International Journal of Case Reports and Images - IJCRI - Case Reports, Case Series, Case in Images, Clinical Images

     
Case Report
 
Unusually large sialolith of submandibular gland
Haci Taner Bulut
Department of Radiology, Medical Faculty of Adiyaman University, Adiyaman, Turkey.

doi:10.5348/ijcri-2014110-CR-10421

Address correspondence to:
Haci Taner Bulut
MD, Department of Radiology, Medical Faculty of Adiyaman University
Adiyaman
Turkey
Phone: +90 530 207 15 17
Fax: +90 416 227 08 63
Email: taner.bulut02@gmail.com

Access full text article on other devices

  Access PDF of article on other devices

[HTML Abstract]   [PDF Full Text] [Print This Article]
[Similar article in Pumed] [Similar article in Google Scholar]


How to cite this article
Bulut HT. Unusually large sialolith of submandibular gland. Int J Case Rep Images 2014;5(9):625–628.


Abstract
Introduction: Pathologically sialolithiasis is a disease which results in the obstruction of a salivary gland by a sialolith. Sialolith is generally seen in small size and their sizes range from 1 mm to 1 cm. Large salivary gland calculi are infrequent and defined as the size of 1.5 cm or larger. Only a few cases of large sialolith of the submandibular glands have been reported in literature. Imaging methods have an important role in making a diagnosis and in planning further management, operative or otherwise.
Case Report: A 52-year-old male patient with multiple stones in the submandibular gland admitted to our hospital with swelling at submandibular region. Plain films show large stones at the region of submandibular gland. The maximum stone size was 1.8x0.8 mm. Submandibular gland size increased due to the sialoadenitis caused by stones. After surgery, the patient had a nearly normal function of the glands for three months.
Conclusion: The swelling which is seen in the submandibular region most commonly originates from sialolithiasis of submandibular gland, so it should be carefully evaluated by clinicians. Diagnostic imaging methods may complement each other in examining glands with sialolithiasis and may offer a promising diagnostic strategy for treatment and follow-up studies in sialolithiasis.

Keywords: Sialolithiasis, Large sialolith, Submandibular gland, Imaging


Introduction

Sialolithiasis accounts for the most common cause of diseases of salivary glands [1] [2] [3] [4] [5] [6] [7]. Pathologically, sialolithiasis is a disease results in the obstruction of a salivary gland by a sialolith [8] [9]. The clinical symptoms of sialolithiasis are pain and swelling due to enlargement of involved gland [1] [2] [3] [7] [9]. Thoese symptoms help clinicians to diagnose easily. Nonetheless, pain is not seen in all the cases [2] [10]. About 80% of all reported cases of sialolith occur in the submandibular gland [1] [2] [3][6][9]. The sublingual and minor salivary glands are seldom involved [1] [2] [3] [4] [5][7][9]. Sialolithiasis is generally found between 30 and 60 years of age and it has a frequent prevalence in male patients [9] [11]. Sialoliths are generally seen in small size and their sizes range from 1 mm to 1 cm. Large salivary gland sialolith which is large 15 mm are considered rare [7] [9]. Only a few cases of large sialolith of the submandibular or parotid glands have been reported in literature. Imaging methods have an important role in making a diagnosis and in planning management. Plain radiography and sialography, magnetic resonance imaging (MRI) scan, computed tomography (CT) scan, and ultrasound all have a role [12] . The aim of this study is to evaluate the clinical and radiographical findings of the patient with large sialolith of submandibular gland.


Case Report

A 52-year-old male patient admitted to our hospital with painless swelling at submandibular region. Extra-oral examination revealed swelling and palpable mass. In intraoral examination, bimanual palpation revealed a hard elongated mass and multiple stones in a large size. Plain film, axial CT, and MRI scans were obtained for radiological examination. Stone location, shape, and size were estimated on plain film, MRI and CT scans. Posterior-anterior and sagittal plain films show large calculi at the region of submandibular gland (Figure 1). Non-enhanced axial CT-scan showed large hyperdense masses (sialoliths), localized within the left Wharton duct and enlargement of the affected submandibular gland. The maximum stone size was 1.8x0.8 mm, as measured directly on the axial CT scan (Figure 2). Submandibular gland size increased due to the sialadenitis caused by stones. Due to sialadenitis, coronal Short tau inversion recovery (STIR) and enhanced axial T1-weighted magnetic resonance images of affected gland showed higher signal intensity compared with normal gland on right side (Figure 3). Sialoliths removed with surgery. After surgery, the patient had a nearly normal function of the glands for three months.


Cursor on image to zoom/Click text to open image
Figure 1: A 52-year-old male with left submandibular sialolithiasis. Posterior-anterior (A) Sagittal, (B) Plain films showing large stones at the region of submandibular gland.



Cursor on image to zoom/Click text to open image
Figure 2: (A, B) Non-enhanced axial computed tomography scan showing sialoliths and enlargement of the affected submandibular gland.



Cursor on image to zoom/Click text to open image
Figure 3: Coronal short tau inversion recovery (STIR) (A) Enhanced T1-weighted, (B) Magnetic resonance images of affected gland showing higher signal intensity compared with normal gland on right side.



Discussion

The most widespread illnesses of the salivary gland are sialoliths [1] [3][9][13]. Sialoliths are generally seen in small size and their sizes range from 1 mm to 1 cm [1] [2] [3] [5][9]. The mean size of sialoliths is reported as 6 to 9 mm. They infrequently measure more than 1.5 cm. Large salivary gland calculi are infrequent and defined as the size of 1.5 cm or larger [2] [9] [10] [11]. Most of the studies have conducted that the common symptoms of sialoliths are recurrent pain and swelling of the associated gland, because sialoliths generally does not block the flow of saliva fully [1] [2] [3]. Nonetheless, large sialoliths have been frequently reported in the body of salivary glands, they have infrequently been described in the salivary ducts, particularly without any complaints from the patients [2] [4][14]. In this study, clinical and radiological features of one case which have large sialoliths in the size of 1.8 cm were presented. The sialoliths were located into Wharton ducts and the patient complained painless swelling. Some uncommon large salivary stones may be noticed unless the patient has a long history, due to the fact that lesions are usually asymptomatic. It is conducted that the stones may expand in the proportion of about 1 to 1.5 mm each year [2]. Hence, it is possible to presume that sialoliths of our case began to develop many years ago.

In the diagnosis of sialoliths, history and careful examination come to the fore. Pain and swelling of involved gland at the time of meal are of great importance. Bimanual examination in the floor of the mouth may show a palpable stone in a great number of cases of submandibular sialoliths. Bimanual palpation of the gland is very useful because a uniformly solid and hard gland indicates a hypo-functional or non-functional gland [1] [2][8]. A case in the this study has a history of painless swelling in the floor of the mouth at mealtimes. Extra-oral examination revealed swelling and palpable mass. In intraoral examination, bimanual palpation revealed a hard elongated mass and multiple stones in a large size.

In the diagnoses of sialolithiasis, imaging methods are very useful. Plain radiographs are useful in showing radiopaque stones. It is very uncommon for patients to have a combination of radiopaque and radiolucent stones and 40% of parotid stones may be radiolucent [15]. Sialography is thus useful in patients showing signs of sialadenitis related to radiolucent stones or deep submandibular stones. Sialography is, however, contraindicated in acute infection or in significant patient contrast allergy. Nowadays, magnetic resonance sialography (MR sialography) imaging is recommended in diagnosis of sialoliths, but this method is not appropriate to see the inner duct of the salivary glands. Developed in the 1990's as an endoscopic method, sialoendoscopy technique enables clinicians to examine the ductal system completely and it can be used not only for diagnosis but also for treatment [16]. The CT scan is useful in identifying small calculi within the salivary gland or duct. It can also show localization and number of stones in the gland and measure size of stones. Contrast-enhanced CT scan has a potential to show enlargement of gland due to sialadenitis. Features of the submandibular glands affected by sialolithiasis can well evaluate with MRI scan [12] . Hence, it can possible to differentiate, acute or chronic stage of sialadenitis with MRI scan. It can also show the location of stones and shapes of ducts. In this context, MRI scan using T1-weighted and STIR sequences, can provide effective information about the pathologic status of the gland parenchyma affected by sialolithiasis [12]. Moreover, the extent, acute and chronic nature of this obstruction may reflect by MRI findings of the gland parenchyma [12]. In this study, the sialoliths were observed clearly in plain radiographs, but estimation of size and location of stones is limited. The MRI scan and CT scan were suitable in precise preoperative estimation of stone's size and location. These results suggest that MRI features may reflect acute obstruction, and a combination of CT and MRI scans in examining glands with sialolithiasis may offer a promising diagnostic strategy for treatment and follow-up studies in sialolithiasis.

Different treatment options may be selected according to the size and location of the sialolith. The treatment of choice of small sialolith should be medical instead of surgical. However, if the stone is too large or located in the proximal of duct, piezoelectric extracorporal shock wave lithotripsy or surgical removal of the stone or gland may be required [1] [2] [3]. Sialoendoscopy is a new way and minimally invasive technique for treating obstructions of the ductal system and can be used with operation in large salivary stones [17]. Recurrent or continuous obstruction of the salivary duct may lead to acute or chronic sialadenitis or even to the perforation of the oral mucosa [18]. In this case, the sialoliths were removed by surgical excision. The clinicians should evaluate carefully the painful or painless swellings in submandibular area. This condition seems to be the most common disease in submandibular gland and Wharton duct due to the presence of gland lithias. Large submandibular sialoliths should be treated by appropriate approach to avoid possible severe postoperative complications.


Conclusion

The swelling which is seen in the submandibular region most commonly originates from sialolithiasis of submandibular gland, so it should be carefully evaluated by clinicians. Diagnostic imaging methods may complement each other in examining glands with sialolithiasis and may offer a promising diagnostic strategy for treatment and follow-up studies in sialolithiasis.


References
  1. Siddiqui SJ. Sialolithiasis: An unusually large submandibular salivary stone. Br Dent J 2002;193(2):89–1.   [CrossRef]   [Pubmed]    Back to citation no. 1
  2. Alkurt MT, Peker I. Unusually large submandibular sialoliths: Report of two cases. Eur J Dent 2009;3(2):135–9.   [Pubmed]    Back to citation no. 2
  3. Iqbal A, Gupta AK, Natu SS, Gupta AK. Unusually large sialolith of Wharton's duct. Ann Maxillofac Surg 2012;2(1):70–3.   [CrossRef]   [Pubmed]    Back to citation no. 3
  4. 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(3):331–3.   [Pubmed]    Back to citation no. 4
  5. Parkar MI, Vora MM, Bhanushali DH. A Large Sialolith Perforating the Wharton's Duct: Review of Literature and a Case Report. J Maxillofac Oral Surg 2012;11(4):477–82.   [CrossRef]   [Pubmed]    Back to citation no. 5
  6. Singhal A, Singhal P, Ram R, Gupta R. Self-exfoliation of large submandibular stone-report of two cases. Contemp Clin Dent 2012;3(Suppl 2):S185–7.   [CrossRef]   [Pubmed]    Back to citation no. 6
  7. Gupta A, Rattan D, Gupta R. Giant sialoliths of submandibular gland duct: Report of two cases with unusual shape. Contemp Clin Dent 2013;4(1):78–80.   [CrossRef]   [Pubmed]    Back to citation no. 7
  8. 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(1):128–30.   [CrossRef]   [Pubmed]    Back to citation no. 8
  9. Oteri G, Procopio RM, Cicciu M. Giant Salivary Gland Calculi (GSGC): Report Of Two Cases. Open Dent J 2011;5:90–5.   [CrossRef]   [Pubmed]    Back to citation no. 9
  10. Lustmann J, Regev E, Melamed Y. Sialolithiasis. A survey on 245 patients and a review of the literature. Int J Oral Maxillofac Surg 1990;19(3):135–8.   [Pubmed]    Back to citation no. 10
  11. Bodner L. Giant salivary gland calculi: Diagnostic imaging and surgical management. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002;94(3):320–3.   [CrossRef]   [Pubmed]    Back to citation no. 11
  12. Sumi M, Izumi M, Yonetsu K, Nakamura T. The MR imaging assessment of submandibular gland sialoadenitis secondary to sialolithiasis: Correlation with CT and histopathologic findings. AJNR Am J Neuroradiol 1999;20(9):1737–43.   [Pubmed]    Back to citation no. 12
  13. Akin I, Esmer N. A submandibular sialolith of unusual size: A case report. J Otolaryngol 1991;20(2):123–5.   [Pubmed]    Back to citation no. 13
  14. Graziani F, Vano M, Cei S, Tartaro G, Mario G. Unusual asymptomatic giant sialolith of the submandibular gland: A clinical report. J Craniofac Surg 2006;17(3):549–2.   [CrossRef]   [Pubmed]    Back to citation no. 14
  15. Isacsson G, Isberg A, Haverling M, Lundquist PG. Salivary calculi and chronic sialoadenitis of the submandibular gland: A radiographic and histologic study. Oral Surg Oral Med Oral Pathol 1984;58(5):622–7.   [CrossRef]   [Pubmed]    Back to citation no. 15
  16. Becker M, Marchal F, Becker CD, et al. Sialolithiasis and salivary ductal stenosis: Diagnostic accuracy of MR sialography with a three-dimensional extended-phase conjugate-symmetry rapid spin-echo sequence. Radiology 2000;217(2):347–58.   [Pubmed]    Back to citation no. 16
  17. Yu CQ, Yang C, Zheng LY, Wu DM, Zhang J, Yun B. Selective management of obstructive submandibular sialadenitis. Br J Oral Maxillofac Surg 2008;46(1):46–9.   [CrossRef]   [Pubmed]    Back to citation no. 17
  18. Sutay S, Erdag TK, Ikiz AO, Guneri EA. Large submandibular gland calculus with perforation of the floor of the mouth. Otolaryngol Head Neck Surg 2003;128(4):587–8.   [CrossRef]   [Pubmed]    Back to citation no. 18

[HTML Abstract]   [PDF Full Text]

Author Contributions
Haci Taner Bulut – Substantial contributions to conception and design, Acquisition of data, Analysis and interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published
Guarantor of submission
The corresponding author is the guarantor of submission.
Source of support
None
Conflict of interest
Authors declare no conflict of interest.
Copyright
© 2014 Haci Taner Bulut. This article is distributed under the terms of Creative Commons Attribution License which permits unrestricted use, distribution and reproduction in any medium provided the original author(s) and original publisher are properly credited. Please see the copyright policy on the journal website for more information.



About The Author

Haci Taner Bulut is Medical Faculty in Department of Radiology, Adiyaman University, Adiyaman, Turkey. He has published many research papers in national and international academic journals. His research interests include neuroradiology, MR imaging. He/she intends to pursue Postdoocs in future.




  Home line About IJCRI line Aim and Scope line Sections line Open Access line Archives
Apply as Editor line Apply as Reviewer line Submit Reviews - Editors line Submit Reviews - Reviewers
Instructions for Authors line Templates to Use line Copyright Form line Author Checklist
Online Submission line Email Submission line Submit Revision line Submit All Forms line Submit Page Proofs
Terms of Service line Privacy policy line Disclaimer line FAQ line Contact: Journal line Contact: Edorium Journals line Site Map
 
  Copyright © 2017. Edorium. All rights reserved.