and apparently the swelling resolved completely with no treatment. The swelling was not related to food intake, with no aggravating or relieving factors and no history of associated pain.

On our initial evaluation, neck examination revealed asymmetry along the left submandibular area. On palpation, swollen area corresponded to the anatomic location of submandibular salivary gland. The swollen area palpated bimanually (extra orally and through the mouth), it was hard and tender. The floor of mouth along the submandibular duct in left side was swollen, with no color changes of the surrounding mucosa. Neck ultrasound showed left submandibular duct stone with an associated abscess confirmed by computed tomography (CT) scan [Figure 1]. Findings on blood and serum biochemistry were within normal limits. Medical management was started by intravenous antibiotic for 10 days, followed by left submandibular intraoral stone extraction with marsuplization of the duct two weeks later under general anesthesia. The calculus was dissected free [Figure 2]. The sialoliths was about 3.6 cm [Figure 3]. The patient had smooth recovery with no complications in follow-up. The symptoms resolved after operation.
Figure 1: Postoperative left submandibular stone, 4 cm size

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Figure 2: Oral examtion shows left submandibular mass

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Figure 3: CT scan head and neck without contrast, axial cut at level of lower mandibular bone shows hyperdense mass in left submandibular area

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  Discussion Top


The GSGS are rare findings in clinical oral pathology, all of them occurring in male patients.[5],[6],[7],[8],[9] The GSGS have been reported both in salivary glands and salivary ducts. Stones larger than 3 cm are rare.[10] In 2002, Bodner reported that only few documented cases have been published in literature.[11] A review of literature in 2007[12] found 16 reported cases of salivary stones having a size up to 35 mm with the largest stone reported measured 55 mm. The ability of a calculus to grow and become a giant depends mostly on the reaction of the affected duct. If the duct adjacent to the sialolith we can able to dilate it, so we allow nearly normal secretion of saliva around the stone by doing that, it might be asymptomatic for a long period till giant calculus will be created when stagnating happen, bacteria accumulate that cause a sialo-oral fistula. The inflammatory debris obstructs the residual narrowed duct lumen, resulting in an exacerbation of the inflammation. The resulting inflammatory process around a large stone may lead to tissue breakdown and spontaneous stone extrusion with developing complication as intraoral fistula formation.

Ultrasonography is one of the standard modalities used to diagnosis submandibular stone. On the other hand, 80.0% of long-standing giant submandibular stones are easily seen as most of them were calcified with time and became radiopaque on standard X-ray films. Only one-fifth of the submandibular stones will be missed radiologically on plain X-rays and mostly found with small stones.[13] The ultrasonography in addition of being an operator-dependent, it does not provide the surgeon with clear and direct anatomic localization of the stone.[14] Although CT scan can pick up both small and large stones depending on the thickness of the cuts, accurate stones' localization is lacking.[15] Sialography conventionally has been considered the gold diagnostic standard and provides a good image of the ductal system.[15] However, sialography carries an increased risk of ductal perforation and retrograde displacement of the stone with an effect of injection. Giant sialoliths of a remarkable size is a diagnostic and therapeutic challenge for the clinician; it needs careful evaluation prior management.[16]

Submandibular stones are typically removed surgically by two ways either intraoral or extraoral approach. The decision of the most appropriate mode of surgical treatment depends on multiple factors, mainly the location of the stone. Intraoral approach is often utilized when the stone is located anterior to the lingual nerve and artery because this method can lead to lingual nerve anesthesia.[16] The anatomical location of the lingual nerve loops around the mid to distal portion of Warthin's duct before it enters the tongue, making the lingual nerves vulnerable for injury. As a general rule, for treating stones that are located entirely in the duct and close to the papillae, intraoral approach is ideal. On the other hand, for treating intraglandular stones and stones embedded within the hilum of the gland, extraoral approach is preferred with excision of the submandibular gland. Newer treatment methods are currently available such as extracorporeal short-wave lithotripsy and interventional sialendoscopy. The newer management options are effective alternatives to conventional surgical excision, especially for smaller stone.[16] Transoral sialolithotomy with sialodochoplasty or sialadenectomy remains the gold standard management technique for giant stones.


  Conclusion Top


Giant submandibular gland stone are rare. There are various methods available for the management of salivary stones, depending on the gland affected and stone location. Asymptomatic giant sialolith of remarkable size may pose both diagnostic and therapeutic challenge for the clinician Newer treatment modalities are effective alternatives to conventional surgical excision for smaller sialoliths. However, for giant sialoliths, transoral sialolithotomy with sialodochoplasty or sialadenectomy remains the mainstay of management.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
McKenna JP, Bostock DJ, McMenamin PG. Sialolithiasis. Am Fam Physician 1987;36:119-25.  Back to cited text no. 1
    
2.
Bodner L. Giant salivary gland calculi: Diagnostic imaging and surgical management. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002;94:320-3.  Back to cited text no. 2
    
3.
Soares EC, Costa FW, Pessoa RM, Bezerra TP. Giant salivary calculus of the submandibular gland. Otolaryngol Head Neck Surg 2009;140:128-9.  Back to cited text no. 3
    
4.
Ledesma-Montes C, Garcés-Ortíz M, Salcido-García JF, Hernández-Flores F, Hernández-Guerrero JC. Giant sialolith: Case report and review of the literature. J Oral Maxillofac Surg 2007;65:128-30.  Back to cited text no. 4
    
5.
Mustard TA. Calculus of unusual size in Wharton's duct. Br Dent J 1945;79:129.  Back to cited text no. 5
    
6.
Cavina C, Santoli A. Some cases of salivary calculi of particular interest. Minerva Stomatol 1965;14:90-5.  Back to cited text no. 6
[PUBMED]    
7.
Raksin SZ, Gould SM, Williams AC. Submandibular duct sialolith of unusual size and shape. J Oral Surg 1975;33:142-5.  Back to cited text no. 7
[PUBMED]    
8.
Tinsley G. An extraordinarily large asymptomatic submandibular salivary calculus. Br Dent J 1989;166:199.  Back to cited text no. 8
    
9.
Leung AK, Choi MC, Wagner GA. Multiple sialoliths and a sialolith of unusual size in the submandibular duct: A case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1999;87:331-3.  Back to cited text no. 9
    
10.
Mandel L, Hatzis G. The role of computerized tomography in the diagnosis and therapy of parotid stones: A case report. J Am Dent Assoc 2000;131:479-82.  Back to cited text no. 10
    
11.
Cawson RA, Odell EW. Neoplastic and non-neoplastic diseases of the salivary glands. In: Essentials of Oral Pathology and Oral Medicine. 6th ed. Edinburgh: Churchill Livingstone; 1998. p. 239-40.  Back to cited text no. 11
    
12.
Cockrell DJ, Rout PG. An adverse reaction following sialography. Dentomaxillofac Radiol 1993;22:41-2.  Back to cited text no. 12
    
13.
Thoma KH, Gorlin RJ, Goldman HM, editors. Thomas' Oral Pathology. St. Louis: Mosby; 1970.  Back to cited text no. 13
    
14.
Paul D, Chauhan SR. Salivary megalith with a sialo-cutaneous and a sialo-oral fistula: A case report. J Laryngol Otol 1995;109:767-9.  Back to cited text no. 14
    
15.
Baurmash HD. Submandibular salivary stones: Current management modalities. J Oral Maxillofac Surg 2004;62:369-78.  Back to cited text no. 15
    
16.
Walvekar RR, Bomeli SR, Carrau RL, Schaitkin B. Combined approach technique for the management of large salivary stones. Laryngoscope 2009;119:1125-9.  Back to cited text no. 16
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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